Patient, carer and family experiences of seeking redress and reconciliation following a life-changing event: Systematic review of qualitative evidence.

INTRODUCTION: We conducted a systematic review of qualitative evidence to improve understanding of the processes and outcomes of redress and reconciliation following a life-changing event from the perspectives of individuals experiencing the event and their families. METHODS: We searched six bibliographic databases for primary qualitative evidence exploring the views of individuals who have experienced a life-changing event, and/or their family or carers, of redress or reconciliation processes. This was supplemented with targeted database searches, forward and backward citation chasing and searches of Google Scholar and relevant websites. Title and abstract and full-text screening were undertaken independently by two reviewers. Data extraction and quality appraisal were conducted by one reviewer and checked by a second. We used a best-fit framework synthesis approach, drawing upon procedural and restorative justice concepts. FINDINGS: Fifty-three studies (61 papers) were eligible for inclusion. Forty-one studies (47 papers) were included in the synthesis, from which we identified four themes. Three themes 'Transparency', 'Person-centered' and 'Trustworthy' represent the procedural elements required to support a fair and objective process. The fourth, 'Restorative justice' encapsulates how a fair process feels to those who have experienced a life-changing event. This theme highlights the importance of an empathic relationship between the different parties involved in the redress-reconciliation process and the significance of being able to engage in meaningful action. CONCLUSION: Our findings highlight the procedural aspects and context of redress-reconciliation processes required to ensure that the process and outcomes are experienced as fair. These criteria may be applied to the processes used to investigate both recent and historical patient safety events. PUBLIC CONTRIBUTION: One member of the public affiliated with the Exeter Policy Research Programme Evidence Review Facility helped develop the review protocol. Two people with experience of medically life-changing events provided insight which corroborated our findings and identified important limitations of the evidence included in this review.

New horizons for caring for people with dementia in hospital: the DEMENTIA CARE pointers for service change.

Approximately two-thirds of hospital admissions are older adults and almost half of these are likely to have some form of dementia. People with dementia are not only at an increased risk of adverse outcomes once admitted, but the unfamiliar environment and routinised practices of the wards and acute care can be particularly challenging for them, heightening their confusion, agitation and distress further impacting the ability to optimise their care. It is well established that a person-centred care approach helps alleviate some of the unfamiliar stress but how to embed this in the acute-care setting remains a challenge. In this article, we highlight the challenges that have been recognised in this area and put forward a set of evidence-based 'pointers for service change' to help organisations in the delivery of person-centred care. The DEMENTIA CARE pointers cover areas of: dementia awareness and understanding, education and training, modelling of person-centred care by clinical leaders, adapting the environment, teamwork (not being alone), taking the time to 'get to know', information sharing, access to necessary resources, communication, involving family (ask family), raising the profile of dementia care, and engaging volunteers. The pointers extend previous guidance, by recognising the importance of ward cultures that prioritise dementia care and institutional support that actively seeks to raise the profile of dementia care. The pointers provide a range of simple to more complex actions or areas for hospitals to help implement person-centred care approaches; however, embedding them within the organisational cultures of hospitals is the next challenge.

Improving uncertainty tolerance in medical students: A scoping review.

INTRODUCTION: Uncertainty is an inevitable part of medical practice. An ability to tolerate uncertainty is viewed as a key competency across many health-care systems. Poor uncertainty tolerance (UT) has been linked to negative outcomes including reduced psychological well-being in medical students. A variety of medical education interventions have been developed with the intention of increasing medical students' UT. However, there is no synthesis of these studies available to inform education and research practice. Our aim was to conduct a scoping review of medical education interventions that evaluate their impact on UT. METHODS: Medline, PsycInfo, Embase and ERIC databases were searched for articles published from inception to December 2020. An extensive supplementary search was conducted and both quantitative and qualitative evaluations were included. For each intervention, we categorised the stimulus of uncertainty (ambiguity, complexity and/or probability) and mapped the students' reported cognitive, behavioural, and/or emotional response(s) to uncertainty onto an existing conceptual framework. RESULTS: Twenty-two of 24 included studies reported a positive impact on medical student UT in at least one domain (cognitive, behavioural or emotional). Interventions included problem based learning-based curricula, medical humanities, simulation, reflection and assessment. We found in four studies that a negative response in the emotional domain was reported despite positive responses also being reported in the cognitive and/or behavioural domains. CONCLUSION: We identified a range of medical education interventions which report a positive impact on medical student UT. Further research is required to understand why a single intervention may stimulate a negative emotional response alongside a positive cognitive or behavioural response. In turn, this could support stakeholders such as policymakers and institutions to adapt the medical curriculum to better prepare their medical students for practice by enhancing their UT.

Use of a search summary table to improve systematic review search methods, results, and efficiency.

BACKGROUND: Systematic reviews are comprehensive, robust, inclusive, transparent, and reproducible when bringing together the evidence to answer a research question. Various guidelines provide recommendations on the expertise required to conduct a systematic review, where and how to search for literature, and what should be reported in the published review. However, the finer details of the search results are not typically reported to allow the search methods or search efficiency to be evaluated. CASE PRESENTATION: This case study presents a search summary table, containing the details of which databases were searched, which supplementary search methods were used, and where the included articles were found. It was developed and published alongside a recent systematic review. This simple format can be used in future systematic reviews to improve search results reporting. CONCLUSIONS: Publishing a search summary table in all systematic reviews would add to the growing evidence base about information retrieval, which would help in determining which databases to search for which type of review (in terms of either topic or scope), what supplementary search methods are most effective, what type of literature is being included, and where it is found. It would also provide evidence for future searching and search methods research.

An alternative screening approach for Google Search identifies an accurate and manageable number of results for a systematic review (case study).

BACKGROUND: A challenge when using Google Search to identify studies for a systematic review is managing the high number of results, which can number in the hundreds of thousands or even more. Studies and guidance on web searching suggest limiting the screening process, e.g. to the first 100 results. OBJECTIVES: Our objective in this case study is to demonstrate an alternative approach to screening the results retrieved by Google Search which is based on our experience that the viewable number of results is often far fewer than the estimated number calculated by the search engine. METHODS: We screened the results of three searches of Google Search using our approach, which involves increasing the number of results displayed per page from 10 to the maximum of 100. We then calculated the viewable number of results and compared this with the estimated number. RESULTS: The mean of the estimated number of results for the three searches was 569,454,000. The mean of the viewable number results was 463 (0.00008% of the mean of the estimated number of results). CONCLUSION: Our findings challenge the commonly reported view that the number of results retrieved when using Google Search is too high to screen in full.

Epilepsy-specific patient-reported outcome measures of children's health-related quality of life: A systematic review of measurement properties.

OBJECTIVE: To identify and appraise published evidence of the measurement properties for epilepsy-specific patient-reported outcome measures (PROMs) of children's health-related quality of life (HRQoL). METHODS: We searched multiple databases for studies evaluating the measurement properties of English-language epilepsy-specific PROMs of children's HRQoL. We assessed the methodological quality using the COnsensus-based Standards for the selection of health Measurement INstruments (COSMIN) guidance. We extracted data about the content validity, construct validity, internal consistency, test-retest reliability, proxy reliability, responsiveness, and precision, and assessed the measurement properties with reference to standardized criteria. RESULTS: We identified 27 papers that evaluated 11 PROMs. Methodological quality was variable. Construct validity, test-retest reliability, and internal consistency were more commonly assessed. Quality of Life in Childhood Epilepsy (QoLCE) questionnaires are parent-reported and evaluated more than other PROMs; QoLCE-55 has good and replicated evidence for structural and construct validity and internal consistency. Health-Related Quality of Life Measure for Children with Epilepsy (CHEQoL) has both child and parent-reported versions and good evidence of content, structural, and construct validity. SIGNIFICANCE: This review identified two leading candidate epilepsy-specific PROMs for measuring health-related quality of life in children. Establishing evidence of the responsiveness of PROMs is a priority to help the interpretation of meaningful change scores.

Patient-initiated appointment systems for adults with chronic conditions in secondary care.

BACKGROUND: Missed hospital outpatient appointments is a commonly reported problem in healthcare services around the world; for example, they cost the National Health Service (NHS) in the UK millions of pounds every year and can cause operation and scheduling difficulties worldwide. In 2002, the World Health Organization (WHO) published a report highlighting the need for a model of care that more readily meets the needs of people with chronic conditions. Patient-initiated appointment systems may be able to meet this need at the same time as improving the efficiency of hospital appointments. OBJECTIVES: To assess the effects of patient-initiated appointment systems compared with consultant-led appointment systems for people with chronic or recurrent conditions managed in secondary care. SEARCH METHODS: We searched the Cochrane Central Register of Controlled Trials (CENTRAL), MEDLINE, Embase, and six other databases. We contacted authors of identified studies and conducted backwards and forwards citation searching. We searched for current/ongoing research in two trial registers. Searches were run on 13 March 2019. SELECTION CRITERIA: We included randomised trials, published and unpublished in any language that compared the use of patient-initiated appointment systems to consultant-led appointment systems for adults with chronic or recurrent conditions managed in secondary care if they reported one or more of the following outcomes: physical measures of health status or disease activity (including harms), quality of life, service utilisation or cost, adverse effects, patient or clinician satisfaction, or failures of the 'system'. DATA COLLECTION AND ANALYSIS: Two review authors independently screened all references at title/abstract stage and full-text stage using prespecified inclusion criteria. We resolved disagreements though discussion. Two review authors independently completed data extraction for all included studies. We discussed and resolved discrepancies with a third review author. Where needed, we contacted authors of included papers to provide more information. Two review authors independently assessed the risk of bias using the Cochrane Effective Practice and Organisation of Care 'Risk of bias' tool, resolving any discrepancies with a third review author. Two review authors independently assessed the certainty of the evidence using GRADE. MAIN RESULTS: The 17 included randomised trials (3854 participants; mean age 41 to 76 years; follow-up 12 to 72 months) covered six broad health conditions: cancer, rheumatoid arthritis, asthma, chronic obstructive pulmonary disease, psoriasis and inflammatory bowel disease. The certainty of the evidence using GRADE ratings was mainly low to very low. The results suggest that patient-initiated clinics may make little or no difference to anxiety (odds ratio (OR) 0.87, 95% confidence interval (CI) 0.68 to 1.12; 5 studies, 1019 participants; low-certainty evidence) or depression (OR 0.79 95% CI 0.51 to 1.23; 6 studies, 1835 participants; low-certainty evidence) compared to the consultant-led appointment system. The results also suggest that patient-initiated clinics may make little or no difference to quality of life (standardised mean difference (SMD) 0.12, 95% CI 0.00 to 0.25; 7 studies, 1486 participants; low-certainty evidence) compared to the consultant-led appointment system. Results for service utilisation (contacts) suggest there may be little or no difference in service utilisation in terms of contacts between the patient-initiated and consultant-led appointment groups; however, the effect is not certain as the rate ratio ranged from 0.68 to 3.83 across the studies (median rate ratio 1.11, interquartile (IQR) 0.93 to 1.37; 15 studies, 3348 participants; low-certainty evidence). It is uncertain if service utilisation (costs) are reduced in the patient-initiated compared to the consultant-led appointment groups (8 studies, 2235 participants; very low-certainty evidence). The results suggest that adverse events such as relapses in some conditions (inflammatory bowel disease and cancer) may have little or no reduction in the patient-initiated appointment group in comparison with the consultant-led appointment group (MD -0.20, 95% CI -0.54 to 0.14; 3 studies, 888 participants; low-certainty evidence). The results are unclear about any differences the intervention may make to patient satisfaction (SMD 0.05, 95% CI -0.41 to 0.52; 2 studies, 375 participants) because the certainty of the evidence is low, as each study used different questions to collect their data at different time points and across different health conditions. Some areas of risk of bias across all the included studies was consistently high (i.e. for blinding of participants and personnel and blinding of outcome assessment, other areas were largely of low risk of bias or were affected by poor reporting making the assessment unclear). AUTHORS' CONCLUSIONS: Patient-initiated appointment systems may have little or no effect on patient anxiety, depression and quality of life compared to consultant-led appointment systems. Other aspects of disease status and experience also appear to show little or no difference between patient-initiated and consultant-led appointment systems. Patient-initiated appointment systems may have little or no effect on service utilisation in terms of service contact and there is uncertainty about costs compared to consultant-led appointment systems. Patient-initiated appointment systems may have little or no effect on adverse events such as relapse or patient satisfaction compared to consultant-led appointment systems.

Activity interventions to improve the experience of care in hospital for people living with dementia: a systematic review.

BACKGROUND: An increasingly high number of patients admitted to hospital have dementia. Hospital environments can be particularly confusing and challenging for people living with dementia (Plwd) impacting their wellbeing and the ability to optimize their care. Improving the experience of care in hospital has been recognized as a priority, and non-pharmacological interventions including activity interventions have been associated with improved wellbeing and behavioral outcomes for Plwd in other settings. This systematic review aimed at evaluating the effectiveness of activity interventions to improve experience of care for Plwd in hospital. METHODS: Systematic searches were conducted in 16 electronic databases up to October 2019. Reference lists of included studies and forward citation searching were also conducted. Quantitative studies reporting comparative data for activity interventions delivered to Plwd aiming to improve their experience of care in hospital were included. Screening for inclusion, data extraction and quality appraisal were performed independently by two reviewers with discrepancies resolved by discussion with a third where necessary. Standardized mean differences (SMDs) were calculated where possible to support narrative statements and aid interpretation. RESULTS: Six studies met the inclusion criteria (one randomized and five non-randomized uncontrolled studies) including 216 Plwd. Activity interventions evaluated music, art, social, psychotherapeutic, and combinations of tailored activities in relation to wellbeing outcomes. Although studies were generally underpowered, findings indicated beneficial effects of activity interventions with improved mood and engagement of Plwd while in hospital, and reduced levels of responsive behaviors. Calculated SMDs ranged from very small to large but were mostly statistically non-significant. CONCLUSIONS: The small number of identified studies indicate that activity-based interventions implemented in hospitals may be effective in improving aspects of the care experience for Plwd. Larger well-conducted studies are needed to fully evaluate the potential of this type of non-pharmacological intervention to improve experience of care in hospital settings, and whether any benefits extend to staff wellbeing and the wider ward environment.

Effectiveness of pharmacist home visits for individuals at risk of medication-related problems: a systematic review and meta-analysis of randomised controlled trials.

BACKGROUND: Medication mismanagement is a major cause of both hospital admission and nursing home placement of frail older adults. Medication reviews by community pharmacists aim to maximise therapeutic benefit but also minimise harm. Pharmacist-led medication reviews have been the focus of several systematic reviews, but none have focussed on the home setting. REVIEW METHODS: To determine the effectiveness of pharmacist home visits for individuals at risk of medication-related problems we undertook a systematic review and meta-analysis of randomised controlled trials (RCTs). Thirteen databases were searched from inception to December 2018. Forward and backward citation of included studies was also performed. Articles were screened for inclusion independently by two reviewers. Randomised controlled studies of home visits by pharmacists for individuals at risk of medication-related problems were eligible for inclusion. Data extraction and quality appraisal were performed by one reviewer and checked by a second. Random-effects meta-analyses were performed where sufficient data allowed and narrative synthesis summarised all remaining data. RESULTS: Twelve RCTs (reported in 15 articles), involving 3410 participants, were included in the review. The frequency, content and purpose of the home visit varied considerably. The data from eight trials were suitable for meta-analysis of the effects on hospital admissions and mortality, and from three trials for the effects on quality of life. Overall there was no evidence of reduction in hospital admissions (risk ratio (RR) of 1.01 (95%CI 0.86 to 1.20, I2 = 69.0%, p = 0.89; 8 studies, 2314 participants)), or mortality (RR of 1.01 (95%CI 0.81 to 1.26, I2 = 0%, p = 0.94; 8 studies, 2314 participants)). There was no consistent evidence of an effect on quality of life, medication adherence or knowledge. CONCLUSION: A systematic review of twelve RCTs assessing the impact of pharmacist home visits for individuals at risk of medication related problems found no evidence of effect on hospital admission or mortality rates, and limited evidence of effect on quality of life. Future studies should focus on using more robust methods to assess relevant outcomes.

Feasibility of School-Based Identification of Children and Adolescents Experiencing, or At-risk of Developing, Mental Health Difficulties: a Systematic Review.

Under-identification of mental health difficulties (MHD) in children and young people contributes to the significant unmet need for mental health care. School-based programmes have the potential to improve identification rates. This systematic review aimed to determine the feasibility of various models of school-based identification of MHD. We conducted systematic searches in Medline, Embase, PsycINFO, ERIC, British Education Index, and ASSIA using terms for mental health combined with terms for school-based identification. We included studies that assessed feasibility of school-based identification of students in formal education aged 3-18 with MHD, symptomatology of MHD, or exposed to risks for MHD. Feasibility was defined in terms of (1) intervention fit, (2) cost and resource implications, (3) intervention complexity, flexibility, manualisation, and time concerns, and (4) adverse events. Thirty-three studies met inclusion criteria. The majority focused on behavioural and socioemotional problems or suicide risk, examined universal screening models, and used cross-sectional designs. In general, school-based programmes for identifying MHD aligned with schools' priorities, but their appropriateness for students varied by condition. Time, resource, and cost concerns were the most common barriers to feasibility across models and conditions. The evidence base regarding feasibility is limited, and study heterogeneity prohibits definitive conclusions about the feasibility of different identification models. Education, health, and government agencies must determine how to allocate available resources to make the widespread adoption of school-based identification programmes more feasible. Furthermore, the definition and measurement of feasibility must be standardised to promote any future comparison between models and conditions.

A systematic review of effectiveness and cost-effectiveness of school-based identification of children and young people at risk of, or currently experiencing mental health difficulties.

BACKGROUND: Although school-based programmes for the identification of children and young people (CYP) with mental health difficulties (MHD) have the potential to improve short- and long-term outcomes across a range of mental disorders, the evidence-base on the effectiveness of these programmes is underdeveloped. In this systematic review, we sought to identify and synthesise evidence on the effectiveness and cost-effectiveness of school-based methods to identify students experiencing MHD, as measured by accurate identification, referral rates, and service uptake. METHOD: Electronic bibliographic databases: MEDLINE, Embase, PsycINFO, ERIC, British Education Index and ASSIA were searched. Comparative studies were included if they assessed the effectiveness or cost-effectiveness of strategies to identify students in formal education aged 3-18 years with MHD, presenting symptoms of mental ill health, or exposed to psychosocial risks that increase the likelihood of developing a MHD. RESULTS: We identified 27 studies describing 44 unique identification programmes. Only one study was a randomised controlled trial. Most studies evaluated the utility of universal screening programmes; where comparison of identification rates was made, the comparator test varied across studies. The heterogeneity of studies, the absence of randomised studies and poor outcome reporting make for a weak evidence-base that only generate tentative conclusions about the effectiveness of school-based identification programmes. CONCLUSIONS: Well-designed pragmatic trials that include the evaluation of cost-effectiveness and detailed process evaluations are necessary to establish the accuracy of different identification models, as well as their effectiveness in connecting students to appropriate support in real-world settings.

Core Health Outcomes in Childhood Epilepsy (CHOICE): Development of a core outcome set using systematic review methods and a Delphi survey consensus.

OBJECTIVE: Establishing a core set of outcomes to be evaluated and reported in intervention trials aims to improve the usefulness of health research. There is no established core outcome set (COS) for childhood epilepsies. The aim of this study was to select a COS to be used in evaluative research of interventions for children with rolandic epilepsy (RE). METHODS: We followed guidance from the COMET (Core Outcome Measures in Effectiveness Trials) Initiative. First, we identified outcomes that had been measured in research through a systematic review. Second, young people with RE, parents, and professionals were invited to take part in a Delphi survey in which participants rated the importance of candidate outcomes. Last, a face-to-face meeting was convened to seek consensus on which outcomes were critical to include and to ratify the final COS. RESULTS: From 37 eligible papers in the review, we identified and included 48 candidate outcomes in the survey. We sent invitations to 165 people registered to take part in the survey; of these, 102 (62%) completed Round 1, and 80 (78%) completed Round 2 (three young people, 16 parents, 61 professionals). In Round 2 we included four additional outcomes suggested by participants in Round 1. The consensus meeting included two young people, four parents, and nine professionals who were eligible to vote and ratified the COS as 39 outcomes across 10 domains. SIGNIFICANCE: Our methodology was a proportionate and pragmatic approach toward producing a COS for evaluating research on interventions aiming to improve the health of children with RE.

Effectiveness of nonpharmacological interventions to reduce procedural anxiety in children and adolescents undergoing treatment for cancer: A systematic review and meta-analysis.

OBJECTIVE: Children and young people (CYP) with cancer undergo painful and distressing procedures. We aimed to systematically review the effectiveness of nonpharmacological interventions to reduce procedural anxiety in CYP. METHODS: Extensive literature searches sought randomised controlled trials that quantified the effect of any nonpharmacological intervention for procedural anxiety in CYP with cancer aged 0 to 25. Study selection involved independent title and abstract screening and full text screening by two reviewers. Anxiety, distress, fear, and pain outcomes were extracted from included studies. Where similar intervention, comparator, and outcomes presented, meta-analysis was performed, producing pooled effect sizes (Cohen's d) and 95% confidence intervals (95% CI). All other data were narratively described. Quality and risk of bias appraisal was performed, based on the Cochrane risk of bias tool. RESULTS: Screening of 11 727 records yielded 56 relevant full texts. There were 15 included studies, eight trialling hypnosis, and seven nonhypnosis interventions. There were large, statistically significant reductions in anxiety and pain for hypnosis, particularly compared with treatment as usual (anxiety: d = 2.30; 95% CI, 1.30-3.30; P < .001; pain: d = 2.16; 95% CI, 1.41-2.92; P < .001). Evidence from nonhypnosis interventions was equivocal, with some promising individual studies. There was high risk of bias across included studies limiting confidence in some positive effects. CONCLUSIONS: Evidence suggests promise for hypnosis interventions to reduce procedural anxiety in CYP undergoing cancer treatment. These results largely emerge from one research group, therefore wider research is required. Promising evidence for individual nonhypnosis interventions must be evaluated through rigorously conducted randomised controlled trials.

Dissemination and implementation research in dementia care: a systematic scoping review and evidence map.

BACKGROUND: The need to better understand implementing evidence-informed dementia care has been recognised in multiple priority-setting partnerships. The aim of this scoping review was to give an overview of the state of the evidence on implementation and dissemination of dementia care, and create a systematic evidence map. METHODS: We sought studies that addressed dissemination and implementation strategies or described barriers and facilitators to implementation across dementia stages and care settings. Twelve databases were searched from inception to October 2015 followed by forward citation and grey literature searches. Quantitative studies with a comparative research design and qualitative studies with recognised methods of data collection were included. Titles, abstracts and full texts were screened independently by two reviewers with discrepancies resolved by a third where necessary. Data extraction was performed by one reviewer and checked by a second. Strategies were mapped according to the ERIC compilation. RESULTS: Eighty-eight studies were included (30 quantitative, 34 qualitative and 24 mixed-methods studies). Approximately 60% of studies reported implementation strategies to improve practice: training and education of professionals (94%), promotion of stakeholder interrelationships (69%) and evaluative strategies (46%) were common; financial strategies were rare (15%). Nearly 70% of studies reported barriers or facilitators of care practices primarily within residential care settings. Organisational factors, including time constraints and increased workload, were recurrent barriers, whereas leadership and managerial support were often reported to promote implementation. Less is known about implementation activities in primary care and hospital settings, or the views and experiences of people with dementia and their family caregivers. CONCLUSION: This scoping review and mapping of the evidence reveals a paucity of robust evidence to inform the successful dissemination and implementation of evidence-based dementia care. Further exploration of the most appropriate methods to evaluate and report initiatives to bring about change and of the effectiveness of implementation strategies is necessary if we are to make changes in practice that improve dementia care.

Development and testing of a medline search filter for identifying patient and public involvement in health research.

BACKGROUND: Research involving the public as partners often proves difficult to locate due to the variations in terms used to describe public involvement, and inability of medical databases to index this concept effectively. OBJECTIVE: To design a search filter to identify literature where patient and public involvement (PPI) was used in health research. METHODS: A reference standard of 172 PPI papers was formed. The references were divided into a development set and a test set. Search terms were identified from common words, phrases and synonyms in the development set. These terms were combined as a search strategy for medline via OvidSP, which was then tested for sensitivity against the test set. The resultant search filter was then assessed for sensitivity, specificity and precision using a previously published systematic review. RESULTS: The search filter was found to be highly sensitive 98.5% in initial testing. When tested against results generated by a 'real-life' systematic review, the filter had a specificity of 81%. However, sensitivity dropped to 58%. Adjustments to the population group of terms increased the sensitivity to 73%. CONCLUSION: The PPI filter designed for medline via OvidSP could aid information specialists and researchers trying to find literature specific to PPI.

Measurement properties of multidimensional patient-reported outcome measures in neurodisability: a systematic review of evaluation studies.

AIM: To identify and appraise the quality of studies that primarily assessed the measurement properties of English language versions of multidimensional patient-reported outcome measures (PROMs) when evaluated with children with neurodisability, and to summarize this evidence. METHOD: MEDLINE, Embase, PsycINFO, CINAHL, AMED, and the National Health Service Economic Evaluation Database were searched. The methodological quality of the papers was assessed using the COnsensus-based Standards for selection of health Measurement INstruments checklist. Evidence of content validity, construct validity, internal consistency, test-retest reliability, proxy reliability, responsiveness, and precision was extracted and judged against standardized reference criteria. RESULTS: We identified 48 studies of mostly fair to good methodological quality: 37 papers for seven generic PROMs (CHIP, CHQ, CQoL, KIDSCREEN, PedsQL, SLSS, and YQOL), seven papers for two chronic-generic PROMs (DISABKIDS and Neuro-QOL), and four papers for three preference-based measures (HUI, EQ-5D-Y, and CHSCS-PS). INTERPRETATION: On the basis of this appraisal, the DISABKIDS appears to have more supportive evidence in samples of children with neurodisability. The overall lack of evidence for responsiveness and measurement error is a concern when using these instruments to measure change, or to interpret the findings of studies in which these PROMs have been used to assess change.

A systematic review of generic multidimensional patient-reported outcome measures for children, part I: descriptive characteristics.

OBJECTIVES: To identify generic, multidimensional patient-reported outcome measures (PROMs) for children up to 18 years old and describe their characteristics and content assessed using the International Classification of Functioning, Disability and Health Children and Youth version (ICF-CY). METHODS: The search strategy, developed by an information specialist, included four groups of terms related to "measure," "health," "children and young people," and "psychometric performance." The search was limited to publications from 1992. Five electronic databases and two online-specific PROM databases were searched. Two groups of reviewers independently screened all abstracts for eligible PROMs. Descriptive characteristics of the eligible PROMs were collected, and items and domains of each questionnaire were mapped onto the ICF-CY chapters. RESULTS: We identified 35 PROMs, of which 29 were generic PROMs and 6 were preference-based measures. Many PROMs cover a range of aspects of health; however, social functioning is represented most often. Content covered differs both in which aspects of health are assessed and whether individual questions focus on functioning (what the subject can or does do) and/or well-being (how the subject feels about a certain aspect of his or her health). CONCLUSIONS: A broad variety of PROMs is available to assess children's health. Nevertheless, only a few PROMs can be used across all age ranges to 18 years. When mapping their content on the ICF-CY, it seems that most PROMs exclude at least one major domain, and all conflate aspects of functioning and well-being in the scales.

A systematic review of generic multidimensional patient-reported outcome measures for children, part II: evaluation of psychometric performance of English-language versions in a general population.

OBJECTIVES: The objectives of this systematic review were 1) to identify studies that assess the psychometric performance of the English-language version of 35 generic multidimensional patient-reported outcome measures (PROMs) for children and young people in general populations and evaluate their quality and 2) to summarize the psychometric properties of each PROM. METHODS: MEDLINE, EMBASE, and PsycINFO were searched. The methodological quality of the articles was assessed using the COnsensus-based Standards for selection of health Measurement INstruments checklist. For each PROM, extracted evidence of content validity, construct validity, internal consistency, test-retest reliability, proxy reliability, responsiveness, and precision was judged against standardized reference criteria. RESULTS: We found no evidence for 14 PROMs. For the remaining 21 PROMs, 90 studies were identified. The methodological quality of most studies was fair. Quality was generally rated higher in more recent studies. Not reporting how missing data were handled was the most common reason for downgrading the quality. None of the 21 PROMs has had all psychometric properties evaluated; data on construct validity and internal consistency were most frequently reported. CONCLUSIONS: Overall, consistent positive findings for at least five psychometric properties were found for Child Health and Illness Profile, Healthy Pathways, KIDSCREEN, and Multi-dimensional Student Life Satisfaction Scale. None of the PROMs had been evaluated for responsiveness to detect change in general populations. Further well-designed studies with transparent reporting of methods and results are required.

Meaningful health outcomes for paediatric neurodisability: Stakeholder prioritisation and appropriateness of patient reported outcome measures.

BACKGROUND: Health services are increasingly focused on measuring and monitoring outcomes, particularly those that reflect patients' priorities. To be meaningful, outcomes measured should be valued by patients and carers, be consistent with what health professionals seek to achieve, and be robust in terms of measurement properties. The aim of this study was (i) to seek a shared vision between families and clinicians regarding key aspects of health as outcomes, beyond mortality and morbidity, for children with neurodisability, and (ii) to appraise which multidimensional patient reported outcome measures (PROMs) could be used to assess salient health domains. METHODS: Relevant outcomes were identified from (i) qualitative research with children and young people with neurodisability and parent carers, (ii) Delphi survey with health professionals, and (iii) systematic review of PROMs. The International Classification of Functioning Disability and Health provided a common language to code aspects of health. A subset of stakeholders participated in a prioritisation meeting incorporating a Q-sorting task to discuss and rank aspects of health. RESULTS: A total of 33 pertinent aspects of health were identified. Fifteen stakeholders from the qualitative and Delphi studies participated in the prioritisation meeting: 3 young people, 5 parent carers, and 7 health professionals. Aspects of health that emerged as more important for families and targets for health professionals were: communication, emotional wellbeing, pain, sleep, mobility, self-care, independence, mental health, community and social life, behaviour, toileting and safety. Whilst available PROMs measure many aspects of health in the ICF, no single PROM captures all the key domains prioritised as for children and young people with neurodisability. The paucity of scales for assessing communication was notable. CONCLUSIONS: We propose a core suite of key outcome domains for children with neurodisability that could be used in evaluative research, audit and as health service performance indicators. Future work could appraise domain-specific PROMs for these aspects of health; a single measure assessing the key aspects of health that could be applied across paediatric neurodisability remains to be developed.

Sleep positioning systems for children with cerebral palsy.

BACKGROUND: Sleep positioning systems can be prescribed for children with cerebral palsy to help reduce or prevent hip migration, provide comfort to ease pain and/or improve sleep. As sleep disturbance is common in children with developmental disabilities, with impact on their carers' sleep, and as sleep positioning systems can be expensive, guidance is needed to support decisions as to their use. OBJECTIVES: To determine whether commercially-available sleep positioning systems, compared with usual care, reduce or prevent hip migration in children with cerebral palsy. Any negative effect of sleep positioning systems on hip migration will be considered within this objective.Secondary objectives were to determine the effect of sleep positioning systems on: (1) number or frequency of hip problems; (2) sleep patterns and quality; (3) quality of life of the child and family; (4) pain; and (5) physical functioning. We also sought to identify any adverse effects from using sleep positioning systems. SEARCH METHODS: In December 2014, we searched CENTRAL, Ovid MEDLINE, Embase, and 13 other databases. We also searched two trials registers. We applied no restrictions on date of publication, language, publication status or study design. We checked references and contacted manufacturers and authors for potentially relevant literature, and searched the internet using Google. SELECTION CRITERIA: We included all randomised controlled trials (RCTs) evaluating whole body sleep positioning systems for children and adolescents (up to 18 years of age) with cerebral palsy. DATA COLLECTION AND ANALYSIS: Two review authors independently screened reports retrieved from the search against pre-determined inclusion criteria and assessed the quality of eligible studies.Members of the public (parent carers of children with neurodisability) contributed to this review by suggesting the topic, refining the research objectives, interpreting the findings, and reviewing the plain language summary. MAIN RESULTS: We did not identify any randomised controlled trials that evaluated the effectiveness of sleep positioning systems on hip migration.We did find two randomised cross-over trials that met the inclusion criteria in respect of secondary objectives relating to sleep quality and pain. Neither study reported any important difference between sleeping in sleep positioning systems and not for sleep patterns or sleep quality (two studies, 21 children, very low quality evidence) and pain (one study, 11 children, very low quality evidence). These were small studies with established users of sleep positioning systems and were judged to have high risk of bias.We found no eligible trials that explored the other secondary objectives (number or frequency of hip problems, quality of life of the child and family, physical functioning, and adverse effects). AUTHORS' CONCLUSIONS: We found no randomised trials that evaluated the effectiveness of sleep positioning systems to reduce or prevent hip migration in children with cerebral palsy. Nor did we find any randomised trials that evaluated the effect of sleep positioning systems on the number or frequency of hip problems, quality of life of the child and family or on physical functioning.Limited data from two randomised trials, which evaluated the effectiveness of sleep positioning systems on sleep quality and pain for children with cerebral palsy, showed no significant differences in these aspects of health when children were using and not using a sleep positioning system.In order to inform clinical decision-making and the prescription of sleep positioning systems, more rigorous research is needed to determine effectiveness, cost-effectiveness, and the likelihood of adverse effects.