Strategies used for childhood chronic functional constipation: the SUCCESS evidence synthesis.

Background: Up to 30% of children have constipation at some stage in their life. Although often short-lived, in one-third of children it progresses to chronic functional constipation, potentially with overflow incontinence. Optimal management strategies remain unclear. Objective: To determine the most effective interventions, and combinations and sequences of interventions, for childhood chronic functional constipation, and understand how they can best be implemented. Methods: Key stakeholders, comprising two parents of children with chronic functional constipation, two adults who experienced childhood chronic functional constipation and four health professional/continence experts, contributed throughout the research. We conducted pragmatic mixed-method reviews. For all reviews, included studies focused on any interventions/strategies, delivered in any setting, to improve any outcomes in children (0-18 years) with a clinical diagnosis of chronic functional constipation (excluding studies of diagnosis/assessment) included. Dual reviewers applied inclusion criteria and assessed risk of bias. One reviewer extracted data, checked by a second reviewer. Scoping review: We systematically searched electronic databases (including Medical Literature Analysis and Retrieval System Online, Excerpta Medica Database, Cumulative Index to Nursing and Allied Health Literature) (January 2011 to March 2020) and grey literature, including studies (any design) reporting any intervention/strategy. Data were coded, tabulated and mapped. Research quality was not evaluated. Systematic reviews of the evidence of effectiveness: For each different intervention, we included existing systematic reviews judged to be low risk of bias (using the Risk of Bias Assessment Tool for Systematic Reviews), updating any meta-analyses with new randomised controlled trials. Where there was no existing low risk of bias systematic reviews, we included randomised controlled trials and other primary studies. The risk of bias was judged using design-specific tools. Evidence was synthesised narratively, and a process of considered judgement was used to judge certainty in the evidence as high, moderate, low, very low or insufficient evidence. Economic synthesis: Included studies (any design, English-language) detailed intervention-related costs. Studies were categorised as cost-consequence, cost-effectiveness, cost-utility or cost-benefit, and reporting quality evaluated using the consensus health economic criteria checklist. Systematic review of implementation factors: Included studies reported data relating to implementation barriers or facilitators. Using a best-fit framework synthesis approach, factors were synthesised around the consolidated framework for implementation research domains. Results: Stakeholders prioritised outcomes, developed a model which informed evidence synthesis and identified evidence gaps. Scoping review: 651 studies, including 190 randomised controlled trials and 236 primary studies, conservatively reported 48 interventions/intervention combinations. Effectiveness systematic reviews: studies explored service delivery models (n = 15); interventions delivered by families/carers (n = 32), wider children's workforce (n = 21), continence teams (n = 31) and specialist consultant-led teams (n = 42); complementary therapies (n = 15); and psychosocial interventions (n = 4). One intervention (probiotics) had moderate-quality evidence; all others had low to very-low-quality evidence. Thirty-one studies reported evidence relating to cost or resource use; data were insufficient to support generalisable conclusions. One hundred and six studies described implementation barriers and facilitators. Conclusions: Management of childhood chronic functional constipation is complex. The available evidence remains limited, with small, poorly conducted and reported studies. Many evidence gaps were identified. Treatment recommendations within current clinical guidelines remain largely unchanged, but there is a need for research to move away from considering effectiveness of single interventions. Clinical care and future studies must consider the individual characteristics of children. Study registration: This study is registered as PROSPERO CRD42019159008. Funding: This award was funded by the National Institute for Health and Care Research (NIHR) Health Technology Assessment programme (NIHR award ref: 128470) and is published in full in Health Technology Assessment; Vol. 28, No. 5. See the NIHR Funding and Awards website for further award information.

Between 5% and 30% of children experience constipation at some stage. In one-third of these children, this progresses to chronic functional constipation. Chronic functional constipation affects more children with additional needs. We aimed to find and bring together published information about treatments for chronic functional constipation, to help establish best treatments and treatment combinations. We did not cover assessment or diagnosis of chronic functional constipation. This project was guided by a 'stakeholder group', including parents of children with constipation, people who experienced constipation as children, and healthcare professionals/continence experts. We carried out a 'scoping review' and a series of 'systematic reviews'. Our 'scoping review' provides an overall picture of research about treatments, with 651 studies describing 48 treatments. This helps identify important evidence gaps. 'Systematic reviews' are robust methods of bringing together and interpreting research evidence. Our stakeholder group decided to structure our systematic reviews to reflect who delivered the interventions. We brought together evidence about how well treatments worked when delivered by families/carers (32 studies), the wider children's workforce (e.g. general practitioner, health visitor) (21 studies), continence teams (31 studies) or specialist consultant-led teams (42 studies). We also considered complementary therapies (15 studies) and behavioural strategies (4 studies). Care is affected by what is done and how it is done. We brought together evidence about different models of delivering care (15 studies), barriers and facilitators to implementation of treatments (106 studies) and costs (31 studies). Quality of evidence was mainly low to very low. Despite numerous studies, there was often insufficient information to support generalisable conclusions. Our findings generally agreed with current clinical guidelines. Management of childhood chronic functional constipation should be child-centred, multifaceted and adapted according to the individual child, their needs, the situation in which they live and the health-care setting in which they are looked after. Research is needed to address our identified evidence gaps.

Protocol for the development of guidance for collaborator and partner engagement in health care evidence syntheses.

BACKGROUND: Involving collaborators and partners in research may increase relevance and uptake, while reducing health and social inequities. Collaborators and partners include people and groups interested in health research: health care providers, patients and caregivers, payers of health research, payers of health services, publishers, policymakers, researchers, product makers, program managers, and the public. Evidence syntheses inform decisions about health care services, treatments, and practice, which ultimately affect health outcomes. Our objectives are to: A. Identify, map, and synthesize qualitative and quantitative findings related to engagement in evidence syntheses B. Explore how engagement in evidence synthesis promotes health equity C. Develop equity-oriented guidance on methods for conducting, evaluating, and reporting engagement in evidence syntheses METHODS: Our diverse, international team will develop guidance for engagement with collaborators and partners throughout multiple sequential steps using an integrated knowledge translation approach: 1. Reviews. We will co-produce 1 scoping review, 3 systematic reviews and 1 evidence map focusing on (a) methods, (b) barriers and facilitators, (c) conflict of interest considerations, (d) impacts, and (e) equity considerations of engagement in evidence synthesis. 2. Methods study, interviews, and survey. We will contextualise the findings of step 1 by assessing a sample of evidence syntheses reporting on engagement with collaborators and partners and through conducting interviews with collaborators and partners who have been involved in producing evidence syntheses. We will use these findings to develop draft guidance checklists and will assess agreement with each item through an international survey. 3. CONSENSUS: The guidance checklists will be co-produced and finalised at a consensus meeting with collaborators and partners. 4. DISSEMINATION: We will develop a dissemination plan with our collaborators and partners and work collaboratively to improve adoption of our guidance by key organizations. CONCLUSION: Our international team will develop guidance for collaborator and partner engagement in health care evidence syntheses. Incorporating partnership values and expectations may result in better uptake, potentially reducing health inequities.

Exploring eye care pathways, patient priorities and economics in Pakistan: A scoping review and expert consultation study with thematic analysis.

PURPOSE: As the prevalence of eye diseases increases, demand for effective, accessible and equitable eye care grows worldwide. This is especially true in lower and middle-income countries, which have variable levels of infrastructure and economic resources to meet this increased demand. In the present study we aimed to review the literature on eye care in Pakistan comprehensively, with a particular focus on eye care pathways, patient priorities and economics. METHODS: A systematic scoping review was performed to identify literature relating to eye care in Pakistan. Searches of relevant electronic databases and grey literature were carried out. The results were analysed through a mixed methods approach encompassing descriptive numerical summary and thematic analysis. To consolidate results and define priority areas for future study, expert consultation exercises with key stakeholders were conducted using qualitative semi-structured interviews. RESULTS: One hundred and thirty-two papers (published and unpublished) were included in the final review. The majority (n = 93) of studies utilised a quantitative design. Seven interlinked themes were identified: eye care pathways, burden of eye disease, public views on eye-related issues, workforce, barriers to uptake of eye care services, quality of eye care services and economic impact of blindness. Research priorities included investigating the eye care workforce, the quality and efficiency of current eye care services, eye care services available in rural Pakistan and the costs and benefits related to eye care provision and sustaining eye care programmes. CONCLUSIONS: To the best of our knowledge, this is the first review to synthesise evidence from papers across the field relating to eye care in Pakistan. As such, this work provides new insights into the achievements of the national eye health programme, challenges in eye care in Pakistan and priority areas for future research.

Protocol for the development of guidance for stakeholder engagement in health and healthcare guideline development and implementation.

BACKGROUND: Stakeholder engagement has become widely accepted as a necessary component of guideline development and implementation. While frameworks for developing guidelines express the need for those potentially affected by guideline recommendations to be involved in their development, there is a lack of consensus on how this should be done in practice. Further, there is a lack of guidance on how to equitably and meaningfully engage multiple stakeholders. We aim to develop guidance for the meaningful and equitable engagement of multiple stakeholders in guideline development and implementation. METHODS: This will be a multi-stage project. The first stage is to conduct a series of four systematic reviews. These will (1) describe existing guidance and methods for stakeholder engagement in guideline development and implementation, (2) characterize barriers and facilitators to stakeholder engagement in guideline development and implementation, (3) explore the impact of stakeholder engagement on guideline development and implementation, and (4) identify issues related to conflicts of interest when engaging multiple stakeholders in guideline development and implementation. DISCUSSION: We will collaborate with our multiple and diverse stakeholders to develop guidance for multi-stakeholder engagement in guideline development and implementation. We will use the results of the systematic reviews to develop a candidate list of draft guidance recommendations and will seek broad feedback on the draft guidance via an online survey of guideline developers and external stakeholders. An invited group of representatives from all stakeholder groups will discuss the results of the survey at a consensus meeting which will inform the development of the final guidance papers. Our overall goal is to improve the development of guidelines through meaningful and equitable multi-stakeholder engagement, and subsequently to improve health outcomes and reduce inequities in health.

Effectiveness of Motivational Interviewing on adult behaviour change in health and social care settings: A systematic review of reviews.

BACKGROUND: The challenge of addressing unhealthy lifestyle choice is of global concern. Motivational Interviewing has been widely implemented to help people change their behaviour, but it is unclear for whom it is most beneficial. This overview aims to appraise and synthesise the review evidence for the effectiveness of Motivational Interviewing on health behaviour of adults in health and social care settings. METHODS: A systematic review of reviews. Methods were pre-specified and documented in a protocol (PROSPERO-CRD42016049278). We systematically searched 7 electronic databases: CDSR; DARE; PROSPERO; MEDLINE; CINAHL; AMED and PsycINFO from 2000 to May 2018. Two reviewers applied pre-defined selection criteria, extracted data using TIDIER guidelines and assessed methodological quality using the ROBIS tool. We used GRADE criteria to rate the strength of the evidence for reviews including meta-analyses. FINDINGS: Searches identified 5222 records. One hundred and four reviews, including 39 meta-analyses met the inclusion criteria. Most meta-analysis evidence was graded as low or very low (128/155). Moderate quality evidence for mainly short term (<6 months) statistically significant small beneficial effects of Motivational Interviewing were found in 11 of 155 (7%) of meta-analysis comparisons. These outcomes include reducing binge drinking, frequency and quantity of alcohol consumption, substance abuse in people with dependency or addiction, and increasing physical activity participation. CONCLUSIONS: We have created a comprehensive map of reviews relating to Motivational Interviewing to signpost stakeholders to the best available evidence. More high quality research is needed to be confident about the effectiveness of Motivational Interviewing. We identified a large volume of low quality evidence and many areas of overlapping research. To avoid research waste, it is vital for researchers to be aware of existing research, and the implications arising from that research. In the case of Motivational Interviewing issues relating to monitoring and reporting fidelity of interventions need to be addressed.

A scoping review of trials of interventions led or delivered by cancer nurses.

BACKGROUND: Advances in research and technology coupled with an increased cancer incidence and prevalence have resulted in significant expansion of cancer nurse role, in order to meet the growing demands and expectations of people affected by cancer (PABC). Cancer nurses are also tasked with delivering an increasing number of complex interventions as a result of ongoing clinical trials in cancer research. However much of this innovation is undocumented, and we have little insight about the nature of novel interventions currently being designed or delivered by cancer nurses. OBJECTIVES: To identify and synthesise the available evidence from clinical trials on interventions delivered or facilitated by cancer nurses. DATA SOURCES AND REVIEW METHODS: A systematic review of randomised controlled trials (RCT), quasi-RCTs and controlled before and after studies (CBA) of cancer nursing interventions aimed at improving the experience and outcomes of PABC. Ten electronic databases (CENTRAL, MEDLINE, AMED, CINAHL, EMBASE, Epistemonikos, CDSR, DARE, HTA, WHO ICTRP) were searched between 01 January 2000 and 31 May 2016. No language restrictions were applied. Bibliographies of selected studies and relevant Cochrane reviews were also hand-searched. Interventions delivered by cancer nurses were classified according to the OMAHA System. Heat maps were used to highlight the volume of evidence available for different cancer groups, intervention types and stage of cancer care continuum. RESULTS: The search identified 22,450 records; we screened 16,169 abstracts and considered 925 full papers, of which 214 studies (247,550 participants) were included in the evidence synthesis. The majority of studies were conducted in Europe (n = 79) and USA (n = 74). Interventions were delivered across the cancer continuum from prevention and risk reduction to survivorship, with the majority of interventions delivered during the treatment phase (n = 137). Most studies (131/214) had a teaching, guidance or counselling component. Cancer nurse interventions were targeted at primarily breast, prostate or multiple cancers. No studies were conducted in brain, sarcoma or other rare cancer types. The majority of the studies (n = 153) were nurse-led and delivered by specialist cancer nurses (n = 74) or advanced cancer nurses (n = 29), although the quality of reporting was poor. CONCLUSIONS: To the best of our knowledge, this is the first review to synthesise evidence from intervention studies across the entire cancer spectrum. As such, this work provides new insights into the nature of the contribution that cancer nurses have made to evidence-based innovations, as well as highlighting areas in which cancer nursing trials can be developed in the future.

Optimisation of complex health interventions prior to a randomised controlled trial: a scoping review of strategies used.

BACKGROUND: Many complex intervention trials fail to show an intervention effect. Although this may be due to genuine ineffectiveness, it may also be the result of sub-optimal intervention design, implementation failure or a combination of these. Given current financial constraints and the pressure to reduce waste and increase value in health services research, pre-trial strategies are needed to reduce the likelihood of design or implementation failure and to maximise the intervention's potential for effectiveness. In this scoping review, we aimed to identify and synthesise the available evidence relating to the strategies and methods used to 'optimise' complex interventions at the pre-trial stage. METHODS: We searched MEDLINE, CINAHL, AMED, PsycINFO and ProQuest Nursing & Allied Health Source for papers published between January 2000 and March 2015. We included intervention development and optimisation studies that explored potential intervention weaknesses and limitations before moving to a definitive randomised controlled trial (RCT). Two reviewers independently applied selection criteria and systematically extracted information relating to the following: study characteristics; intervention under development; framework used to guide the development process; areas of focus of the optimisation process, methods used and outcomes of the optimisation process. Data were tabulated and summarised in a narrative format. RESULTS: We screened 3968 titles and 231 abstracts for eligibility. Eighty-nine full-text papers were retrieved; 27 studies met our selection criteria. Optimisation strategies were used for a range of reasons: to explore the feasibility and acceptability of the intervention to patients and healthcare professionals; to estimate the effectiveness and cost-effectiveness of different combinations of intervention components; and to identify potential barriers to implementation. Methods varied widely across studies, from interviews and focus groups to economic modelling and probability analysis. CONCLUSIONS: The review identifies a range of optimisation strategies currently used. Although a preliminary classification of these strategies can be proposed, a series of questions remain as to which methods to use for different interventions and how to determine when the intervention is ready or 'optimised enough' to be tested in a RCT. Future research should explore potential answers to the questions raised, to guide researchers in the development and evaluation of more effective interventions.

Pediatric non-neuronopathic Gaucher disease: presentation, diagnosis and assessment. Consensus statements.

UNLABELLED: Gaucher disease is caused by defective activity of glucocerebrosidase. The resulting accumulation of glucocerebroside in the lysosomes of visceral macrophages in various tissue and organ compartments leads to multiple manifestations, including hepatosplenomegaly, anemia, thrombocytopenia, growth retardation and skeletal disease. The most prevalent form of Gaucher disease is the non-neuronopathic (type 1) variant, which lacks primary involvement of the central nervous system. Traditionally, this has been referred to as the 'adult type'; however, 66% of individuals with symptomatic non-neuronopathic Gaucher disease manifest in childhood. Onset in childhood is usually predictive of a severe, rapidly progressive phenotype and children with non-neuronopathic Gaucher disease are at high risk for morbid complications. Enzyme therapy with recombinant human glucocerebrosidase in childhood can restore health in reversible manifestations and prevent the development of irreversible symptoms. A heightened focus on pediatric Gaucher disease is therefore needed. Although some correlation has been found between genotype and phenotype, mutation analysis is of limited value in disease prognosis. Management of pediatric Gaucher disease should be underpinned by a thorough assessment of the phenotype at baseline with regular monitoring thereafter. Excluding neuronopathic disease is recommended as the first step. Subsequently, baseline evaluation should focus on staging of different storage tissues, particularly the bone the involvement of which results in the greatest long-term morbidity. These organ assessments are recommended because bone disease severity may not correlate with disease severity in other organs and vice versa. In addition, different organs may respond differently to therapy. Initial assessment of each organ system can enable setting of realistic and individualized goals. CONCLUSION: A thorough approach to baseline assessment will improve the understanding of childhood Gaucher disease, optimizing management to minimize impairment of growth and development and prevent irreversible symptoms.