a systematic review with narrative synthesis

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Narrative’ synthesis’ refers to an approach to the systematic review and synthesis of findings from multiple studies that relies primarily on the use of words and text to summarise and explain the findings of the synthesis. Whilst narrative synthesis can involve the manipulation of statistical data, the defining characteristic is that it adopts a textual approach to the process of synthesis to ‘tell the story’ of the findings from the included studies. As used here ‘narrative synthesis’ refers to a process of synthesis that can be used in systematic reviews focusing on a wide range of questions, not only those relating to the effectiveness of a particular intervention. (Popay et al. 2006)

A systematic literature review and narrative synthesis on the risks of medical discharge letters for patients’ safety

Christine Maria Schwarz 1 ,
Magdalena Hoffmann ORCID: orcid.org/0000-0003-1668-4294 1 , 2 ,
Petra Schwarz 3 ,
Lars-Peter Kamolz 1 ,
Gernot Brunner 1 &
Gerald Sendlhofer 1 , 2

BMC Health Services Research volume 19 , Article number: 158 ( 2019 ) Cite this article

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The medical discharge letter is an important communication tool between hospitals and other healthcare providers. Despite its high status, it often does not meet the desired requirements in everyday clinical practice. Occurring risks create barriers for patients and doctors. This present review summarizes risks of the medical discharge letter.

The research question was answered with a systematic literature research and results were summarized narratively. A literature search in the databases PubMed and Cochrane Library for Studies between January 2008 and May 2018 was performed. Two authors reviewed the full texts of potentially relevant studies to determine eligibility for inclusion. Literature on possible risks associated with the medical discharge letter was discussed.

In total, 29 studies were included in this review. The major identified risk factors are the delayed sending of the discharge letter to doctors for further treatments, unintelligible (not patient-centered) medical discharge letters, low quality of the discharge letter, and lack of information as well as absence of training in writing medical discharge letters during medical education.

Conclusions

Multiple risks factors are associated with the medical discharge letter. There is a need for further research to improve the quality of the medical discharge letter to minimize risks and increase patients’ safety.

Peer Review reports

The medical discharge letter is an important communication medium between hospitals and general practitioners (GPs) and an important legal document for any queries from insurance carriers, health insurance companies, and lawyers [ 1 ]. Furthermore, the medical discharge letter is an important document for the patient itself.

A timely transmission of the letter, a clear documentation of findings, an adequate assessment of the disease as well as understandable recommendations for follow-up care are essential aspects of the medical discharge letter [ 2 ]. Despite this importance, medical discharge letters are often insufficient in content and form [ 3 ]. It is also remarkable that writing of medical discharge letters is often not a particular subject in the medical education [ 4 ]. Nevertheless, the medical discharge letter is an important medical document as it contains a summary of the patient’s hospital admission, diagnosis and therapy, information on the patient’s medical history, medication, as well as recommendations for continuity of treatment. A rapid transmission of essential findings and recommendations for further treatment is of great interest to the patient (as well as relatives and other persons that are involved in the patients’ caring) and their current and future physicians. In most acute care hospitals, patients receive a preliminary medical discharge letter (short discharge letter) with diagnoses and treatment recommendations on the day of discharge [ 5 ]. Unfortunately, though, the full hospital medical discharge letter, which is often received with great delay, is an area of constant conflict between GPs and hospital doctors [ 1 ]. Thus the medical discharge letter does not only represent a feature of process and outcome quality of a clinic, but also influences confidence building and binding of resident physicians to the hospital [ 6 ].

Beside the transmission of patients’ findings from physician to physician, the delivery of essential information to the patient is an underestimated purpose of the medical discharge letter [ 7 ]. The medical discharge letter is often characterized by a complex medical language that is often not understood by the patients. In recent years, patient-centered/patient-directed medical discharge letters are more in discussion [ 8 ]. Thus, the medical discharge letter points out risks for patients and physicians while simultaneously creating barriers between them.

A systematic review of the literature was undertaken to identify patient safety risks associated with the medical discharge letter.

Search strategy

A systematic literature search was conducted using the electronic databases PubMed and Cochrane Database. Additionally, we scanned the reference lists of selected articles (snowballing). The following search terms were used: “discharge summary AND risks”, “discharge summary AND risks AND patient safety” and “discharge letter AND risks” and “discharge letter AND risks AND patient safety”. We reviewed relevant titles and abstracts on English and German literature published between January 2008 and May 2018 and started the search at the beginning of February 2018 and finished it at the end of May 2018.

Eligibility criteria

In this systematic review, articles were included if the title and/or abstract indicated the report of results of original research studies using quantitative, qualitative, or mixed method approaches. Studies in paediatric settings or studies that do not handle possible risks of the medical discharge letter were excluded, as well as reports, commentaries and letters. Electronic citations, including available abstracts of all articles retrieved from the search, were screened by two authors to select reports for full-text review. Duplicates were removed from the initial search. Nevertheless, during the search of articles the selection, publication as well as language bias must be considered. Thereafter, full-texts of potentially relevant studies were reviewed to determine eligibility for inclusion. In the following Table 1 inclusion and exclusion criteria for the studies are listed. Afterwards, key outcomes and main results were summarized. Differences were resolved by consensus. Finally, a narrative synthesis of studies meeting the inclusion criteria was conducted. Reference management software MENDELEY (Version 1.19.3) was used to organise and store the literature.

Data extraction

The data extraction in form of a table was used to summarize study results. The two authors extracted the data relating to author, country, year, study design, and outcome measure as well as potential risk factors to patient safety directly into a pre-formatted data collection form. After data extraction, the literature was discussed and synthesized into themes. The evaluation of the single studies was done using checklists [STROBE (combined) and the Cochrane Data collection form for intervention reviews (RCTs and non-RCTs)]. Meta-analysis was not considered appropriate for this body of literature because of the wide variability of studies in relation to research design, study population, types of interventions and outcomes.

Then a narrative synthesis was performed to synthesize the findings of the different studies. Because of the range of very different studies that were included in this systematic review, we have decided that a narrative synthesis constitutes the best instrument to synthesise the findings of the studies. First, a preliminary synthesis was undertaken in form of a thematic analysis involving searching of studies, listing and presenting results in tabular form. Then the results were discussed again and structured into themes. Afterwards, summarizing of included studies in a narrative synthesis within a framework was performed by one author.

This framework consisted of the following factors: the individuals and the environment involved in the studies (doctors, hospitals), the tools and technology (such as discharge letter delivery systems), the content of the medical discharge letter (such as missing content, quality of content), the accuracy and timeliness of transfer. These themes were discussed in relation to potential risks for patient’s safety. All articles that were included in this review were published before. The framework of this study was chosen following a previously published systematic review dealing with patient risks associated with telecare [ 9 ].

The initial literature search in the two online databases identified 940 records. From these records, 65 full text articles were screened for eligibility. Then 36 full-text articles were excluded because they pertained to patient transfer within the hospital or to another hospital, or to patient hand-over situations. Finally, 29 studies were included in this review. Included studies are listed in Table 2 . All document types were searched with a focus on primary research studies. The results of the search strategy are shown in Fig. 1 .

Flow chart literature search strategy

From these 29 studies, 13 studies dealt with the quality analysis of discharge letters, 12 studies with delayed transmission of medical discharge letters and just as many with the lack of information in medical discharge letters. Only few studies dealt with training on writing medical discharge letters and with understanding of patients of their medical discharge letters. The descriptive information of the included articles is presented in Table 2 . Overall quality of the articles was found to be acceptable, with clearly stated research questions and appropriate used methods.

Risk factors

In the following the identified major risk factors concerning the medical discharge letter are presented in a narrative summary.

Delayed delivery

The medical discharge letters should arrive at the GP soon after hospital discharge to ensure the quickest possible further treatment [ 4 ]. If letters are delivered weeks after the hospital stay, a continuous treatment of the patient cannot be ensured. Furthermore, the author of the medical discharge letter will no longer have current data after the discharge of the patient, which may result in a loss of important information [ 10 ]. Interfaces between different treatment areas and organizational units are known to cause a loss of information and a lack of quality in patient handling [ 11 ]. The improvement of information transfer between different healthcare providers during the transition of patients has been recommended to improve patient care [ 12 , 13 ]. Delayed communication of findings may lead to a lack of continuity of care and suboptimal outcomes, as well as decreased satisfaction levels for both patients and GPs [ 14 , 15 , 16 ]. In a review of Kripalani et al., it was shown that 25% of discharge summaries were never received by GPs [ 17 ]. This has several negative consequences for patients. Li et al. [ 18 ] found that a delayed transmission or absence of the medical discharge summary is related to patient readmission, and a study by Gilmore-Bykovskyi [ 19 ] found a strong relationship between patients whose discharge summaries omitted designation of a responsible clinician/clinic for follow-up care and re-hospitalisation and/or death. A Swedish study by Carlsson et al. [ 20 ] points out that a lack of accuracy and continuity in discharge information on eating difficulties may increase risk of undernutrition and related complications. A study of Were et al. [ 18 ] investigated pending lab results in medical discharge summaries and found that only 16% of tests with pending results were mentioned in the discharge summaries, and Walz et al. [ 21 ] found that approximately one third of the sub-acute care patients had pending lab results at discharge, but only 11% of these were documented in the medical discharge summaries.

Quality, lack of information

Medical discharge letters are a key communication tool for patient safety issues [ 17 ]. Incomplete and insufficient medical discharge letters increase the risks of readmission and myriad other complications [ 22 ]. Langelaan et al. (2017) evaluated more than 2000 medical discharge letters and found that in about 60% of the letters essential information was missing, such as a change of the existing medication, laboratory data, and even data on the patients themselves [ 23 ]. Accurate and complete medical discharge summaries are essential for patient safety [ 17 , 24 , 25 ]. Addresses; patient data, including duration of stay; diagnoses; procedures; operations; epicrisis and therapy recommendations; as well as findings in the appendix; are minimum requirements that are supposed to be included in the medical discharge letter [ 4 ]. However, it was found that key components are often lacking in medical discharge letters, including information about follow-up and management plans [ 23 , 26 ], test results [ 27 , 28 , 29 ], and medication adjustments [ 30 , 31 , 32 , 33 , 34 , 35 ]. In a review of Wimsett et al. [ 36 ] key components of a high-quality medical discharge summary were identified in 32 studies. These important components were discharge diagnosis, the received treatment, results of investigations as well as follow-up plans.

Accuracy of patients’ medication information is important to ensure patient safety. Hospital doctors expect GPs to continue with the prescribed (or modified) drug therapy. However, the selection of certain drugs is not always transparent for the GPs. A study by Grimes et al. [ 30 ] found that a discrepancy in medication documentation at discharge occurred in 10.8% of patients. From these patients nearly 65.5% were affected by discrepancies in medication documentation. The most prevalent inconsistency was drug omission (20.9%). Only 2% of patients were contacted, although general patient harm was assessed. A Swedish study of 2009 [ 37 ] investigated the quality improvement of medical discharge summaries. A higher quality of discharge letter led to an average of 45% fewer medication errors per patient.

A recent study by Tong et al. [ 38 ] revealed a reduced rate of medication errors in medical discharge summaries that were completed by a hospital pharmacist. Hospital pharmacists play a key role in preparing the discharge medication information transferred to GPs upon patient discharge and should work closely with hospital doctors to ensure accurate medication information that is quickly communicated to GPs at transitions of care [ 39 ]. Most hospitals have introduced electronic systems to improve the discharge communication, and many studies found a significant overall improvement in electronic transfer systems due to better documentation of information about follow-up care, pending test results, and information provided to patients and relatives [ 40 , 41 , 42 ]. Mehta et al. [ 43 ] found that the changeover to a new electronic system resulted in an increased completeness of discharge summaries from 60.7 to 75.0% and significant improvements in levels of completeness in certain categories.

Writing of medical discharge letter is missing in medical education

Both junior doctors as well as medical students reported that they received inadequate guidance and training on how to write medical discharge summaries [ 44 , 45 ] and recognized that higher priority is often given to pressing clinical tasks [ 46 ]. Research into the causes of prescribing errors by junior doctors at hospitals in the UK has revealed that latent conditions like organizational processes, busy environments, and medical care for complex patients can lead to medication errors in the medical discharge summary [ 47 ].

Fortunately, some study results demonstrate that information and education on writing medical discharge letters would enhance communication to the GPs and prevent errors during the patient discharge process [ 37 ]. Minimal formal teaching about writing medical discharge summaries is common in most medical schools [ 39 , 46 ]; however, a study by Shivji et al. has shown that simple, intensive educational sessions can lead to an improvement in the writing process of medical discharge summaries and communication with primary care [ 48 ].

Since the medical discharge letter should meet specific quality criteria, senior physicians and/or the head physician correct(s) and validate(s) the letter. The medical discharge letter therefore represents an essential learning target [ 8 ]. Training activities and workshops are necessary for junior doctors to improve writing medical discharge letters [ 44 , 49 ]. It might be also useful for young doctors to use checklists or other structured procedures to improve writing [ 4 ]. Maher et al. showed that the use of a checklist enhanced the quality (content, structure, and clarity) of medical discharge letters written by medical students [ 50 ].

In the following Table 3 main risk factors of the medical discharge letter are summarized.

The results of this systematic literature research indicate notable risk factors relating to the medical discharge letter. In a study by Sendlhofer et al., 360 risks were identified in hospital settings [ 51 ]. From these, 176 risks were scored as strategic and clustered into “top risks”. Top risks included medication errors, information errors, and lack of communication, among others. During this review, these potential risk factors were also identified in terms of the medical discharge letter.

Delayed sending and low quality of medical discharge letters to the referring physicians, may adversely affect the further course of treatment. However, a study of Spencer et al. has determined rates of failures in processing actions requested in hospital discharge summaries in general practice. It was found that requested medication changes were not made in 17% and patient harm occurred in 8% in relation to failures [ 52 ].

Despite the existence of reliable standards [ 53 ] many physicians are not adequately trained for writing medical discharge letters during their studies. Regular trainings and workshops and standardized checklists may optimize the quality of the medical discharge letter. Furthermore, electronic discharge letters have the potential to easily and quickly extract important information such as diagnoses, medication, and test results into a structured discharge document, and offer important advantages such as reliability, speed of information transfer, and standardization of content. Comprehensive discharge letters reduce the readmission rate and increase safety and quality by discharging of the patient. A missing structure, as well as a complex language, illegible handwriting, and unknown abbreviations, make reading medical discharge letters more complicated [ 4 ]. At least, poor patient understanding of their diagnosis and treatment plans and incomprehensible recommendations can adversely impact clinical outcome following hospital discharge. Many studies confirm that inadequate communication of findings [ 3 , 39 , 54 ] is an important risk factor in patients’ safety [ 51 ].

Most medical information in the discharge letter is not understood by patients (as well as relatives and other persons that are involved in the patients’ caring) and patients themselves do not receive a comprehensible medical discharge letter. The content of the medical discharge letter is often useless for the patient due to its medical terminology and content that is not matching with the patient’s level of knowledge or health literacy [ 55 , 56 , 57 ]. Poor understanding of diagnoses and related discharge plans are common among patients and family members and often accompanied by unplanned hospital readmissions [ 58 , 59 , 60 , 61 ]. In a study by Lin et al., it was shown that a patient-directed discharge letter enhanced understanding for hospitalization and for recommendations. Furthermore, verbal communication of the letter contents, explanation of every section of the medical discharge letter, and the opportunity for discussion and asking questions improved patient comprehension [ 7 ]. A study by O’Leary et al. showed that roughly 80–95% of patients with breast tumours want to be informed and educated about their illness, treatment, and prognosis [ 62 ].

High quality of care is characterized by a patient-centered communication, where the patient’s personal needs are also in focus [ 63 ]. Translation of medical terms in reports and letters leads to a better understanding of the disease and, interestingly, the avoidance of medical terms did not lead to deterioration in the transmission of information between the treating physicians. Moreover, it was found that the minimisation of medical terminology in medical discharge letters improved understanding and perception of patients’ ability to manage chronic health conditions [ 64 ]. In effect, it is clear that patient-centered communication improves outcome, mental health, patient satisfaction and reduces the use of health services [ 65 ].

Strengths and limitations

We have identified key problems with the medical discharge summaries that negatively impact patients’ safety and wellbeing. However, there is a heterogeneous nature of the included studies in terms of study design, sample size, outcomes, and language. Only two reviewers screened the studies for eligibility and only full-text articles were included in the literature review; furthermore, only the databases Pubmed and Cochrane library were screened for appropriate studies. Due to these constraints, there is a chance that other relevant studies may have been missed.

High-quality medical discharge letters are essential to ensure patient safety. To address this, the current review identified the major risk factors as delayed sending and low quality of medical discharge letters, lack of information and patient understanding, and inadequate training in writing medical discharge letters. In future, research studies should focus on improving the communication of pending test results and findings at discharge, and on evaluating the impact that this improved communication has on patient outcomes. Moreover, a simple patient-centered medical discharge letter may improve the patient’s (as well as family members’ and other caregivers’) understanding of disease, treatment and post-discharge recommendations.

Abbreviations

General practitioner

Randomized Controlled Trial

STrengthening the Reporting of OBservational studies in Epidemiology

United Kingdom

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Published: 21 July 2020

A systematic review and narrative synthesis of data-driven studies in schizophrenia symptoms and cognitive deficits

Tesfa Dejenie Habtewold ORCID: orcid.org/0000-0003-4476-518X 1 , 2 ,
Lyan H. Rodijk 1 , 3 ,
Edith J. Liemburg 2 ,
Grigory Sidorenkov 1 ,
H. Marike Boezen 1 ,
Richard Bruggeman 2 , 4 &
Behrooz Z. Alizadeh ORCID: orcid.org/0000-0002-1415-8007 1 , 2

Translational Psychiatry volume 10 , Article number: 244 ( 2020 ) Cite this article

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To tackle the phenotypic heterogeneity of schizophrenia, data-driven methods are often applied to identify subtypes of its symptoms and cognitive deficits. However, a systematic review on this topic is lacking. The objective of this review was to summarize the evidence obtained from longitudinal and cross-sectional data-driven studies in positive and negative symptoms and cognitive deficits in patients with schizophrenia spectrum disorders, their unaffected siblings and healthy controls or individuals from general population. Additionally, we aimed to highlight methodological gaps across studies and point out future directions to optimize the translatability of evidence from data-driven studies. A systematic review was performed through searching PsycINFO, PubMed, PsycTESTS, PsycARTICLES, SCOPUS, EMBASE and Web of Science electronic databases. Both longitudinal and cross-sectional studies published from 2008 to 2019, which reported at least two statistically derived clusters or trajectories were included. Two reviewers independently screened and extracted the data. In this review, 53 studies (19 longitudinal and 34 cross-sectional) that conducted among 17,822 patients, 8729 unaffected siblings and 5520 controls or general population were included. Most longitudinal studies found four trajectories that characterized by stability, progressive deterioration, relapsing and progressive amelioration of symptoms and cognitive function. Cross-sectional studies commonly identified three clusters with low, intermediate (mixed) and high psychotic symptoms and cognitive profiles. Moreover, identified subgroups were predicted by numerous genetic, sociodemographic and clinical factors. Our findings indicate that schizophrenia symptoms and cognitive deficits are heterogeneous, although methodological limitations across studies are observed. Identified clusters and trajectories along with their predictors may be used to base the implementation of personalized treatment and develop a risk prediction model for high-risk individuals with prodromal symptoms.

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Introduction.

In psychiatry, phenotypic heterogeneity of disorders and their overlapping symptoms that may presumably share some fundamental biologic underpinnings is a major challenge for tailoring individualized therapies 1 . Similarly, the course and phenotypic expression of schizophrenia are variable 2 . Schizophrenia is a complex polygenic psychotic disorder with a lifetime morbidity risk of 0.7% 3 . The twin- and SNP-based heritability estimate of schizophrenia was 80% 4 and 30% 5 , respectively.

According to the diagnostic and statistical manual of mental disorders (DSM) criteria, the clinical manifestations of schizophrenia are positive (e.g., hallucinations, delusions and disorganized behaviour) and negative (e.g., emotional expressive deficit, social amotivation, social withdrawal and difficulty in experiencing pleasure) symptoms 6 . Cognitive deficit is also one of the hallmark manisfestations of schizophrenia that occur in 75–80% of patients and often associated with poor daily functioning and quality of life 7 . Cognitive impairment in schizophrenia can be selective or general though the most common deficits occur in executive function, processing speed, memory (e.g. episodic, verbal and working), attention, verbal fluency, problem-solving and social cognition 8 , 9 , 10 , 11 . Patients harbor a wide range of subjectively defined symptoms, which together yields instinctively heterogeneous groups of people who are collectively diagnosed with schizophrenia. Subclinical or prodromal symptoms are also evident in relatives of patients with schizophrenia and healthy general population 12 , 13 , 14 .

Despite a century of efforts, understanding the heterogeneity in the clinical presentation and course of schizophrenia has been unsuccessful. This can be due to the subjective measurement of its clinical symptoms, variation in response to treatment, lack of valid, stable, and meaningful sub-phenotyping methods, and molecular complexity with limited understanding of the pathophysiology 15 , 16 , 17 . Phenotypic heterogeneity can be related to several intrinsic and extrinsic factors and expressed in patients, time, and disease sub-phenotypes 16 , 18 . Identification of meaningful homogeneous subgroups of people based on their symptoms or endophenotypes (e.g. neuropsychological markers, neural substrates, and neurological soft signs) requires the use of both supervised and unsupervised analyses. Distinguishing heterogeneous patients to more behaviorally and biologically similar subgroups is expedient not only to unveil common etiologies but also to examine the patterns of clinical symptoms, understand the biology of disease, predict treatment response and develop a new targeted treatment that improves recovery and functional outcomes 15 , 16 , 19 , 20 .

For tackling heterogeneity, in the past decade, numerous efforts have been undertaken by carefully designing studies and developing statistical models implemented in various programming languages and software 16 . In 2013, the American Psychiatric Association also endorsed a dimensional approach to identify intermediate categories based on the subjective report of severity of symptoms 6 . As a result, researchers have been using latent class cluster analyses and growth mixture models to explore clusters of individuals and trajectories of clinical symptoms in various settings 15 , 21 , 22 . Statistical methods can be used to identify subgroups and describe within and between individual variations to guide clinicians and statisticians to explore the relationship of diseases with various clinical and functional outcomes, treatment response, and neuropathological change. Moreover, subtyping using imaging, biological and symptom data is a recognizable method and widely used in psychiatric research 21 .

Several reviews have been conducted on positive symptoms 23 , negative symptoms 24 , 25 , 26 and cognitive dysfunction 7 , 9 , 27 , 28 , 29 , 30 , 31 , 32 , 33 , 34 , 35 . However, these reviews have largely focused on the conventional approach for determining an average change in the course of symptoms over time and the difference between subjects (e.g., patient vs sibling, sibling vs control, or patient vs control) and diagnosis. Reviewed studies are also based on correlation analysis, which is believed not to be a strong measure of association between predictors and outcomes 36 . Besides, these primary studies vary in terms of study population and use of assessment tools, scoring and standardization techniques, and have several limitations, such as small sample size, short duration of follow-up and limited use of data from healthy siblings and/or controls 9 , 37 , 38 . Of interest, none of these reviews fully addressed evidence from both longitudinal and cross-sectional data-driven studies on schizophrenia symptoms and cognitive deficits among patients with schizophrenia spectrum disorders, relatives and healthy controls. Taken together, thus far, our understanding of the heterogeneity of the course of schizophrenia symptoms and cognitive deficits is still limited. In the present systematic review, we summarized the contemporary evidence from cross-sectional and longitudinal studies on positive and negative symptoms and cognitive deficits among patients with schizophrenia spectrum disorders, their unaffected siblings and healthy people. Additionally, we explored the extent and origin of heterogeneity across studies. We further highlighted common methodological gaps and point out future directions to optimize the translatability of evidence from data-driven studies within the outlook of a personalized approach.

Registration and reporting

This systematic review was conducted and reported based on a registered protocol 39 and the Preferred Reporting Items for Systematic Review and Meta-Analysis (PRISMA) statement (Supplementary File 1 ), respectively 40 , 41 . The screening and selection process of the reviewed articles are further illustrated using a PRISMA flow diagram.

Databases and search terms

A systematic search of PubMed, PsycINFO, PsycTESTS, PsycARTICLES, SCOPUS, EMBASE and Web of Science electronic databases was performed. A comprehensive search strategy was developed for PubMed and adapted for each database in consultation with a medical information specialist (Supplementary File 2 ). The following search terms were used in their singular or plural form in the title, abstract, keywords and text fields of the articles: “schizophrenia”, “psychosis”, “non-affective psychosis”, “cognitive deficit”, “cognitive dysfunction”, “cognitive alteration”, “negative symptoms”, “deficit syndrome”, “positive symptoms”, “psychopathology”, “cognit*”, “neuropsycholog*”, “neurocognition”, “longitudinal”, “follow-up”, “course”, “heterogeneity”, “endophenotype”, “profile”, “cluster analysis”, “siblings”, “healthy controls”, “latent class analyses”, “Symptom trajectories”, “traject*”, “group modelling” and “trajectory”. Cross-references of included articles and grey literature were also hand-searched. Furthermore, we searched the table of contents of the journals of Schizophrenia Research, Schizophrenia Bulletin, Acta Psychiatrica Scandinavica, BMC Psychiatry, American Journal of Psychiatry and British Journal of Psychiatry to explore relevant studies. The freezing date for the final search was August 2019. In this review, we use ‘trajectory’ for groups identified in longitudinal studies and “cluster” for groups identified in cross-sectional studies.

Inclusion and exclusion criteria

Studies which met the following criteria were included: (1) longitudinal and cross-sectional studies; (2) studies that reported at least two clusters or trajectory groups of individuals using a statistical method based on a distinct positive symptom, negative symptom, and cognitive deficit or a combination of these symptoms; (3) studies conducted in patients with schizophrenia spectrum disorders, unaffected relatives, or healthy individuals irrespective of their clinical (e.g. medication status, severity of illness) and sociodemographic characteristics; and (4) studies published in English from 2008 to 2020. The publication year was limited to the last decade to capture the latest available evidence, which is likely to provide statistically powerful estimates and successfully subtyping schizophrenia symptoms given the increased number of large cohorts. To maximize the number of searched articles, the follow-up period in longitudinal studies was not restricted. Longitudinal studies based on the analyses of the mean levels of change of symptom scores were excluded because they did not capture individuals’ patterns of change over time by treating between-subject variation as an error, so that the actual heterogeneity of groups cannot be revealed 42 . Also, studies based on the non-statistical methods of clustering (e.g. family-based clustering) were excluded. Review papers, commentaries, conference abstracts, duplicate studies, editorials, and qualitative studies were excluded as well. Furthermore, we excluded studies in which the trajectory groups or clusters were generated based on scores constructed using a combination of schizophrenia symptoms and other unspecified psychotic symptoms.

Data retrieval and synthesis

Studies retrieved from all databases were exported to RefWorks version 2.0 for Windows web-based citation manager, which followed by the removal of close and exact duplicates. All independent studies were exported to a Microsoft Excel spreadsheet to screen for further inclusion criteria. Authors T.D.H. and L.H.R. independently screened the titles and abstracts. The two reviewers had a substantial agreement (Kappa statistic ( κ ) = 0.62). Inconsistent decisions were discussed and solved with consensus. Finally, full-text was reviewed, and the following data were independently extracted by T.D.H. and L.H.R.: first author name, publication year, country, cohort/research center, study population, sample size, symptom dimension(s), assessment tool, study design, duration of follow-up for longitudinal studies, frequency of assessment, method of calculating composite score, method of clustering/trajectory analysis, number of identified clusters or trajectory groups and significant correlates of clusters and predictors of trajectories 43 . The corresponding author was contacted by email if the full-text of included article was not accessible. When studies did not report the cohort or research center, we extracted the institutional affiliation of the first or corresponding author.

Search results

In total, 2262 articles were identified through database searching and an additional 26 articles were obtained through manual searching of cross-references and tables of content of relevant journals. After removing duplicate and unrelated articles, the titles and abstracts of 1292 articles were screened. The evaluation of titles and abstracts resulted in the exclusion of 1231 articles. In total, 61 articles were selected for full-text review, and eight articles 44 , 45 , 46 , 47 , 48 , 49 , 50 , 51 were excluded due to unclear outcomes, mixed diagnosis of the study population and use of a non-statistical method of clustering or clustering based on different phenotypes of schizophrenia. Finally, data were extracted from 53 longitudinal and cross-sectional studies. The PRISMA flow diagram of screening and the selection process is shown in Fig. 1 .

PRISMA flow diagram illustrating the screening and selection of literature.

Overview of included studies

The included 53 studies were conducted globally in 30 countries and published over a decade from 2009 to 2020. Seventeen studies were conducted in the USA and few studies were internationally conducted. Of these, 19 studies were longitudinal that involved 11,684 patients, 1059 siblings and 2194 controls or general population from more than eight countries, whereas 34 studies were cross-sectional that involved 6138 patients, 7670 siblings, and 3326 controls from 14 countries. Most of the longitudinal studies examined trajectories of positive and negative symptoms in patients, whereas most of the cross-sectional studies explored cognitive subtypes in patients. Only one longitudinal study 52 and three cross-sectional studies 53 , 54 , 55 examined cognitive subtypes among siblings. Overall, two to six subtypes of positive and negative symptoms and cognitive deficits were identified.

Longitudinal studies

In total, 19 longitudinal studies were reviewed that included all population age groups with the duration of follow-up ranged from six weeks to 10 years. The sample size ranged from 138 to 1990 subjects. Even though all studies had a similar aim, they have used slightly different models of trajectory analysis and model selection criteria. Growth mixture modelling (GMM) 17 , 56 , 57 , latent class growth analysis (LCGA) 16 , 19 , 20 , 58 , 59 , 60 , 61 , mixed-mode latent class regression modelling 22 , 62 , 63 , group-based trajectory modelling (GBTM) 52 , 64 , 65 , 66 and Ward’s method 67 were reported data-driven methods. Akaike’s Information Criterion (AIC), Bayesian information criterion (BIC) (i.e., used in most studies), deviance information criterion (DIC), logged Bayes factor, sample size adjusted BIC (aBIC), bootstrap likelihood ratio test [BLRT], Gap statistic, Lo–Mendell–Rubin Likelihood Ratio Test (LMR-LRT) and entropy were reported model selection indices.

Most longitudinal studies, Table 1 , investigated the trajectory of positive, negative or both symptoms in patients whereas one study 68 explored the trajectory of schizotypy in a nonclinical population. Another study 57 examined the association between positive and negative symptom trajectories in patients. Moreover, three studies examined the long-term trajectories of cognitive impairment in patients, their unaffected siblings and healthy controls 16 , 52 , 66 . One study 52 investigated the association between patients’ and siblings’ cognitive trajectories as well. Overall, these studies characterized the general pattern of identified trajectories as progressive deterioration, relapsing, progressive amelioration and stable, and the detail results are presented per symptom domains as follows.

Positive symptoms

As presented in Table 1a , four studies 19 , 20 , 57 , 65 investigated the trajectory of positive symptoms in patients with first-episode schizophrenia spectrum disorders with no or prior antipsychotics treatment for less than three months. The duration of follow-up and frequency of assessment ranged from six weeks to 10 years and five to seven times, respectively. Two studies 19 , 65 have used the Scale for the Assessment of Positive Symptoms (SAPS) to assess positive symptoms and identified five trajectories with more than one-third of patients subtyped as decrease positive symptoms or good responders. The other two studies used the Positive and Negative Syndrome Scale (PANSS) tool to assess positive symptoms and identified three trajectories that most of them grouped to class two 57 and two trajectories being in the most of the cases early response and stable trajectory over time 20 . The identified predictors were male gender, low educational status, substance use, diagnosis with schizophrenia, long duration of untreated psychosis, poor global functioning, and severe baseline positive and negative symptoms (Fig. 2 ).

Schizophrenia spectrum circle illustrating the schizophrenia symptoms and cognitive deficits (innermost circle), sample groups (inner circle), identified trajectories (outer circle) and predictors (outermost circle) in longitudinal studies. Findings are read and interpreted based on the line up in the circle.

Negative symptoms

Eight longitudinal studies 19 , 20 , 57 , 58 , 61 , 64 , 65 , 67 explored negative symptom trajectories among patients with first-episode non-affective psychosis with no prior or minimal treatment up to three months (Table 1b ). Two studies 19 , 65 used the Scale for the Assessment of Negative Symptoms (SANS), four studies 20 , 57 , 61 , 64 used the PANSS scale and two studies used the High Royds Evaluation of Negativity Scale 58 and Clinical Global Impressions-Schizophrenia scale 67 to assess negative symptoms. The duration of follow-up and frequency of assessment ranged from 6 weeks to 10 years and three to 64 times, respectively. Five studies 19 , 20 , 57 , 61 , 64 identified four trajectories of negative symptoms with variable patterns, whereas one study 65 found five trajectories with approximately half of them had persistent symptoms or poor response to treatment. The other two studies 58 , 67 found three trajectories with most of the participants had minimally stable negative symptoms. Our review depicted that trajectories of negative symptoms were predicted by older age, male gender, low educational status, ethnic minority, being unmarried, family history of psychosis, long duration of untreated psychosis, poor premorbid adjustment, severe depressive and disorganized symptoms, diagnosis of schizophrenia, unemployment, poor functioning and quality of life, high antipsychotics dosage, low cognitive performance, and high level of baseline negative and positive symptoms (Fig. 2 ).

Positive and negative symptoms

Combining both positive and negative symptom dimensions, which is illustrated in Table 1c , eight studies 17 , 22 , 56 , 57 , 59 , 60 , 62 , 63 inspected trajectories in patients with first-episode or chronic schizophrenia with antipsychotics treatment for more than three months and all of these studies used the PANSS scale to measure positive and negative symptoms. The duration of follow-up and frequency of assessment ranged from three months to two years and five to ten times, respectively. Among these studies, four 22 , 56 , 60 , 62 of them revealed five trajectories, two 57 , 63 of them revealed three trajectories, one study 17 found four trajectories and another study 59 found two trajectories with substantial difference in the nature, pattern and distribution of trajectories. Symptom trajectories were predicted by older age, male gender, ethnic minority, increased weight, diagnosis with schizophrenia, late age of illness onset, depressive and extrapyramidal symptoms, general psychopathology, type of antipsychotics treatment (e.g., aripiprazole, olanzapine), exacerbation, long duration of illness, poor premorbid and cognitive functioning, low global functioning and quality of life, living situation, involuntary admission, previous hospitalization and severe baseline positive and negative symptoms (Fig. 2 ).

Cognitive deficits

As shown in Table 1d , three studies investigated the trajectories of global cognitive deficits in patients with first-episode psychosis patients, their siblings and healthy controls 52 , 66 , and clinically stable outpatients with schizophrenia (SCZ) together with healthy controls 16 . The first six-year longitudinal study 52 , which cognitive function was assessed by the cognitive battery test, depicted five trajectories of cognitive impairment in patients (i.e., most of them with mild to moderate deficits) and four trajectories in healthy siblings (i.e., most of them had normal cognitive function). The second study 66 , which was the follow-up of the previous study, found six cognitive trajectories (i.e., nearly half of the population had mild to severe cognitive impairment) by combining patients, siblings and controls. The third longitudinal study 16 have used the Mattis Dementia Rating Scale and reported three trajectories (i.e., half of them with high and stable trajectory) of global cognitive function by combining patients and controls. Two studies found that patients with poor cognitive trajectories had younger age, low educational status, non-Caucasian ethnicity, lived in a sheltered facility, low IQ, poor premorbid adjustment, severe positive and negative symptoms, and low baseline cognitive performance 16 , 52 . Likewise, siblings with poor cognitive trajectories had younger age, female gender, low educational status, non-Caucasian ethnicity, low IQ, poor premorbid adjustment, severe schizotypy, frequent positive psychotic experience, and low baseline cognitive performance (Fig. 2 ) 52 . One study discovered that polygenic risk score for schizophrenia significantly predicted poor long-term cognitive trajectory in combined samples of patients, siblings and controls 66 .

A single longitudinal study assessed schizotypy in healthy college students using the Chapman Psychosis Proneness Scales (CPPS) and found four trajectories, in which nearly three-fourths of students were categorized as non-schizotypal 68 . This study also found that male gender and a high level of baseline schizotypy significantly predicted trajectories (Table 1e , Fig. 2 ).

In summary, when we inspecting the longitudinal study’s findings shown in Table 1 , studies that found the same number of trajectories were substantially different concerning participants composition (patient, sibling and controls), assessment instruments, symptom dimensions, frequency of assessment, duration of follow-up, methods used to generate a composite score, data-driven methods applied, label, proportion, pattern and type of trajectories, and identified predictors. In addition, there was no link between the numbers and types of trajectories and the use of trajectory analysis methods, study population and symptom dimensions.

Cross-sectional studies

Of the 53 included studies, 34 studies were cross-sectional (Table 2 ) that conducted in different groups of population. The total sample size per study ranged from 62 to 8231 individuals irrespective of participants’ diagnostic status. The reported clustering methods were K-means or non-hierarchical clustering analysis 21 , 53 , 55 , 69 , 70 , 71 , 72 , 73 , 74 , 75 , 76 , Ward’s method or hierarchical analysis 77 , 78 , 79 , 80 , 81 , 82 , 83 , K-means clustering and Ward’s method 18 , 38 , 54 , 84 , 85 , 86 , 87 , 88 , 89 , latent class or profile analysis 15 , 90 , 91 and two-step cluster analysis 92 , 93 , 94 . One study 95 identified clusters using a combination of clinical/empirical and statistical clustering methods. The model selection criteria or similarity metrics were visual inspections of the dendrogram, Pearson correlation, squared Euclidean distance (i.e., the most common index), agglomeration coefficients, Dunn index, Silhouette width, Duda and Hart index, elbow test, variance explained, inverse scree plot, average proportion of non-overlap, AIC, BIC, aBIC, Schwarz’s BIC, Lo–Mendell–Rubin (LMR) test, adjusted LMR and BLRT.

Among the 34 studies (Table 2 ), 22 studies 18 , 38 , 53 , 54 , 70 , 71 , 75 , 76 , 78 , 79 , 80 , 81 , 82 , 84 , 86 , 87 , 88 , 90 , 91 , 92 , 93 , 94 , 95 reported cognitive clusters in patients with first-episode, stable or chronic schizophrenia with or without antipsychotics treatment and one study 54 reported cognitive clusters in unaffected siblings. Other studies investigated trajectories of negative symptoms 15 , 85 , positive symptoms 83 , positive and negative symptoms 21 , 69 , 77 in patients and positive and negative schizotypy in a nonclinical population 55 , 72 , 73 , 83 . Furthermore, two studies 75 , 90 investigate the data-driven clusters by combining cognitive deficit and negative symptoms. Details on clusters and correlates of clusters presented per symptom dimensions as follows.

Only one study 83 assessed hallucinatory experience in patients with schizophrenia using Launay–Slade Hallucination Scale-Revised (LSHS-R) and identified three clusters (Table 2a ) 83 . Given this was an explanatory study, correlates of clusters were not studied.

Two studies 15 , 85 reported three clusters of patients with (chronic)schizophrenia based on the negative symptoms that assessed by the SANS scale 85 and Schedule for the Deficit Syndrome (Table 2b ) 15 . Identified clusters were significantly correlated with male gender, ethnic minority, low educational status, summer season of birth, early age onset of illness, severity of positive and negative symptoms, poor cognitive performance, poor functioning, high level of general psychopathology, previous hospitalization, poor premorbid adjustment, social anhedonia and poor attitude (Fig. 3 ).

Schizophrenia spectrum circle illustrating the schizophrenia symptoms and cognitive deficits (innermost circle), sample groups (inner circle), identified clusters (outer circle) and correlates (outermost circle) in cross-sectional studies. Findings are read and interpreted based on the line up in the circle.

Two studies 21 , 77 assessed positive and negative symptoms in patients with childhood-onset or first-episode schizophrenia using the SAPS and SANS scales, respectively and found three clusters, while another study 69 used the PANSS scale and found four clusters (Table 2c ). Reported symptom clusters were characterized as low positive and negative symptoms, high positive and low negative, low positive and high negative, and high positive and high negative though the patterns and distributions of clusters were different across studies. Identified clusters were significantly correlated with male gender, low IQ, poor global functioning, poorer metacognitive ability, and high level of positive and negative symptoms (Fig. 3 ).

Of the 22 studies conducted on neurocognitive deficits, 17 studies 38 , 53 , 70 , 71 , 76 , 78 , 79 , 80 , 81 , 82 , 84 , 90 , 92 , 93 , 94 , 95 found three clusters, five studies 18 , 75 , 86 , 87 , 91 reported four clusters and one study 88 discovered five clusters among patients (Table 2d ). Most studies assessed global cognitive function using a comprehensive neuropsychological test that included three to 18 cognitive subtests. Poor cognitive function in patients was associated with age, gender, non-Caucasian ethnicity, low socioeconomic and educational status, poor premorbid adjustment, low premorbid and current IQ, early age of illness onset, long duration of illness, severe positive and negative symptoms, poor social cognition, high antipsychotics dosage, use of second-generation antipsychotics, and poor functioning and poor quality of life (Fig. 3 ). In siblings, one study 54 found three cognitive clusters in unaffected siblings that associated with young age, low educational status, low IQ, poor premorbid adjustment and severe positive schizotypy (Table 2d , Fig. 3 ) 54 . One study 92 found that polygenic score (PRS) for schizophrenia, cognition, educational attainment and attention deficit hyperactivity disorder (ADHD) correlated with cognitive clusters in patients and their unaffected siblings.

Negative symptoms and cognitive deficits

One study 89 found three clusters of (out)patients with stable schizophrenia spectrum disorder by combining social cognition that assessed by the Mayer-Salovey-Caruso Emotional Intelligence Test and negative symptoms that assessed by the PANSS scale, whereas another study 74 found four clusters in patients by combining neurocognition that assessed by Continuous Performance Tests and negative symptom that assessed by the PANSS scale (Table 2e ). Clusters were significantly correlated with being unmarried, poor self-esteem, low cognitive (attention, social) performance, stigma, severity of positive and negative symptoms, poor social functioning and quality of life, and previous hospitalization (Fig. 3 ).

Three studies investigated schizotypy in unaffected first-degree relatives of patients with schizophrenia 55 and healthy college students 72 , 73 using the CPPS scale and found four clusters, whereas another study 83 found two clusters based on hallucinatory experience that assessed by LSHS-R scale in healthy general population (Table 2f ). Schizotypy clusters were significantly associated with male gender, lack of pleasure experiences, difficulty of emotional expression, psychotic-like symptoms, severity of positive and negative schizotypy, depressive, schizoid and somatic symptoms, poor social and cognitive functioning, substance abuse and poor personality (Fig. 3 ).

To summarize, as we observed in longitudinal studies, cross-sectional studies that found the same number of clusters were conducted in a different group of samples and used various assessment instruments and methods of generating composite scores and clustering. The labeling, pattern, proportion, and type of clusters were remarkably different. Generally, three clusters were the most replicated number of clusters and characterized by low (severe deficits), mixed (intermediate deficits) and high (intact or normal performance) cognitive function. In addition, cognitive clustering, such as verbal fluency deficit, verbal memory and executive function deficit, face memory and processing deficits, or global cognitive deficits were revealed. Cross-sectional studies that found the same number of clusters were largely different in the characteristics of study population, pattern of identified clusters, symptom dimensions, methodology of assessment, applied data-driven methods and identified associated factors.

Overall, as shown in Table 3 , the reviewed studies reported two to six clusters or trajectories and 58 factors that linked with identified clusters and/or trajectories across all study participants and symptom dimensions. The most common associated factors were old age, male gender, non-Caucasian ethnicity, low educational status, late age of illness onset, diagnosis of schizophrenia, several general psychopathology and depressive symptoms, severe positive and negative symptoms, low cognitive performance, and poor premorbid functioning, quality of life and global functioning.

To our knowledge, this is the first comprehensive systematic review based on recent cross-sectional and longitudinal data-driven studies in positive and negative symptoms, and cognitive deficits in patients with schizophrenia spectrum disorders, their relatives and healthy people. Our review has three major findings. First, longitudinal trajectory-based studies found two to five positive and negative symptoms trajectories in patients and four to six cognitive trajectories in patients, siblings, controls, or combined samples. Second, cross-sectional cluster-based studies identified three positive and negative symptoms clusters among patients and four positive and negative schizotypy clusters among healthy siblings. Additionally, three to five cognitive clusters were discovered in patients and their unaffected relatives. Third, numerous sociodemographic, clinical and genetic factors that determine trajectories and/or clusters were identified.

We showed that longitudinal and cross-sectional studies in patients, their siblings and healthy general population have inconsistently identified two to five trajectories/clusters and various predictors across the schizophrenia symptoms and cognitive deficits. Several shortcomings across studies may cause this inconsistency. Previous longitudinal studies did not uniformly research symptoms and cognitive deficits. For example, only three studies 16 , 52 , 66 longitudinally investigated cognitive trajectories, but 22 cross-sectional studies investigated cognitive clusters. Utterly, none of the reviewed longitudinal and cross-sectional studies also validated their model using empirical methods or comparable statistical methods though they have used different complex data-driven methods. Accumulating evidence showed that the number of classes in the optimal model derived from one method can be remarkably different compared to the other method 96 . Given that these studies were conducted in patients at a different stage at diagnosis, disease course or severity of illness and treatment status, the results may not be expectedly consistent as well. For example, studies that included only first-episode psychosis, chronic or stable patients may identify smaller clusters than studies that included a mixture of patients with first-episode and chronic psychosis or patients with severe illness. Additionally, since the reported studies were conducted in more than 20 countries, the use of different treatment strategies and assessment methods in different countries could further confound the assessment of symptoms and clinical heterogeneity. Obviously, in patients who are treated, the observed symptoms and cognitive characteristics are the product of those features that were present before treatment and the response to treatment. Moreover, the different measurement tools may lead to discrepant results. For instance, the discrepancy of negative and positive symptoms trajectories (or cross-sectional clusters) might partly be attributable to the use of a specific negative (e.g. SANS) and positive (e.g. SAPS) symptom scale or a more general symptom scale (e.g. PANSS) that included items measuring cognitive or disorganization symptoms. Additionally, some studies administered up to 18 different neuropsychological tests to measure cognition while others have used as few as two or three cognitive assessment tests.

We further observed common methodological limitations across studies. Firstly, the reviewed studies included various groups of participants from different age groups and ethnicities. Secondly, while the comparison of patient clusters and trajectories with healthy siblings or controls could provide an accurate means of disentangling the heterogeneity and causes of heterogeneity of schizophrenia symptoms, only four studies (three were cross-sectional studies) examined clusters in siblings. Likewise, most studies used healthy controls to standardize patients neurocognitive composite scores, and a few other studies used controls to compare the distribution of patient clusters or trajectory groups. Thirdly, substantial differences between studies were also noted in constructing composite scores, use of model selection criteria and method of parameter estimation. Fourthly, we observed several ways of subtyping and nomenclature for clusters or trajectories, which may be difficult for clinicians to translate the evidence in diagnosing and treating diseases. This is due to the lack of a standard for designing a study (e.g. adequate sample size), reporting data analysis approaches and publishing results 42 .

Generally, we saw that studies conducted in patients with similar stages of illness (i.e, first-episode, stable, chronic stage or with or without treatment) and used similar assessment methods (i.e., SANS, SAPS or PANSS) showed some level of similarity in results with respect to identified trajectories and predictors, but studies are largely different in duration of follow-up, frequency of assessment and methods used to assess symptoms or cognition. By the same token, studies that used the similar data-driven statistical methods showed similarity in the number of identified trajectories/clusters, but largely different in study population, stage of illness, use of measurement tool, duration of follow-up, frequency of assessment and identified factors. Moreover, studies with duration of follow-up less than two years and above two years showed a similar level of heterogeneity in symptoms and cognitive deficits and identified predictors. In addition, a 10-year study with five times assessment showed similar findings with a 6 week study with every week assessment on positive symptoms. On the other hand, a 2-year study with five times assessment identified only two trajectories. Despite these facts, all studies interestingly showed heterogeneity of symptoms and cognitive deficits at various level with “four trajectories” is the most replicated in longitudinal studies and “three clusters” is the most replicated in cross-sectional studies. Besides, these studies consistently reported age, gender, ethnicity, educational status, age of illness onset, diagnosis, general psychopathology and depressive symptoms, positive and negative symptoms, cognitive performance, functioning and quality of life as determinant factors of trajectories and/or clusters.

In the era of team science and big data, the use of data-driven statistical methods is becoming increasingly popular for the analysis of longitudinal repeated measures (i.e., latent growth mixture models (LGMMs)) and cross-sectional (i.e., cluster analysis) data (Fig. 4 ). In our review, we observed that LGMMs, such as GMM, latent class growth analysis (LCGA), mixed mode latent class regression modelling and group-based trajectory modelling (GBTM) were commonly used data-driven methods in longitudinal studies. LGMMs can identify realistic categories based on temporal patterns of change in outcome by assuming the existence of latent classes or subgroups of subjects exhibiting similarity with regard to unobserved (latent) variables 19 , 97 . LGMMs have four advantages for modelling longitudinal data. First, they are flexible and data-driven methods that can accurately reveal actual heterogeneity. Second, they allow the classification of individual subjects into latent classes based on the largest probability of class membership. Third, they are sensitive to the pattern of change over time and robust in the presence of missing data. Fourth, subject-level predictors can be directly assessed for association with class membership and hence with different trajectory subtypes 16 , 19 , 97 . Cluster analysis, which is commonly used in cross-sectional studies, is also a data-driven approach for classifying people into homogeneous groups by determining clusters of participants that display less within-cluster variation relative to the between-cluster variation 81 . Among the reviewed cross-sectional studies, K-means and Ward’s method clustering analyses were commonly used alone or in combination. K-means cluster analysis is a non-hierarchical form of cluster analysis appropriate when previous evidence or hypotheses exist regarding the number of clusters in a sample 74 . On the other hand, Ward’s method is a hierarchical cluster analysis aiming to determine group assignment without prior hypothesis 74 . It is believed, K-means cluster analysis can handle larger data sets compared with Ward’s method 73 .

A hypothetical model for driving big multidimensional data towards a personalized selection of treatments in schizophrenia spectrum disorders. GBTM: Group-based trajectory modeling; LCGA: Latent class growth analysis; CBT: Cognitive behavioural therapy.

The results of statistical subtyping approaches, such as cluster or trajectory analysis depend on mathematical assumptions, type of data, number of variables or tests, sample size and sampling characteristics. Therefore, the models can be unstable and parameter estimates of clinical symptoms may not converge to a consistent set of subgroups and lack a direct relationship to clinical reality 59 , 87 , 98 . For example, intermediate clusters and trajectories substantially vary between studies that used the same cluster or trajectory analysis method 87 . We advocate that study results from data-driven methods should be applicable, comparable, generalizable and interpretable into clinical practice. As a result, we recommend to validate models using at least one additional comparable statistical methods, combine statistical methods of subtyping with empirical/clinical methods, or work together with clinicians to create a common understanding and clinically relevant clustering or trajectories nomenclatures. Furthermore, it is relevant to replicate clusters or trajectory groups using independent samples, different assessment tools that measure the same construct, or different linkage methods 38 , 99 . Finally, further studies are required that focus on longitudinal study design, unaffected siblings, genetic markers and more detailed measures of brain network function for improving our understanding of the biological mechanism underlying heterogeneity of schizophrenia.

Future clinical advances may benefit from the subgrouping of patients to implement tailored therapy. In our review, we observed that several longitudinal studies were conducted based on drug response. One study found individuals who treated with aripiprazole had delayed response 56 , whereas another study found olanzapine treated patients had good response 63 . Another study also revealed individuals receiving standard treatment, compared to assertive treatment, showed delayed negative symptom trajectory 19 . Furthermore, individuals with substantial cognitive deficit received high dose of antipsychotics 18 , 82 , 87 . Subtyping of symptoms and cognitive deficits can also contribute to uncover the biological basis of individual symptoms, rather than studying constellation of co-occurring symptoms 1 . The identified factors associated with clusters and/or trajectories could be used for developing a clinical risk prediction model for high-risk individuals with prodromal symptoms 100 , 101 .

Thus far, findings from this review showed that data-driven approaches could have substantial role to optimize the efficacy of personalized care by predicting individual susceptibility to disease, providing accurate assessments of disease course, contribute to best-choice of early intervention, and selecting treatments (e.g., antipsychotics, cognitive behavioral therapy, social skill training, family therapy) targeting subgroups of patients with similar phenotypic or psychosocial characteristics (Fig. 4 ) 102 . When data-driven methods are implemented on samples/cohorts following different pharmacological and non-pharmacological interventions, then, we believe that our proposed model (Fig. 4 ) can identify individuals who successfully treated, not treated or even harmed and who needs further intervention and close follow-up to protect from unnecessary cost and side effect of medication(s). Therefore, findings from our review could assist in the implementation of personalized and preventive strategies for clinical practice at least in national or regional level.

Conclusions

Our review indicated a significant heterogeneity in results and conclusions obtained from both cross-sectional and longitudinal studies in terms of the number of group membership for positive and negative symptoms and cognition as well as factors (predictors) associated with the group membership. This review also identified several methodological issues contributing to the discrepant results. Generally, the longitudinal studies identified trajectories characterized by progressive deterioration, relapsing, progressive amelioration and stability, whereas low, mixed (intermediate) and high psychotic symptoms and cognitive clusters were identified by cross-sectional studies. Future studies can be more benefited from data-driven methods if applied based on pharmacological and non-pharmacological treatment responses. The use of empirical methods to distinguish more homogeneous subgroups of patients along heterogeneous symptom dimensions has gained traction in the last several years and it is an essential step toward implementation of a more precise prediction of disease risk and individualized selection of interventions.

Data availability

All relevant data were included in the paper.

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Acknowledgements

We would like to forward our special gratitude to Sjoukje van der Werf, who is a medical information specialist at the University of Groningen, the Netherlands, for her support to develop the search strings and guiding the overall literature retrieval process. Tesfa Dejenie was supported by the Scholarship of University of Groningen, Groningen, the Netherlands. L.H. Rodijk was supported by the Junior Scientific Master Class of the University of Groningen, Groningen, the Netherlands.

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T.D.H. and B.Z.A. conceived and designed the study. T.D.H. and L.H.R. did study selection and data extraction. R.B., B.Z.A. and H.M.B. led the primary work during systematic review and narrative synthesis. T.D.H. and B.Z.A. drafted the paper. L.H.R., G.S. and E.J.L. provide intellectual comments during the revision process of the paper. All co-authors contributed to the review and synthesis of the findings, and reviewed and approved the final paper.

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Correspondence to Tesfa Dejenie Habtewold or Behrooz Z. Alizadeh .

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Habtewold, T.D., Rodijk, L.H., Liemburg, E.J. et al. A systematic review and narrative synthesis of data-driven studies in schizophrenia symptoms and cognitive deficits. Transl Psychiatry 10 , 244 (2020). https://doi.org/10.1038/s41398-020-00919-x

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Research Article

A systematic review and narrative synthesis of the research provisions under the Mental Capacity Act (2005) in England and Wales: Recruitment of adults with capacity and communication difficulties

Roles Data curation, Formal analysis, Methodology, Writing – original draft, Writing – review & editing

* E-mail: [email protected]

Affiliation School of Health Sciences, University of East Anglia, Norwich Research Park, Norwich, United Kingdom

Roles Data curation, Methodology, Writing – review & editing

Affiliation Norwich Medical School, University of East Anglia, Norwich Research Park, Norwich, United Kingdom

Roles Conceptualization, Methodology, Writing – review & editing

Roles Methodology, Writing – review & editing

Affiliation Centre of Research Excellence in Aphasia Recovery and Rehabilitation, La Trobe University, Melbourne Victoria, Australia

Affiliation Centre for Educational Development, Appraisal and Research, New Education Building, Westwood Campus, University of Warwick, Coventry, United Kingdom

Affiliation School of Law, University of East Anglia, Norwich Research Park, Norwich, United Kingdom

Roles Conceptualization, Data curation, Funding acquisition, Methodology, Project administration, Writing – review & editing

Oluseyi Florence Jimoh,
Hayley Ryan,
Anne Killett,
Ciara Shiggins,
Peter E. Langdon,
Rob Heywood,
Karen Bunning

Published: September 1, 2021
https://doi.org/10.1371/journal.pone.0256697
Peer Review
Reader Comments

The Mental Capacity Act (MCA, 2005) and its accompanying Code of Practice (2007), govern research participation for adults with capacity and communication difficulties in England and Wales. We conducted a systematic review and narrative synthesis to investigate the application of these provisions from 2007 to 2019.

Methods and findings

We included studies with mental capacity in their criteria, involving participants aged 16 years and above, with capacity-affecting conditions and conducted in England and Wales after the implementation of the MCA. Clinical trials of medicines were excluded. We searched seven databases: Academic Search Complete, ASSIA, MEDLINE, CINAHL, PsycArticles, PsycINFO and Science Direct. We used narrative synthesis to report our results. Our review follows Preferred Reporting Items for Systematic Reviews and is registered on PROSPERO, CRD42020195652.

28 studies of various research designs met our eligibility criteria: 14 (50.0%) were quantitative, 12 (42.9%) qualitative and 2 (7.1%) mixed methods. Included participants were adults with intellectual disabilities (n = 12), dementia (n = 9), mental health disorders (n = 2), autism (n = 3) and aphasia after stroke (n = 2). We found no studies involving adults with acquired brain injury. Diverse strategies were used in the recruitment of adults with capacity and communication difficulties with seven studies excluding individuals deemed to lack capacity.

Conclusions

We found relatively few studies including adults with capacity and communication difficulties with existing regulations interpreted variably. Limited use of consultees and exclusions on the basis of capacity and communication difficulties indicate that this group continue to be under-represented in research. If health and social interventions are to be effective for this population, they need to be included in primary research. The use of strategic adaptations and accommodations during the recruitment process, may serve to support their inclusion.

Citation: Jimoh OF, Ryan H, Killett A, Shiggins C, Langdon PE, Heywood R, et al. (2021) A systematic review and narrative synthesis of the research provisions under the Mental Capacity Act (2005) in England and Wales: Recruitment of adults with capacity and communication difficulties. PLoS ONE 16(9): e0256697. https://doi.org/10.1371/journal.pone.0256697

Editor: Muhammad Shahzad Aslam, Xiamen University - Malaysia Campus: Xiamen University - Malaysia, MALAYSIA

Received: May 9, 2021; Accepted: July 13, 2021; Published: September 1, 2021

Copyright: © 2021 Jimoh et al. This is an open access article distributed under the terms of the Creative Commons Attribution License , which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.

Data Availability: All relevant data are within the manuscript and its Supporting information files.

Funding: KB OSAP/43239 NUFFIELD Foundation https://www.nuffieldfoundation.org/ The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.

Competing interests: The authors have declared that no competing interests exist.

Introduction

Informed consent is a requirement of intrusive research [ 1 ], which upholds the principles of autonomous decision-making with provisions for the protection of those who lack capacity [ 2 , 3 ]. It requires that the person can understand and retain relevant information, weigh up the implications of participation, and communicate a decision [ 4 – 6 ]. However, our society also includes people who lack mental capacity and people with communication difficulties, either as separate impairments or in combination, referred to in this review as adults with capacity and communication difficulties (CCDs). The number of people affected by such difficulties is rising and include people with dementia [ 7 ], stroke [ 8 ], acquired brain injury [ 9 ], mental health difficulties [ 10 ], autism and intellectual disabilities [ 11 , 12 ]. In the context of a rising prevalence of people living CCD, there is a need for research to advance our understanding of these conditions and to improve evidence-based interventions. However, research shows that people living with CCDs continue to be under-represented in research [ 13 , 14 ].

In England and Wales, the Mental Capacity Act (MCA) (2005) [ 2 ] and its accompanying Code of Practice (CoP) [ 2 , 15 ] were originally introduced to protect the rights of adults who may lack capacity for autonomous decision-making in relation to treatment, welfare and finance. There are separate provisions for research (CoP: Chapter 11). Different legislation is provided in other countries of the UK: the Adults with Incapacity (Scotland) Act 2000 (AWIA); the Mental Capacity Act (Northern Ireland) (2016). In Ireland, it is the Assisted Decision Making (Capacity) Act 2015. However, the current review pertains to the Mental Capacity Act (2005) in England and Wales. The MCA applies to ‘intrusive’ research, which refers to research that would require consent if it were conducted on persons with mental capacity [ 2 ]. It does not apply to clinical trials of medicines which is governed by different legislation (The Medicines for Human Use Clinical Trials Regulations) [ 16 ].

For the purposes of research, there is the presumption of capacity unless there is a reason to believe that a person lacks capacity (CoP 2007). Before deciding that someone lacks capacity, the CoP (2007) recommends the provision of relevant information, communicated in the most appropriate way [ 15 ]. Whilst practical details are not given, there is general encouragement for presenting project information to suit the processing capabilities of potential participants. For example, support for the person’s understanding of what research participation entails might include: information sheets rendered in simple language with or without pictorial support; a simulated data collection procedure shown on video; questions and answer opportunities in conversations about a project; and use of manual sign and gesture to augment meanings [ 15 , 17 – 19 ]. Relevance theory [ 20 ] argues that people find it easier to engage with and understand information that is most relevant to them and requires the least cognitive effort. The form of the message interacts with the person’s cognitive abilities, prior experience and underlying knowledge. On this latter point, the person’s familiarity with the subject matter contributes to their perception of possible cognitive gain, which in turn optimises the potential relevance of information to them [ 20 ]. This asserts the importance of addressing the information-processing needs of the target population for successful recruitment to studies, particularly where CCDs are present.

Notwithstanding the presumption of capacity [CoP: 11.4; MCA S.1(2)], an assessment of an individual’s capacity is a requirement [ 2 , 15 ] when concerns are raised about capacity. For this purpose, a two-stage test is recommended [MCA S.3; CoP 4:10]. There is no one standard method for the purpose, with many researchers using locally-developed initiatives [ 21 , 22 ]. Capacity is defined as time and decision-specific, variable according to complexity of information [ 23 ], and possibly fluctuating over time [ 24 ]. The distinction between capacity and lack of capacity is far from straightforward [ 24 , 25 ]. Furthermore, the presence of communication and cognitive impairments may complicate the informed consent process [ 26 – 30 ] by masking true competence in people with, for example, early stage dementia, moderate intellectual disability [ 31 , 32 ], aphasia following stroke [ 33 , 34 ] and autistic spectrum disorder [ 35 ]. To circumnavigate some of these difficulties, researchers have developed person-centred approaches [ 24 ] characterised by flexibility and support from family and friends [ 36 ].

A proven lack of capacity requires the advice of a consultee, either personal (e.g. relatives, friends, unpaid carer) or nominated (e.g. healthcare professionals) [ 36 , 37 ], about the individual’s likely wishes and feelings concerning research participation (CoP: 11.20) [ 2 ]. In the context of a consultee’s affirmative advice, researchers are required to prioritise the interest of the participant above that of science and the society (CoP: 11.20; CoP 11.29), considering their wishes and feelings throughout the research process (CoP 11.29) [ 15 ]. In such cases, expressions of: assent (a person’s ‘permission or affirmative agreement to something) [ 38 ]; and dissent (a person’s disagreement or refusal), are recognised appropriately [ 36 ]. This aligns with the principle of partial participation [ 39 ], which acknowledges that gradations of involvement are possible. Gatekeepers such as residential home managers, carers and health professionals, are uniquely placed to facilitate access to those with CCD because of an existing relationship with the person [ 40 ]. Thus, the individual’s participation in research is not only dependent on autonomous decision-making or consultee advice, but upon overcoming additional barriers such as permission from gatekeepers.

There has been limited consideration of intrusive research under the MCA [ 41 , 42 ]. Previous reviews have focused on MCA provisions in relation to health and social care practice [ 22 , 43 ] and clinical trials of medicines, which is governed by different legislation (The Medicines for Human Use Clinical Trials Regulations [ 16 ]. Provisions for intrusive research under the MCA have been criticised for a lack of clarity leading to variable interpretations [ 21 , 44 , 45 ]. Considering these challenges, the aim of this systematic review was to develop an understanding of how adults with CDD have been included and accommodated within research studies within England and Wales following the implementation of the MCA, 2005.

This systematic review of the literature was carried out following PRISMA guidance [ 46 ]. The review protocol (See S1 File ) was prospectively registered in Prospero with Registration number CRD42020195652 [ 47 ]. In the protocol, we used the term “adults with impairments of capacity and/or communication (ICC)”. This has been refined and modified through our interactions with our stakeholders to “adults with capacity and communication difficulties”.

Search strategy and eligibility criteria

We included studies conducted in England and/or Wales from 2007 (the year the Mental Capacity Act 2005, was implemented; CoP: DfCA, 2007) to 2019. The search framework focused on adults with CCD and the MCA (2005). Multiple terms, representative of the primary stakeholder groups (i.e., autism; aphasia; dementia; head injury (OR brain injury); learning disability (OR intellectual disability), were used in combination with (AND) mental capacity (OR) informed consent and applied to the following databases: Academic Search Complete, ASSIA, MEDLINE, CINAHL, PsycArticles, PsycINFO and Science Direct. The initial search strategy was developed in MEDLINE and adjusted according to the indexing systems of other databases (See S2 File ). The first search was carried out on 11 th December 2019 and an updated search on 13 th July 2020, to identity any additional papers.

Study selection

Search results were combined into a single Endnote file, citations were screened, and duplicates removed in accordance with the PRISMA statement [ 46 ]. Two researchers (FJ and HR) then independently screened all titles to identify relevant studies according to the eligibility criteria ( Table 1 ). Then, abstracts were reviewed to identify studies to undergo full-text review. Disagreements were resolved by discussion between the two researchers. We did not search grey literature sources but supplemented searches with backwards and forward searches of the references listed in the included studies.

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Data extraction and quality assessment

The review set out to identify, describe and synthesise the procedures and accommodations used by researchers to support the inclusion and participation of adults with impairments of capacity and communication in research. The data extraction table was therefore designed to capture this information and is presented in the supplementary material ( S1 Table ). Two researchers (FJ and HR) extracted data independently using a Microsoft Excel-based broad extraction sheet, which detailed: population-type by diagnosis, inclusion/exclusion criteria, sample size, sampling method, information format, capacity assessment procedure, informed consent procedure, research accommodations, consultee involvement, use of gatekeepers and the year of study. Data were summarised and a third researcher KB reviewed and confirmed the data extraction.

The Mixed Methods Appraisal Tool (MMAT) [ 49 ], for concurrent critical appraisal of quantitative, qualitative and mixed-methods primary research was applied [ 50 ]. The MMAT has established content validity, it has been piloted across all methodologies; quantitative, qualitative and mixed methods research designs [ 50 , 51 ]. Compared with other tools, the MMAT includes specific criteria for appraising mixed methods studies. While critical appraisal tools are more widely available for quantitative and qualitative research, there has not been consensus on quality criteria for mixed methods research [ 52 ].

The tool results in a methodological rating of between one and five (with five being the highest quality), for each study, based on the evaluation of study selection bias, study design, data collection methods, sample size, intervention integrity, and analysis. An overall quality score and a descriptive summary was derived for each study [ 49 ]. A score of 4–5 indicated a ‘high quality’; 3 indicated ‘moderate’; 2 or less indicated ‘low quality’. For mixed-method studies, each methodological element was assessed separately, and the lowest quality score included. A second researcher (KB) independently checked the reliability of the quality assessment on a random sample of studies (17%) [ 53 ], with perfect agreement (k = 1.0) [ 54 ]. As the review is exploratory, no study was excluded based on quality assessment since they may still provide valuable insight [ 53 ].

Data analysis

To account for methodological diversity and sample variability, we employed narrative synthesis in the report of results [ 55 , 56 ]. Using a textual approach, a descriptive summary of the included studies focused on the recorded fields in the broad extraction sheet and the relationships within and between the studies examined.

Search results

Search results are summarised in the Preferred Reporting Items for Systematic Reviews and Meta-analyses (PRISMA) flowchart ( Fig 1 and S2 Table ) [ 46 ].

https://doi.org/10.1371/journal.pone.0256697.g001

Our initial search identified 2116 studies and a repeat search identified a further 614 studies. Following removal of duplicates, screening and full textual review of 126 studies, of which 20 met the inclusion criteria. A further 8 studies were identified after reference and citation searches.

Characteristics of included studies

The key characteristics of the included studies are presented in supplementary S3 Table . Included participants were said to have intellectual disabilities (n = 12; 42.9%); dementia (n = 9; 32.1%); autism spectrum disorders (n = 3; 10.7%); mental health disorders (n = 2; 7.1%); and aphasia after stroke (n = 2; 7.1%). None were said to have brain injury. Study designs included quantitative (n = 14; 50.0%); qualitative (n = 12; 42.9%) and mixed methods (n = 2; 7.1%). Samples were drawn mainly from hospital in-patients or attending outpatient services (n = 13; 46.4%). Others were in receipt of social care services, prisoners, or part of national databases or ongoing studies (n = 15; 53.6%).

Quality assessment scores

Of the fourteen quantitative studies, twelve (85.7%) were evaluated as high-quality, one (7.1%) as moderate-quality and one (7.1%) as low-quality; all qualitative studies (n = 12, 100%), were evaluated as high-quality and both mixed-methods studies (n = 2, 100%) were evaluated as moderate quality.

All the studies articulated clear research questions and appropriate method to address such questions. Quantitative studies benefitted from the clear description of target population, use of validated tools and the use of sensitivity analysis and/or adjustments to reduce bias. However, some quantitative studies were weakened by the lack of sample size calculations and the recruitment of only those who had capacity or could speak English language (a potential source of bias). The strength of qualitative studies was based on appropriate methodology, use of triangulation methods, substantiating data with quotes and coherence between data and its interpretation. The quantitative aspect of the two mixed-method studies lacked rigour and clarity. See S4 Table for full details of the quality assessment of each included paper and S5 Table for synopsis of study quality appraisal.

Identification of participants

In all included studies, participant access was managed through designated gatekeepers, who identified potentially eligible participants. Where specified, the role was variously enacted by clinical practitioners [ 57 – 64 ], other healthcare professionals [ 65 – 72 ], care home managers and staff [ 73 , 74 ], prison staff [ 75 ] or support staff [ 76 ]. In one study, Hall [ 74 ], following a period of acclimatisation in the home, the researcher performed the role of gatekeeper alongside staff and relatives in a residential home for people with dementia.

Inclusion/Exclusion criteria of participants

Participants deemed to lack capacity were included in 15 studies (54%) based on consultee advice [ 57 – 63 , 68 , 72 , 77 – 82 ], and excluded from seven studies as part of eligibility criteria (25%) [ 66 , 67 , 69 , 70 , 75 , 76 , 83 ]. In one of the studies, potential participants judged not able to consent were not even approached [ 83 ]. Of the remaining 6 studies, one made provision for consultee advice but did not use this as all participants were able to give informed consent [ 84 ], while the participants in the remaining five studies were able to give informed consent [ 64 , 65 , 71 , 73 , 85 ]. In addition, three studies excluded potential participants based on cognitive-communicative competence for data collection methods [ 73 , 83 , 84 ], and severe visual and cognitive difficulties [ 78 ]. Furthermore, limitations in English as a second language affected exclusions in 3 studies [ 68 , 70 , 75 ]. The role of personal consultee was fulfilled variously by family members, friends, next of kin, or a close person who knew the participant well [ 57 , 58 , 61 , 63 , 64 , 68 , 72 , 74 , 82 , 86 ] while nominated consultees were either paid carers or healthcare professionals [ 59 , 60 , 77 , 81 ]. Several studies reported checks for verbal and non-verbal signs indicating participant willingness or unwillingness to participate in the research [ 57 , 58 , 67 , 68 , 72 – 75 , 78 , 82 ].

Study information format

A lack of detail concerning the format of study information was evident in 12 studies (42.9%) [ 57 , 58 , 60 – 63 , 66 , 73 , 77 , 79 , 82 , 83 ]. Where detail was provided, the preferred format was text, often combined with verbal explanations [ 70 , 72 , 80 , 84 , 85 , 87 ]. Wray [ 76 ], reported the use of verbal explanation only for those living with aphasia. Eight studies reported adaptations to the participant information sheet in support of communication needs: an ‘aphasia friendly’ format for people with aphasia post-stroke [ 78 ]; ‘easy read’ versions for people with intellectual disabilities [ 59 , 65 , 71 ] and ASD/ID [ 75 ]; and ‘accessible’ information for people with intellectual disabilities [ 67 ] and dementia [ 68 , 69 ]. One study [ 59 ] used graphic images to supplement text. Collaborative development of information sheets by researchers and user group representatives was reported by two studies [ 68 , 88 ] and affected volume of essential information presented [ 81 ] and format accessibility [ 68 ].

Further support for decision making process

Supplementary decision-making processes included communicative support from familiar others (e.g. family members, carers, and healthcare professionals) [ 59 ]; allowing extra time for participants to process information [ 65 , 75 ]; and providing question and answer opportunities [ 58 , 64 , 68 , 78 , 84 ]. Consideration of setting factors for recruitment activities were also reported: familiar places to minimise any anxiety affecting understanding [ 58 ]; and private places to control for distraction [ 75 ]. Some studies used a range of information formats and approaches to recruitment. For example, Stoner [ 69 ] used a full information sheet, abbreviated, and accessible formats for those living with dementia. While Frighi [ 59 ], used a variety of pictures, or ‘easy read’ materials supplemented by support from familiar others.

Capacity assessment procedures

Capacity assessment procedures were not reported in detail in many studies. However, authors of 7 studies [ 57 – 60 , 63 – 65 , 75 ] referred to the MCA functional test (MCA 2005), albeit with variously described procedures. Formal assessments were reported for three studies with variable use of closed questions [ 86 ]; a checklist of items [ 65 , 75 ]; and standardised questions [ 85 ]. Spencer [ 88 ], used the MacArthur Competence Assessment Tool for Clinical Research (MacCAT-CR) with people with mental health disorders. It is a semi-structured tool that measures decision-making competence in terms of understanding, appreciation, reasoning and expressing a choice [ 89 ]. Informal capacity assessments, appeared to be based on conversations between researcher and prospective participants [ 72 ] or on ethnographic observations of the individual’s verbal and behavioural responses [ 67 , 74 ] in some studies. Although researchers’ judged capacity in most studies, this decision was initially taken by clinicians [ 60 , 61 , 63 – 65 , 76 , 82 , 90 ] or other gatekeepers such as care home managers of staff [ 73 , 74 ] or both [ 66 , 74 ]. Individuals deemed to lack capacity were often excluded from research participation without report of a formal assessment [ 66 , 67 , 69 – 71 , 75 , 76 ].

Informed consent procedures

Written informed consent was obtained from participants who had capacity to take part in research [ 57 – 61 , 64 , 65 , 69 , 70 , 72 , 75 – 77 , 80 , 82 , 85 ]. Four studies involving adults with dementia [ 68 , 72 , 80 ], and intellectual disabilities [ 67 ] reported adaptation to the consent process by the use of an enhanced process consent model that monitored ongoing consent through verbal and non-verbal signs, thereby supporting participant autonomy [ 68 , 80 ]. In each case, the researcher maintained a documented ‘audit trail’ of decisions and actions informed by the gatekeepers and consultees, and the communicative behaviours of participants, as did Hall [ 80 ]. Goldsmith [ 67 ] assessed consent in adults with intellectual disabilities, by meeting the potential participant with a supporter in attendance and capturing the process on video to document non-verbal cues. This was then checked by the supporter for non-verbal cues to either confirm or deny capacity and a decision that is free from coercion. In addition, one group recruited from a population case register using an ‘opt-out consent procedure’ and made contact with prospective participants by phone or an ‘opt-in consent procedure’ where participants contacted the study team directly [ 79 ]. A single study [ 76 ] used the Consent Support Tool with adults with aphasia post-stroke to determine the requirements for support and the recommended communication strategies.

Our systematic review revealed variable interpretation of the provisions of the MCA (2005) and its accompanying guidance in the CoP. Capacity was included as part of the eligibility criteria within studies, sometimes as an exclusion criterion. Assessment of capacity is reported inconsistently with some studies adopting formal measures and others making it part of the informed consent procedure. Procedures used for informed and autonomous decision-making appeared to uphold the four defining principles of capacity. Our findings showed that researchers made efforts to maximise individual autonomy through use of various media and tools to support informed consent processes. Beyond seeking a consultee’s advice around the inclusion of incapacitous participants, there is limited report of measures to engage such participants in ongoing decisions about participation in research.

The gatekeeper is attributed a pivotal role in gaining access to participants [ 15 , 40 ]. Thus, there is the authority to facilitate or impede recruitment. Furthermore, it is possible that the inclusion of adults with CCD is affected by the gatekeeper’s own interpretation of mental capacity for decision-making. Communication difficulties in people post-stroke and memory problems in people with dementia may be mistaken for a lack of capacity by gatekeepers [ 91 ]. In one study [ 68 ], where all the participants were able to give informed consent, it was asked whether staff acting as gatekeepers avoided those individuals with dementia who had more complex communication needs. This raises questions about the gatekeeper’s own agenda and whether support for decision-making gives way to protection. The process whereby gatekeepers decide who to nominate as potential participants lacks clear specification, and may be seen as counter to the MCA [ 2 ] requirement for establishing capacity.

A range of strategies were used by researchers to support the accessibility of research information for those with CCD. This is consistent with relevance theory [ 20 ], as understanding of research information will be based on the cognitive load of each strategy. The use of accessible information with participants with intellectual disabilities showed compliance with the MCA’s second statutory requirement [ 2 , 15 ], reinforced by the Department of Health [ 18 ] and the Accessible Information Standards (AIS) [ 17 ]. Previous studies have shown that ‘aphasia-friendly’ study information was preferred by the aphasic participants [ 92 ] and led to 11.2% increase in their understanding [ 93 ]. This resonates the underlying premise of relevance theory that successful engagement with information requires the least cognitive load [ 20 ]. Beyond the use of multiple media to convey information, the support of familiar others and adjusting to individual needs is important [ 15 ]. Whilst there was limited report of tailored approaches to supporting CCD, a role for experts-by-experience was exemplified in one study [ 81 ], where researcher collaboration with patient group representatives informed the development of study information suitable for those with psychoses. Suitably selected images can support understanding [ 15 ]. However, the use of pictures may not be amenable to all participants and interpreted as patronising or misleading [ 86 , 92 ].

Careful consideration and further research are needed to ascertain the best strategies for each group of adults with capacity and communication difficulties.

Recruitment procedures targeting individuals with CCD need to include deliberate measures to achieve the easiest cognitive load possible within the required research framework [ 20 ]. Researchers need to be cognisant of the range of strategies and accommodations that can be used to support autonomous decision-making by engaging with the evidence on augmentation and alternative communication methods [ 19 ]. This includes the use of picture, simple text, object of reference and supported conversation [ 17 ]. In addition, consideration should be given to the individual need of each participant, tailoring accommodations to their preferred way of engaging with researchers [ 15 ].

The MCA (2005) recognises people’s interest in making decisions as much as possible [ 2 ]. An established lack of capacity does not obviate the need to provide opportunities for the participant to express their wishes and feelings. Baumgart proposed the principle of partial participation for individuals with severe developmental disabilities [ 39 ]. The concept embraces the notion of active engagement and advocates ‘interdependence’ such that individualised adaptations may serve to scaffold participation in ongoing decision-making as far as possible [ 39 ].

The lack of detailed description of the MCA’s two-staged assessment of capacity process in our findings may be a matter of reporting rather than reality. The use of both formal and informal methods of assessment allowed the inclusion of a range of adults with CCD in research. However, this type of capacity assessment is reported to be less reliable compared with structured assessment in clinical settings [ 94 ]. In contrast, our findings showed that ethnographic observations contribute to improved understanding of verbal and non-verbal behaviour and enhance capacity assessment [ 72 , 74 ]. While there is no ‘gold standard’ method for accessing capacity, the use of an assessment tool was documented in one study [ 88 ]. Previous research suggests that the MacCAT-CR tool is adaptable and reliable in those living with dementia and mental health difficulties [ 89 ]. There is need for the development and validation of capacity assessment tools in different groups of adults with CCD.

We found that adults who had difficulty communicating and those who were not able to consent to research participation were excluded from research potentially relevant to them. A parallel can be drawn with the clinical trials literature, where similar vulnerable groups were also excluded and therefore remain under-represented in research [ 95 , 96 ]. While eligibility criteria are useful for recruiting participants representative of a target population, exclusions solely based on lack of capacity, without appropriate assessments or adaptations in place are potentially unethical. It is possible that the added demands of consultee procedures and the perceived risks of participation for incapacitous individuals had a negative effect on sample inclusion [ 95 ]. This is contrary to Article 12 of the Convention on the Rights of Persons with Disabilities (CRPD: UN 2006) [ 97 ] which asserts there should be ‘equal recognition before the law’. Their exclusion may skew research sampling and has implications for service provision and policies.

Our findings provide evidence that adults with capacity and communication difficulties can take part in ethically sound research. Adaptations and accommodations are used variously to support both the assessment of capacity and the decision-making process in recruitment of participants, but exclusions still continue on the basis of a lack of capacity.

For the researcher, this means engaging with participants, as well as the gatekeepers and familiar others in their lives who are possible sources of information and support to them. Traditional ways of obtaining informed consent are not appropriate for all, and there is a need to consider the non-traditional ways such as process model of consent. Capacity is relative to a spectrum of decisions. Exercise of capacity can be supported, and its assessment is context- and time-specific. While consultees can facilitate participation in research for those lacking capacity, autonomy through partial participation is possible and to be encouraged. Thus, including people with capacity and communication difficulties in ethically-sound research requires a deliberate approach to devising ways of assessing true capacity and presenting study information.

Limitations

A possible limitation is that we missed some relevant studies because we excluded publications prior to 2011 in keeping with our focus on the implementation of the MCA. By limiting publication language to only English, we might have missed out on research findings reported in Welsh, the other official language apart from English in Wales. Our search did not yield any study involving adults with acquired brain injury, we have therefore not reported on this population.

Including adults with CCD in ethically-sound research is a complex proposition demanding deliberate planning of procedures to support autonomous decision-making as far as possible. Furthermore, the complexities of inclusion may cause researchers to err on the side of caution and exclude those deemed/presumed to be incapacitous. There is a need to further investigate the reasoning underpinning researchers’ decisions about sample inclusion and the development of research protocols and procedures for participant recruitment. Similarity in the provisions made for those living with dementia, intellectual disability and aphasia implies some common ground for future developments ( S1 Fig and S6 Table . Including CCD in research). The use of these strategies may enable researchers to navigate better the recruitment and inclusion adults with CCD in research.

Supporting information

S1 fig. including ccd in research..

https://doi.org/10.1371/journal.pone.0256697.s001

S1 Table. Data extraction table.

Showing characteristics and findings of the 28 included papers.

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S2 Table. PRISMA checklist.

Showing the page numbers on which Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) are reported.

https://doi.org/10.1371/journal.pone.0256697.s003

S3 Table. Summary of the characteristics of included studies with focus on study outcomes.

https://doi.org/10.1371/journal.pone.0256697.s004

S4 Table. Quality appraisal of studies using the Mixed Methods Appraisal Tool (MMAT).

https://doi.org/10.1371/journal.pone.0256697.s005

S5 Table. Summary table of study synopses (MMAT).

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S6 Table. Solutions to CCD recruitment.

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S7 Table. Excluded studies.

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S1 File. PROSPERO protocol.

Review protocol registered with PROSPERO (International prospective register of systematic reviews).

https://doi.org/10.1371/journal.pone.0256697.s009

S2 File. Search strategy.

https://doi.org/10.1371/journal.pone.0256697.s010

Acknowledgments

We would like to thank Marcus Redley for his input into the PROSPERO protocol.

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Hospice care access inequalities: a systematic review and narrative synthesis

Affiliations.

1 School of Clinical Medicine, University of Cambridge, Cambridge, Cambridgeshire, UK.
2 Primary Care Unit, Department of Public Health and Primary Care, University of Cambridge, Cambridge, Cambridgeshire, UK.
3 Medical Library, School of Clinical Medicine, University of Cambridge, Cambridge, Cambridgeshire, UK.
4 Primary Care Unit, Department of Public Health and Primary Care, University of Cambridge, Cambridge, Cambridgeshire, UK [email protected].
PMID: 33608254
PMCID: PMC9125370
DOI: 10.1136/bmjspcare-2020-002719

Background: Inequalities in access to hospice care is a source of considerable concern; white, middle-class, middle-aged patients with cancer have traditionally been over-represented in hospice populations.

Objective: To identify from the literature the demographic characteristics of those who access hospice care more often, focusing on: diagnosis, age, gender, marital status, ethnicity, geography and socioeconomic status.

Design: Systematic literature review and narrative synthesis.

Method: Searches of Medline, PsycINFO, CINAHL, Web of Science, Assia and Embase databases from January 1987 to end September 2019 were conducted. Inclusion criteria were peer-reviewed studies of adult patients in the UK, Australia, New Zealand and Canada, receiving inpatient, day, outpatient and community hospice care. Of the 45 937 titles retrieved, 130 met the inclusion criteria. Narrative synthesis of extracted data was conducted.

Results: An extensive literature search demonstrates persistent inequalities in hospice care provision: patients without cancer, the oldest old, ethnic minorities and those living in rural or deprived areas are under-represented in hospice populations. The effect of gender and marital status is inconsistent. There is a limited literature concerning hospice service access for the LGBTQ+ community, homeless people and those living with HIV/AIDS, diabetes and cystic fibrosis.

Conclusion: Barriers of prognostic uncertainty, institutional cultures, particular needs of certain groups and lack of public awareness of hospice services remain substantial challenges to the hospice movement in ensuring equitable access for all.

Keywords: chronic conditions; cultural issues; hospice care; service evaluation.

Publication types

Systematic Review
Aged, 80 and over
Hospice Care*
Middle Aged

Systematic Review
Open access
Published: 30 May 2024

Patient experiences: a qualitative systematic review of chemotherapy adherence

Amineh Rashidi 1 ,
Susma Thapa 1 ,
Wasana Sandamali Kahawaththa Palliya Guruge 1 &
Shubhpreet Kaur 1

BMC Cancer volume 24 , Article number: 658 ( 2024 ) Cite this article

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Adherence to chemotherapy treatment is recognized as a crucial health concern, especially in managing cancer patients. Chemotherapy presents challenges for patients, as it can lead to potential side effects that may adversely affect their mobility and overall function. Patients may sometimes neglect to communicate these side effects to health professionals, which can impact treatment management and leave their unresolved needs unaddressed. However, there is limited understanding of how patients’ experiences contribute to improving adherence to chemotherapy treatment and the provision of appropriate support. Therefore, gaining insights into patients’ experiences is crucial for enhancing the accompaniment and support provided during chemotherapy.

This review synthesizes qualitative literature on chemotherapy adherence within the context of patients’ experiences. Data were collected from Medline, Web of Science, CINAHL, PsychINFO, Embase, Scopus, and the Cochrane Library, systematically searched from 2006 to 2023. Keywords and MeSH terms were utilized to identify relevant research published in English. Thirteen articles were included in this review. Five key themes were synthesized from the findings, including positive outlook, receiving support, side effects, concerns about efficacy, and unmet information needs. The review underscores the importance for healthcare providers, particularly nurses, to focus on providing comprehensive information about chemotherapy treatment to patients. Adopting recommended strategies may assist patients in clinical practice settings in enhancing adherence to chemotherapy treatment and improving health outcomes for individuals living with cancer.

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Introduction

Cancer can affect anyone and is recognized as a chronic disease characterized by abnormal cell multiplication in the body [ 1 ]. While cancer is prevalent worldwide, approximately 70% of cancer-related deaths occur in low- to middle-income nations [ 1 ]. Disparities in cancer outcomes are primarily attributed to variations in the accessibility of comprehensive diagnosis and treatment among countries [ 1 , 2 ]. Cancer treatment comes in various forms; however, chemotherapy is the most widely used approach [ 3 ]. Patients undergoing chemotherapy experience both disease-related and treatment-related adverse effects, significantly impacting their quality of life [ 4 ]. Despite these challenges, many cancer patients adhere to treatment in the hope of survival [ 5 ]. However, some studies have shown that concerns about treatment efficacy may hinder treatment adherence [ 6 ]. Adherence is defined as “the extent to which a person’s behaviour aligns with the recommendations of healthcare providers“ [ 7 ]. Additionally, treatment adherence is influenced by the information provided by healthcare professionals following a cancer diagnosis [ 8 ]. Patient experiences suggest that the decision to adhere to treatment is often influenced by personal factors, with family support playing a crucial role [ 8 ]. Furthermore, providing adequate information about chemotherapy, including its benefits and consequences, can help individuals living with cancer gain a better understanding of the advantages associated with adhering to chemotherapy treatment [ 9 ].

Recognizing the importance of adhering to chemotherapy treatment and understanding the impact of individual experiences of chemotherapy adherence would aid in identifying determinants of adherence and non-adherence that are modifiable through effective interventions [ 10 ]. Recently, systematic reviews have focused on experiences and adherence in breast cancer [ 11 ], self-management of chemotherapy in cancer patients [ 12 ], and the influence of medication side effects on adherence [ 13 ]. However, these reviews were narrow in scope, and to date, no review has integrated the findings of qualitative studies designed to explore both positive and negative experiences regarding chemotherapy treatment adherence. This review aims to synthesize the qualitative literature on chemotherapy adherence within the context of patients’ experiences.

This review was conducted in accordance with the Joanna Briggs Institute [ 14 ] guidelines for systemic review involving meta-aggregation. This review was registered in PROSPERO (CRD42021270459).

Search methods

The searches for peer reviewed publications in English from January 2006-September 2023 were conducted by using keywords, medical subject headings (MeSH) terms and Boolean operators ‘AND’ and ‘OR’, which are presented in the table in Appendix 1 . The searches were performed in a systematic manner in core databases such including Embase, Medline, PsycINFO, CINAHL, Web of Science, Cochrane Library, Scopus and the Joanna Briggs Institute (JBI). The search strategy was developed from keywords and medical subject headings (MeSH) terms. Librarian’s support and advice were sought in forming of the search strategies.

Study selection and inclusion criteria

The systematic search was conducted on each database and all articles were exported to Endnote and duplicates records were removed. Then, title and abstract of the full text was screened by two independent reviewers against the inclusion criteria. For this review, populations were patients aged 18 and over with cancer, the phenomenon of interest was experiences on chemotherapy adherence and context was considered as hospitals, communities, rehabilitation centres, outpatient clinics, and residential aged care. All peer-reviewed qualitative study design were also considered for inclusion. Studies included in this review were classified as primary research, published in English since 2006, some intervention implemented to improve adherence to treatment. This review excluded any studies that related to with cancer and mental health condition, animal studies and grey literature.

Quality appraisal and data extraction

The JBI Qualitative Assessment and Review Instrument for qualitative studies was used to assess the methodological quality of the included studies, which was conducted by the primary and second reviewers independently. There was no disagreement between the reviews. The qualitative data on objectives, study population, context, study methods, and the phenomena of interest and findings form the included studies were extracted.

Data synthesis

The meta-aggregation approach was used to combine the results with similar meaning. The primary and secondary reviewers created categories based on the meanings and concept. These categories were supported by direct quotations from participants. The findings were assess based on three levels of evidence, including unequivocal, credible, and unsupported [ 15 , 16 ]. Findings with no quotation were not considered for synthesis in this review. The categories and findings were also discussed by the third and fourth reviewers until a consensus was reached. The review was approved by the Edith Cowan University Human Research Ethics Committee (2021–02896).

Study inclusion

A total of 4145 records were identified through a systematic search. Duplicates ( n = 647) were excluded. Two independent reviewers conducted screening process. The remaining articles ( n = 3498) were examined for title and abstract screening. Then, the full text screening conducted, yielded 13 articles to be included in the final synthesis see Appendix 2 .

Methodological quality of included studies

All included qualitative studies scored between 7 and 9, which is displayed in Appendix 3 . The congruity between the research methodology and the research question or objectives, followed by applying appropriate data collection and data analysis were observed in all included studies. Only one study [ 17 ] indicated the researcher’s statement regarding cultural or theoretical perspectives. Three studies [ 18 , 19 , 20 ] identified the influence of the researcher on the research and vice-versa.

Characteristics of included studies

Most of studies conducted semi-structured and in-depth interviews, one study used narrative stories [ 19 ], one study used focus group discussion [ 21 ], and one study combined focus group and interview [ 22 ] to collect data. All studies conducted outpatient’s clinic, community, or hospital settings [ 17 , 18 , 19 , 20 , 21 , 22 , 23 , 24 , 25 , 26 , 27 , 28 , 29 ]. The study characteristics presented in Appendix 4 .

Review findings

Eighteen findings were extracted and synthesised into five categories: positive outlook, support, side effects, concern about efficacy and unmet information needs.

Positive outlook

Five studies discussed the link between positivity and hope and chemotherapy adherence [ 19 , 20 , 23 , 27 , 28 ]. Five studies commented that feeling positive and avoid the negativity and worry could encourage people to adhere in their mindset chemotherapy: “ I think the main thing for me was just keeping a positive attitude and not worrying, not letting myself worry about it ” [ 20 ]. Participants also considered the positive thoughts as a coping mechanism, that would help them to adhere and complete chemotherapy: “ I’m just real positive on how everything is going. I’m confident in the chemo, and I’m hoping to get out of her soon ” [ 23 ]. Viewing chemotherapy as part of their treatment regimen and having awareness of negative consequences of non-adherence to chemotherapy encouraged them to adhere chemotherapy: “ If I do not take medicine, I do not think I will be able to live ” [ 28 ]. Adhering chemotherapy was described as a survivor tool which helped people to control cancer-related symptoms: “ it is what is going to restore me. If it wasn’t this treatment, maybe I wasn’t here talking to you. So, I have to focus in what he is going to give me, life !” [ 27 ]. Similarly, people accepted the medical facts and prevent their life from worsening; “ without the treatment, it goes the wrong way. It is hard, but I have accepted it from the beginning, yes. This is how it is. I cannot do anything about it. Just have to accept it ” [ 19 ].

Finding from six studies contributed to this category [ 20 , 21 , 23 , 24 , 25 , 29 ]. Providing support from families and friends most important to the people. Receiving support from family members enhanced a sense responsibility towards their families, as they believed to survive for their family even if suffered: “ yes, I just thought that if something comes back again and I say no, then I have to look my family and friends in the eye and say I could have prevented it, perhaps. Now, if something comes back again, I can say I did everything I could. Cancer is bad enough without someone saying: It’s your own fault!!” [ 29 ]. Also, emotional support from family was described as important in helping and meeting their needs, and through facilitation helped people to adhere chemotherapy: “ people who genuinely mean the support that they’re giving […] just the pure joy on my daughter’s face for helping me. she was there day and night for me if I needed it, and that I think is the main thing not to have someone begrudgingly looking after you ” [ 20 ]. Another study discussed the role family, friends and social media as the best source of support during their treatment to adhere and continue “ I have tons of friends on Facebook, believe it or not, and it’s amazing how many people are supportive in that way, you know, just sending get-well wishes. I can’t imagine going through this like 10 years ago whenever stuff like that wasn’t around ” [ 23 ]. Receiving support from social workers was particularly helpful during chemotherapy in encouraging adherence to the chemotherapy: “ the social worker told me that love is courage. That was a huge encouragement, and I began to encourage myself ” [ 25 ].

Side effects

Findings from five studies informed this category [ 17 , 21 , 22 , 25 , 26 ]. Physical side effects were described by some as the most unpleasure experience: “ the side effects were very uncomfortable. I felt pain, fatigue, nausea, and dizziness that limited my daily activities. Sometimes, I was thinking about not keeping to my chemotherapy schedule due to those side effect ” [ 17 ]. The impact of side effects affected peoples’ ability to maintain their independence and self-care: “ I couldn’t walk because I didn’t have the energy, but I wouldn’t have dared to go out because the diarrhoea was so bad. Sometimes I couldn’t even get to the toilet; that’s very embarrassing because you feel like you’re a baby ” [ 26 ]. Some perceived that this resulted in being unable to perform independently: “ I was incredibly weak and then you still have to do things and you can’t manage it ” [ 22 ]. These side effect also decreased their quality of life “ I felt nauseated whenever I smelled food. I simply had no appetite when food was placed in front of me. I lost my sense of taste. Food had no taste anymore ” [ 25 ]. Although, the side effects impacted on patients´ leisure and free-time activities, they continued to undertake treatment: “ I had to give up doing the things I liked the most, such as going for walks or going to the beach. Routines, daily life in general were affected ” [ 21 ].

Concern about efficacy

Findings form four studies informed this category [ 17 , 18 , 24 , 28 ]. Although being concerned about the efficacy of the chemotherapy and whether or not chemotherapy treatment would be successful, one participant who undertook treatment described: “the efficacy is not so great. It is said to expect about 10% improvement, but I assume that it declines over time ” [ 28 ]. People were worried that such treatment could not cure their cancer and that their body suffered more due to the disease: “ I was really worried about my treatment effectiveness, and I will die shortly ” [ 17 ]. There were doubts expressed about remaining the cancer in the body after chemotherapy: “ there’s always sort of hidden worries in there that whilst they’re not actually taking the tumour away, then you’re wondering whether it’s getting bigger or what’s happening to it, whether it’s spreading or whatever, you know ” [ 24 ]. Uncertainty around the outcome of such treatment, or whether recovering from cancer or not was described as: “it makes you feel confused. You don’t know whether you are going to get better or else whether the illness is going to drag along further” [ 18 ].

Unmet information needs

Five studies contributed to this category [ 17 , 21 , 22 , 23 , 26 ]. The need for adequate information to assimilate information and provide more clarity when discussing complex information were described. Providing information from clinicians was described as minimal: “they explain everything to you and show you the statistics, then you’re supposed to take it all on-board. You could probably go a little bit slower with the different kinds of chemo and grappling with these statistics” [ 26 ]. People also used the internet search to gain information about their cancer or treatments, “I’ve done it (consult google), but I stopped right away because there’s so much information and you don’t know whether it’s true or not ” [ 21 ]. The need to receive from their clinicians to obtain clearer information was described as” I look a lot of stuff up online because it is not explained to me by the team here at the hospital ” [ 23 ]. Feeling overwhelmed with the volume of information could inhibit people to gain a better understanding of chemotherapy treatment and its relevant information: “ you don’t absorb everything that’s being said and an awful lot of information is given to you ” [ 22 ]. People stated that the need to know more information about their cancer, as they were never dared to ask from their clinicians: “ I am a low educated person and come from a rural area; I just follow the doctor’s advice for my health, and I do not dare to ask anything” [ 17 ].

The purpose of this review was to explore patient’s experiences about the chemotherapy adherence. After finalizing the searches, thirteen papers were included in this review that met the inclusion criteria.

The findings of the present review suggest that social support is a crucial element in people’s positive experiences of adhering to chemotherapy. Such support can lead to positive outcomes by providing consistent and timely assistance from family members or healthcare professionals, who play vital roles in maintaining chemotherapy adherence [ 30 ]. Consistent with our study, previous research has highlighted the significant role of family members in offering emotional and physical support, which helps individuals cope better with chemotherapy treatment [ 31 , 32 ]. However, while receiving support from family members reinforces individuals’ sense of responsibility in managing their treatment and their family, it also instils a desire to survive cancer and undergo chemotherapy. One study found that assuming self-responsibility empowers patients undergoing chemotherapy, as they feel a sense of control over their therapy and are less dependent on family members or healthcare professionals [ 33 ]. A qualitative systematic review reported that support from family members enables patients to become more proactive and effective in adhering to their treatment plan [ 34 ]. This review highlights the importance of maintaining a positive outlook and rational beliefs as essential components of chemotherapy adherence. Positive thinking helps individuals recognize their role in chemotherapy treatment and cope more effectively with their illness by accepting it as part of their treatment regimen and viewing it as a tool for survival. This finding is supported by previous studies indicating that positivity and positive affirmations play critical roles in helping individuals adapt to their reality and construct attitudes conducive to chemotherapy adherence [ 35 , 36 ]. Similarly, maintaining a positive mindset can foster more favourable thoughts regarding chemotherapy adherence, ultimately enhancing adherence and overall well-being [ 37 ].

This review identified side effects as a significant negative aspect of the chemotherapy experience, with individuals expressing concerns about how these side effects affected their ability to perform personal self-care tasks and maintain independent living in their daily lives. Previous studies have shown that participants with a history of chemotherapy drug side effects were less likely to adhere to their treatment regimen due to worsening symptoms, which increased the burden of medication side effects [ 38 , 39 ]. For instance, cancer patients who experienced minimal side effects from chemotherapy were at least 3.5 times more likely to adhere to their treatment plan compared to those who experienced side effects [ 40 ]. Despite experiencing side effects, patients were generally willing to accept and adhere to their treatment program, although one study in this review indicated that side effects made some patients unable to maintain treatment adherence. Side effects also decreased quality of life and imposed restrictions on lifestyle, as seen in another study where adverse effects limited individuals in fulfilling daily commitments and returning to normal levels of functioning [ 41 ]. Additionally, unmet needs regarding information on patients’ needs and expectations were common. Healthcare professionals were considered the most important source of information, followed by consultation with the internet. Providing information from healthcare professionals, particularly nurses, can support patients effectively and reinforce treatment adherence [ 42 , 43 ]. Chemotherapy patients often preferred to base their decisions on the recommendations of their care providers and required adequate information retention. Related studies have highlighted that unmet needs among cancer patients are known factors associated with chemotherapy adherence, emphasizing the importance of providing precise information and delivering it by healthcare professionals to improve adherence [ 44 , 45 ]. Doubts about the efficacy of chemotherapy treatment, as the disease may remain latent, were considered negative experiences. Despite these doubts, patients continued their treatment, echoing findings from a study where doubts regarding efficacy were identified as a main concern for chemotherapy adherence. Further research is needed to understand how doubts about treatment efficacy can still encourage patients to adhere to chemotherapy treatment.

Strengths and limitation

The strength of this review lies in its comprehensive search strategy across databases to select appropriate articles. Additionally, the use of JBI guidelines provided a comprehensive and rigorous methodological approach in conducting this review. However, the exclusion of non-English studies, quantitative studies, and studies involving adolescents and children may limit the generalizability of the findings. Furthermore, this review focuses solely on chemotherapy treatment and does not encompass other types of cancer treatment.

Conclusion and practical implications

Based on the discussion of the findings, it is evident that maintaining a positive mentality and receiving social support can enhance chemotherapy adherence. Conversely, experiencing treatment side effects, concerns about efficacy, and unmet information needs may lead to lower adherence. These findings present an opportunity for healthcare professionals, particularly nurses, to develop standardized approaches aimed at facilitating chemotherapy treatment adherence, with a focus on providing comprehensive information. By assessing patients’ needs, healthcare professionals can tailor approaches to promote chemotherapy adherence and improve the survival rates of people living with cancer. Raising awareness and providing education about cancer and chemotherapy treatment can enhance patients’ understanding of the disease and its treatment options. Utilizing videos and reading materials in outpatient clinics and pharmacy settings can broaden the reach of educational efforts. Policy makers and healthcare providers can collaborate to develop sustainable patient education models to optimize patient outcomes in the context of cancer care. A deeper understanding of individual processes related to chemotherapy adherence is necessary to plan the implementation of interventions effectively. Further research examining the experiences of both adherent and non-adherent patients is essential to gain a comprehensive understanding of this topic.

Data availability

The datasets used and/or analysed during the current study available from the corresponding author on reasonable request. on our submission system as well.

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First author (AR) and second author (ST) conceived the review and the second author oversight for all stages of the review provided by the second author. All authors (AR), (ST), (WG) and (SK) undertook the literature search. Data extraction, screening the included papers and quality appraisal were undertaken by all authors (AR), (ST), (WG) and (SK). First and second authors (AR) and (ST) analysed the data and wrote the first draft of the manuscript and revised the manuscript and all authors (AR), (ST), (WG) and (SK) approved the final version of the manuscript.

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Rashidi, A., Thapa, S., Kahawaththa Palliya Guruge, W. et al. Patient experiences: a qualitative systematic review of chemotherapy adherence. BMC Cancer 24 , 658 (2024). https://doi.org/10.1186/s12885-024-12353-z

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A Systematic Review and Narrative Synthesis: Determinants of the Effectiveness and Sustainability of Measurement-Focused Quality Improvement Trainings

Associated data.

Supplemental Digital Content is Available in the Text.

Introduction:

The ability of health care professionals to measure change is critical for successful quality improvement (QI) efforts. Currently, there are no systematic reviews focusing on continuing education for health care professionals in data skills for QI. The purpose of this systematic review is to define effectiveness and sustainability of QI programs for health care professionals containing a measurement skills component and to identify barriers and facilitators to effectiveness and sustainability.

The systematic review involved study identification, screening, full text review, and data extraction. Four electronics databases and grey literature sources were searched to identify studies published between 2009 and 2019 (11 years). A customized data extraction form was developed. Mixed methods appraisal tool was used for quality assessment and a thematic analysis was conducted for narrative synthesis.

Fifty-three studies from 11 countries were included. Most study designs were quantitative descriptive (17/53) and used a blended learning approach (25/53) combining face-to face and distance learning modes. The programs included basic, intermediate, and advanced data skills concepts. Overall, studies reported positive outcomes for participant reaction, learning, and behavior, but reported variable success in sustainability and spread of QI.

Discussion:

Studies discussed measurement as a key competency for clinical QI. Effectiveness definitions focused on the short-term impact of the programs, whereas sustainability definitions emphasized maintenance of outcomes and skills in the long-term. Factors that influenced effectiveness and sustainability of the included studies were strategic approach to QI, organizational support, intervention design, communication, accountability, leadership support, and learning networks.

Health care organizations worldwide continue to test new systems and ways to enhance health care quality and patient safety. 1 Organizations are using continuing education programs in quality improvement (QI) methodologies to transform care and improve patient safety, reduce variations in care outcomes, and deliver sustainable changes in the health care system. 2 The use of such programs to improve health care has also gained considerable popularity in the health care system. 3 However, the health care system is complex and professional knowledge alone is not enough to engage in QI work to bring about change. 4 Numerous QI training programs have been developed to train health care staff in QI methodology and application.

QI training can improve processes, staff knowledge, and health outcomes. 5 Measurement is an important construct in all QI efforts because unless we measure, it is impossible to demonstrate whether the change has resulted in an improvement or not. 6 For health care staff today, collecting, processing, and understanding data is a part of routine practice. 7 This makes a strong case to train health care staff in quality measurement and to develop their expertise in the use of data. 8

Although there are several systematic reviews evaluating QI training and curricula, 9 – 11 none have focused on the evaluation of measurement for improvement training components. This systematic review differs from previous reviews by focusing on QI curricula and training programs containing a significant data skills component. The concepts of effectiveness and sustainability are critical to assessing the impact of teaching measurement skills to health care staff, but these concepts are underexplored in the QI literature. Effectiveness is a micro concept and refers to the assessment of the usefulness of an output at a certain point, with little reference to context. On the other hand, sustainability is a macro concept which extends over a longer period as the new ways of working or improved outcomes become the norm, with context being an essential element. 12 The purpose of this systematic review is to address this gap in literature and define effectiveness and sustainability of QI programs for health care professionals that have a data for improvement component and to identify the associated barriers and enablers.

Protocol and Registration

Review protocol for this systematic review is registered on PROSPERO (ID: CRD42019122997). This study was approved by the IRB of our institution.

Eligibility Criteria

Studies were included if:

Conducted in health care setting
Intervention was QI-based training and included a measurement component.
Study was about development, evaluation, or implementation of the program
Population was health care staff or postgraduate students
Based on primary research

Studies were excluded if:

There was no measurement for improvement component in intervention
Conference proceedings
Population was undergraduate students

Information Sources

Systematic review protocols were scanned in Prospero and Cochrane library to ensure novelty of the review question. A scoping search of databases was conducted to inform the development of the search strategy. Databases were purposively selected to include health care and education sources. The databases were: PubMed, CINAHL Plus, ERIC (via Pro-Quest), and Web of Science. Grey literature sources included two databases: OAIster and OpenGrey along with websites of leading organizations (see Supplemental File 1, Supplemental Digital Content 1, http://links.lww.com/JCEHP/A103 ). The reference lists of eligible studies were scanned to identify additional papers.

The search strategy (see Supplemental File 2, Supplemental Digital Content 2, http://links.lww.com/JCEHP/A104 ) was optimized toward sensitivity rather than specificity because the scoping search revealed that measurement for improvement was integrated into QI studies rather than being delivered as a standalone training. 13 The authors finalized the search strategy and databases iteratively. The systematic search of the literature was conducted in January 2019 and updated in June 2020. The search was restricted to papers published in last 11 years (Search date: January 1, 2009–December 31, 2019). Foreign language papers with English abstracts were considered at the initial stage but only included in full text review if a complete translation was available.

Study Selection

The systematic review consisted of four stages: study identification, title and abstract screening, full text review, and data extraction. Study screening was completed using Covidence tool. 14 Two reviewers independently conducted title and abstract screening. The reviewers met regularly to resolve disputes. The full text review was also conducted independently by two reviewers and discrepancies resolved via discussion. The two reviewers consulted a third reviewer to assist in making the decision on one paper at the full text review stage. Because the studies were heterogenous, a narrative synthesis was performed.

The database search returned 6184 articles, which were imported into Covidence. The 2499 duplicates were removed, leaving 3685 studies eligible for screening. After screening, 110 studies were shortlisted for full text review. A total of 53 studies were included in the review. The PRISMA flow diagram is presented in Figure Figure1 1 and the checklist is attached in Supplemental Digital Content 3 (see Supplemental File 3, http://links.lww.com/JCEHP/A105 ). 15

An external file that holds a picture, illustration, etc.
Object name is ceh-41-210-g001.jpg

PRISMA Statement. An overview of the study selection process. The 6184 records identified through database searching were screened by 2 reviewers. Nineteen records were identified and included from grey literature sources. Exclusion reasons at each stage are shown.

Assessment of Methodological Quality

The Mixed Methods Appraisal Tool (MMAT) was used to evaluate methodological quality. 16 The validity and reliability of the MMAT has been established and is suitable for appraising mixed method studies. 17 Two reviewers assessed quality independently and results were compared. Studies meeting the screening questions of the MMAT on clarity of research questions and appropriateness of collected data were considered appropriate quality for inclusion. All 53 studies met these criteria and were included in the review. The quality assessment is presented in Table Table1 1 .

Assessment of Methodological Quality Using MMAT *

Data Extraction

Two reviewers completed the data extraction independently. A customized data extraction form was developed (see Supplemental File 4, Supplemental Digital Content 4, http://links.lww.com/JCEHP/A106 ). One reviewer compared the data extraction forms and discrepancies were resolved through discussion between reviewers.

The 53 included studies were published between 2009 and 2019 and set in 11 countries. Most studies (35/53) were based in the United States. Most Study designs were quantitative descriptive (17/53) followed by mixed methods studies (16/53). The population varied widely, ranging from frontline staff, clinical and nonclinical staff, and leaders. A summary of studies is presented in Supplemental File 5, Supplemental Digital Content 5, http://links.lww.com/JCEHP/A131 .

Training Description

Less than half (14/53) of the studies were based on a collaborative approach. Duration of the collaboratives was variable, ranging from 2 months to 72 months. Half of the studies used a blended learning approach (25/53) combining face-to face and distance learning modes, whereas 21 studies relied solely on face-to-face learning modes. Four trainings were delivered online, whereas three studies did not state training modality used. Interventions included multiple training methods; the most common (39/53) one being face-to-face learning sessions. Other methods included teleconferencing (12/53), online modules (10/53), workshops (9/53), webinars (6/53), and emails (6/53).

Curriculum Description

The curricula were summarized into categories of basic, intermediate, and advanced data skills based on complexity of data concepts taught. Figure Figure2 2 summarizes the three categories and highlights the data concepts part of the training and curricula in the included studies. Basic data skills include concepts of measurement and QI knowledge, which are important for all health care staff. Intermediate data skills concepts are useful for staff working in improvement teams, whereas advanced data skills concepts are useful for improvement team leads and advisors.

An external file that holds a picture, illustration, etc.
Object name is ceh-41-210-g002.jpg

Measurement for Improvement Concepts. Summary of basic, intermediate, and advanced measurement and data skills taught in the QI programs.

Study Outcomes

Study outcomes are categorized as participant reaction, participant learning, participant behavior, sustainability, spread, and course design elements (see Supplemental File 6, Supplemental Digital Content 6, http://links.lww.com/JCEHP/A107 ). All studies measuring participation reaction to training and improved learning reported positive outcomes regardless of the study design. Studies measuring participant behavior also reported positive results except two quantitative descriptive studies. 63 , 65 Two randomized control trial studies 24 , 53 and a controlled interrupted time series study 50 reported not achieving the clinical outcomes being measured. A cohort study also reported not achieving the outcome of developing a culture of QI. 42 In spread, one quantitative descriptive study 49 reported no spread of QI methods. For sustainability outcomes, Glasgow et al 37 (Interrupted time series), Doyle et al 30 (Quantitative descriptive), and Cranley et al 26 (Mixed methods) reported a lack of sustainability of QI.

The Role of Measurement

The included studies reported on the role of measurement in QI for tracking progress toward goals and offering a systematic way to test changes to close performance gaps. 20 , 61 Measurement was identified as a key competency for clinical QI 21 to understand variation and improve the design health care. 4 Measurement was used to view data over time and draw conclusions regarding variations. 54 Measurement also played a role in implementation of QI methods 19 and contributed to the success of QI. 49

Knowledge about statistics and statistical process control 4 and additional support for measurement skills was reported by studies as critical. 2 , 33 , 39 Defining clear aims and measuring progress toward them was described as essential for QI. 62 Measurement was used to demonstrate patient outcomes to the host organizations 30 and provide guidance to decision makers. 49 Continuous measurement followed the processes through the project period into daily operations. 4 One study labelled measurement as one of the crucial elements of strategy for QI spread and sustainability. 31 Timely data and measurement are important for assessing progress and evaluation. 30

Challenges in identifying, collecting, and displaying appropriate measures of care impact QI program success. 4 , 18 , 63 Studies cited measurement challenges such as difficulty in obtaining measurable data 34 , 64 and presenting data in run/control chart formats. 35 Many participating hospitals were not equipped for systematic data collection. 31 Data collection and measurement was valued, 29 but perceived to be time consuming by participants. 52 , 53

Defining Effectiveness and Sustainability

The purpose of the review is to define effectiveness and sustainability and identify the barriers and enablers to success, in the context of QI programs with a focus on data and measurement for improvement. There is variability in how effectiveness and sustainability is defined in the studies. Another related concept that emerged was that of spread. It is therefore important to distinguish between effectiveness, spread, and sustainability. Effectiveness and sustainability definitions were extracted as part of the data extraction tool and summarized in Supplemental Digital Content 7 (see Supplemental file 7, http://links.lww.com/JCEHP/A108 ). The aspects addressed by these extracted definitions were then used to synthesize definitions. This was completed via consultation between three reviewers.

Effectiveness definitions focused on the short-term impact of the QI programs and were measured using participant reaction to the program, improved knowledge and skill application of participants, program participation and completion of QI projects by participants, and improvement in clinical outcomes at the end of the intervention period. Sustainability on the other hand, is defined not only as long-term outcomes beyond the intervention period, but also as a continuous process. Spread definitions focused on the diffusion of QI methods, processes, and skills from the intervention setting to nonintervention settings. We synthesized the following definitions of effectiveness, sustainability, and spread for measurement for improvement programs:

Effectiveness

Demonstrating improvement in key process, outcome, or quality measures being tracked, accompanied with an improvement in measurement knowledge, skills, and behaviors of learners during the intervention period.

Sustainability

Ongoing measurement, and development of processes and policies to maintain and improve the achieved gains in outcomes and participant skills and integration of measurement practices into routine after the intervention period, without further support from the trainers.

Active and passive diffusion of measurement skills and practices to areas and staff within and outside the organization that were not exposed to the training intervention.

Barriers and Enablers

A six-phased thematic analysis methodology (familiarization with data, initial coding, identifying themes, reviewing themes, naming themes, and reporting) was used to identify the barriers and enablers of sustainability 66 (see Supplemental file 8, Supplemental Digital Content 8, http://links.lww.com/JCEHP/A109 ). The definitions of effectiveness and sustainability synthesized were used as reference. The coding process was done manually by one reviewer and final themes were discussed and agreed with two other reviewers.

The four themes that emerged in enablers to effectiveness were intervention design, staff engagement, supportive leadership, and organizational support. Intervention design was the most important factor in the effectiveness of the program. Customizing training allows teaching of skills relevant to participant's role. 3 , 31 Considering the implementation context 3 , 39 , 52 , 53 and the challenges and opportunities of the setting 33 , 43 , 62 leads to targeted skill building. 19 A good starting point is to assess the prior knowledge and experience of participants 21 to determine training needs 31 and design a suitable range of resources 57 , 65 corresponding to diversity of experiences and knowledge levels. 64 Offering online modules 44 and online resources 53 also helps bridge this gap.

Intervention effectiveness can be enhanced using multiple learning strategies 21 and evidence-based curricula. 28 An effective intervention is responsive to participant learning styles 65 and improves the training based on feedback. 28 , 64 The best way to learn is by doing 21 and incorporating experiential learning principles 23 through demonstration projects 29 and case studies 65 builds capability. Another aspect of customized content is developing an interdisciplinary and team-based course 46 , 60 , 65 as working in teams prevents participants from becoming overburdened with measurement. 64 Having a participatory, data-driven approach contributes to effectiveness. 19 , 53 Focusing on real-time data increases 20 the program's value as participants can identify gaps in current processes. 23 , 55 Teaching practical data gathering, 21 statistical control charts, 4 data analysis, and comparison contribute to effectiveness. Feedback from fellow participants allows them to learn from each other and adds to effectiveness. 36 , 64 Similarly, informing participants about other team's progress creates healthy competition and prevents redundancy of efforts. 64

Effective coaching also plays an important role. Customized coaching experience through just-in-time coaching 28 and direct onsite, in-person support 33 improves effectiveness. Coaching is more effective when trainers can respond directly to participant concerns. 40 The ability of coaches to provide measurement support 22 in creating data collection processes 27 and data quality troubleshooting 33 adds to effectiveness. Practice facilitation 33 is also an enabler because providing private coaching between learning sessions, 44 ongoing mentorship 21 and tools and resources 39 are valued by participants. Coaches can provide customized feedback and assistance. 29 When participants perceive the training organization to be credible and have a sense of affiliation with it, they consider the training to be more effective. 23

Guiding participants in indicator selection by focusing on establishing clear, realistic, mutually agreed, 18 , 22 and clinically meaningful goals 43 , 47 , 50 is a successful strategy. Encouraging participants to focus on simple solutions 18 , 25 and making small changes 26 , 62 leads to effectiveness. In-person workshops 21 are an effective mode of training as face-to-face contact 33 is preferred by participants. Using technology for designing easy to access, self-paced and self-initiated interventions 57 improves effectiveness.

Successfully engaging health care staff is another important theme in effectiveness. Clinical staff feel empowered when they can identify and address gaps 25 and select relevant QI topics. 41 , 52 Providing dedicated time to participants to attend training sessions 34 , 41 , 42 , 45 , 53 also adds to effectiveness. Demonstrating the value of competency in QI skills 64 and offering maintenance of certification credit 59 also helps in creating enthusiasm among staff. Supportive policies of the organization such as assuring time release recognizes the training as a valued activity. 21 Leadership support is an important factor in the success of such programs. 21

There are four themes in barriers to effectiveness: incompatible intervention design, lack of staff engagement, lack of organizational support, and lack of strategic approach. Fast pace of collaboratives 18 and didactic instruction 30 which did not correspond to learning needs of all participants, especially those in support roles 3 were perceived as barriers. The number of concepts covered in the training made it difficult for participants to keep up and the terminology used was sometimes difficult to understand. 29 A single day of classroom training was an insufficient dose 50 and scheduling a full day training workshop is tiring for participants. 63 When training programs that did not incorporate advice on implementation 53 and leading change, 36 it proved to be a barrier to effectiveness.

Lack of organizational support was visible in cases where participants were not provided protected time and struggled to attend the sessions. 3 , 64 Poor data infrastructure impeded data collection 31 and obtaining baseline measures. 34 , 44 Lack of staff engagement and a negative perception about QI work and training because of previous negative experiences 18 , 45 dampened effectiveness. Some programs failed to incorporate appropriate reward systems to motivate behavior 36 and the lack of interest among participants resulted in low attendance 35 , 50 and in some cases, staff disliked new tools and processes that required learning new methods. 39 Staff struggled with learning measurement skills such as presenting data as run charts/control charts 35 , 36 which decreased collaborative effectiveness. Some did not see any value in investing time in such collaboratives 63 and believed the burdens outweighed the benefits. 29 , 36 Another barrier was the lack of a strategic approach and the participants selected projects that were incompatible with the goals of their institutions. 36 , 51

The themes observed in enablers to sustainability were taking a strategic approach, accountability, communication, learning networks, staff engagement, organizational support, intervention design, and supportive leadership. Taking a strategic approach requires connecting the program to organizational and national priorities, 21 strategic goals, 2 , 27 and teaming up with other departments 64 and organizations 53 with similar agendas. 29 As organizations prioritize and implement QI, 57 they move from sporadic efforts toward performance management systems, 5 which sustains learning. Incorporating strategies to address psychology 20 of change improves sustainability. Using a standard approach to QI ensures a common and clear improvement language. 44

Another aspect of sustainability is to recruit the right people in the project team. 4 , 22 A purposeful participant selection strategy 46 ensures inclusion of individuals who are interested in improvement work. Scale-up plans 18 with a goal of institution-wide diffusion 31 add to sustainability. It is important to integrate QI into programs and services 42 , 53 through updated job descriptions, 33 building QI responsibility into operational responsibilities 47 and continually reinforcing skills. 49 Engaging all stakeholders from an early stage 2 , 18 , 19 , 21 , 53 is also an enabler. In addition, while planning evaluations, it is important to assess learner involvement and QI project outcomes beyond completion of the programme. 52 A strategic approach requires taking a system-level view 20 , 21 of improvements with a blameless culture focusing on systems rather than individuals, 24 , 53 which considers challenges as system issues rather than staff issues. 28

Supportive organizational practices encourage QI by removing barriers, 26 investing in workforce capacity and culture change 28 and providing a conducive environment for teamwork. 38 , 45 It also commits resources 3 , 33 , 46 , 56 and provides opportunities to practice the skills learned. 19 , 22 , 27 Accountability is an important enabler for sustainability. A clear definition of responsibilities, 34 tasks 39 and individual roles 62 is key. Establishing time-bound targets 20 and regular meetings to follow through on action 62 ensures accountability. It is also beneficial to establish measurement guidelines to follow the process through the project period into daily operations. 4 This continuous sharing of numbers leads 53 to motivation and boosts sustainability. 4 The training organization can also provide external accountability 33 and ensure participants see projects to completion. 41

Focus on capacity building also improves sustainability. This includes training staff for specialized QI roles such as QI champion, 28 process coach, 31 and QI advisor. 33 A mentorship framework to support those interested in developing QI skills and encouraging permanent staff to develop coaching skills improves sustainability. 47 Effective communication contributes to sustainability. Recognizing the efforts of QI teams 26 , 34 by showcasing success stories 28 through ongoing promotional activities 56 is a rewarding strategy. Senior leader communication through board letters 31 also supports sustainability. Formal and informal dissemination are vital to communication and sustainability. Formal dissemination can include internal dissemination, 31 dissemination to local, national, and international audiences 2 and toolkits. 55 Informal dissemination can include enthusiastic employees 53 and other informal contacts. 31 Similarly, visual display of data and progress helps in disseminating the message of improvement. 59

Learning networks are an important enabler. 53 Learning from peers by sharing ideas 18 , 21 and building relationships creates a strong learning community for idea exchange. 33 These learning platforms serve as venues for knowledge transfer 57 and repositories for QI. 27 Development of collaborations between organizations leads to networking 56 and solution sharing. 33 Another area in staff engagement is generating awareness about QI 18 beyond the project team 2 , 26 , 42 and its impact on career. 22 Extended support from coaches for implementation sequencing 48 improves sustainability.

Support from leaders is crucial to sustainability. 53 This involves improving leaders' QI skills so they can develop infrastructure for QI in their organizations 28 such as establishing QI teams. 53 Senior leadership support 22 including board executives and chief of the medical staff provide legitimacy to QI. 31 A strong leadership structure championing QI on a daily basis 34 sends a message for sustainability. Leadership support allows staff to try new ideas in a safe environment that does not punish risk-taking. 56 Organizational support plays a role through various strategies such as incentivizing diffusion 48 and providing resources and autonomy to innovate. 3

Themes in barriers to sustainability include lack of accountability, poor communication, lack of leadership support, lack of staff engagement, lack of organizational support, absence of learning networks, and not having a strategic approach. When timelines, roles, and responsibilities are not established, the plan of actions can evaporate leading to slippage in agreed timeframes and a loss of momentum. 50 Because of poor institutional communication, staff lack a shared perception of problems 51 and often lack institutional knowledge to approach the relevant individuals for QI work. 60 Lack of leadership support manifests in the form of a lack of interest from top management 4 and variations in the readiness of senior leaders to engage in QI. 46

Learning networks play a vital role in sustainability; however, they are challenging to establish because few practices reach out to others to learn from them 62 and may also face difficulty in learning from practices with dissimilar QI capacity and patients. 29 Lack of organizational support is a major barrier to sustainability 53 because it represents a culture that is not conducive to making or sustaining change. 33 Presence of administrative red tape 3 , 64 can inhibit innovation and indicate that QI is not a priority for the organization. 55

Poor data infrastructure, 4 , 33 , 53 data quality, and access to data 4 decrease sustainability. Repeated data collection can be cumbersome and labor intensive in the long run. 52 Lack of resource availability 3 , 26 , 46 for QI projects is another barrier. Programs that lack ongoing organizational support are likely to be unsuccessful. 65 Health care staff have competing demands on their time, 46 , 62 , 63 , 63 which interferes with team's ability to meet and work. 3 Because QI teams are a disparate group of staff, 50 a lack of dedicated time for QI work 57 can be a barrier. Failing to engage staff, and leadership effectively and not focusing on motivation and behavior change can be a barrier to sustainability. 53 It is also important to account for the high levels of stress and emotional demands experienced by front-line staff. 1

The purpose of the systematic review is to define effectiveness and sustainability of QI programs with a significant data skills component and to identify the relevant barriers and enablers. Fifty-three studies were included in the review. There was heterogeneity in the content, teaching methods, and program design in the included studies and variability in the way effectiveness, sustainability, and spread were defined and measured in the context of QI programs. The review also highlighted variation in the ability of the programs to achieve desired outcomes. These inconsistencies in program success were attributed to various barriers and enablers to effectiveness and sustainability.

The lack of staff engagement, lack of a strategic approach, and lack of organizational support are barriers common between effectiveness and sustainability, which implies that these factors have implications for the short-term and long-term success of the programs. Poor intervention design affects the effectiveness of the program while poor communication, lack of accountability, and lack of leadership support can plague the ability to sustain the skills and results in the long-term. In enablers, intervention design, supportive leadership, engaged staff, and organizational support can affect positively on both effectiveness and sustainability of programs. Enablers that are relevant to sustainability are learning networks, communication, accountability, and a strategic approach to QI. The barriers and enablers highlight the importance of organizational, 39 learner, teacher, curricular, 35 and contextual factors 3 in the success of QI programs.

The definitions derived for effectiveness and sustainability highlight the importance of measurement. Studies reported measurement as a key competency for clinical QI. 21 Continuous measuring and remeasuring play an important role in maintaining 62 and operationalizing improvements in the long run. Selecting appropriate measures, 18 , 22 data collection 34 and using charts to display data 35 are essential to show effective change. 64 QI programs therefore need to focus on training staff in QI methods and how to measure care and use data to drive change. 62 There is an increasing expectation from health care professionals to measure, report, and continually improve the quality of care. 62 This indicates the need for a cultural shift from traditional academic-focused programs toward programs focusing on measurement and results to develop the capability of health professionals in leading improvement. 44

The findings of this systematic review also advocate for program evaluation to consider impact on participant behavior, patient outcomes, and supporting downstream learning beyond the direct participants of the programme. 43 Instead of solely relying on measuring quantitative outcomes, evaluators should also use qualitative data to assess whether program outcomes are achieved. 29

Measurement emerged as a critical element of QI training programs, which enables health care professionals and organizations to demonstrate effectiveness of improvement efforts and sustain improvements in the long run. Training health care professionals in data skills can have implications for improving health systems. However, health care systems are complex and various actors such as the health care authorities, training organizations, trainers, trainees, and trainee's organization have a collaborative role to play in ensuring effectiveness and sustainability of QI programs. Outputs of the thematic analysis in the form of effectiveness and sustainability barriers and enablers were broken down into inputs, outputs, and short- and long-term outcomes, which were then mapped onto a logic model. This was completed via consultation between three reviewers and presented in Figure Figure3 3 .

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Intervention success factors. Summary of QI program inputs contributing to achievement of short-term and long-term outcomes.

Limitations

A limitation of this review is that there were no stand-alone measurement for improvement training studies. The reviewers overcame this by establishing the presence of measurement component in the QI programs as an inclusion criterion. Because no search strategy is perfect, there is a risk of missing relevant studies; however, we mitigated this risk using a search strategy focused on sensitivity and iteratively testing the search strategy in selected databases.

The review highlighted that measuring the improvement in outcomes and participant knowledge establishes effectiveness while remeasuring continuously helps in sustaining outcomes in the long-term for QI programs with a significant measurement skills component. The review identified staff engagement, strategic approach to QI, organizational support, intervention design, communication, accountability, leadership support, and learning networks as factors that affect effectiveness and sustainability. The review expands current knowledge about the importance of measurement in QI training programs. Ensuring effectiveness and sustainability of measurement for improvement programs requires a collective effort from trainers, trainees, the organizations in which the interventions are implemented and policy makers.

Lessons for Practice

■ Measurement has a central role in demonstrating improvements and maintaining desired improvement outcomes of QI programs in the short- and long-term.
■ Staff engagement, strategic approach to QI, organizational support, intervention design, communication, accountability, leadership support, and learning networks influence effectiveness and sustainability of QI programs.
■ Effectiveness, sustainability, and spread of QI programs with a measurement component requires a collective effort from trainers, trainees, the organizations in which the interventions are implemented, and policy makers.

Supplementary Material

The corresponding author receives a PhD funding from the Health Service Executive Ireland (Project reference 57399). The study is also supported by the Irish Health Research Board (RL-2015-1588).

Disclosures: The authors declare no conflict of interest.

Supplemental digital content is available for this article. Direct URL citations appear in the printed text and are provided in the HTML and PDF versions of this article on the journal's Web site ( www.jcehp.org ).

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S1 Table. Data extraction table.