Title: Strategies Used for Constipation in Children - Evidence Synthesis involving Stakeholders (SUCCESS)
Funder: Chief Scientist Office, Scotland

Abstract: Thirty percent of parents report constipation at some stage in their child’s life. Often short-lived, but in some it progresses to long-term severe constipation, potentially with overflow incontinence. There are often difficulties in identifying the problem and getting a diagnosis. A lack of treatment options can result in considerable distress for the child and family, social isolation and bullying. Laxatives are usually the first treatment given, but often parents and GPs are reluctant to use them. Education and lifestyle advice should be given alongside laxatives but are often not. Many parents use social media for advice. The problem is greater in severity and prevalence if learning disabilities are involved._x000D_ _x000D_ We do not know enough about how best to treat chronic functional constipation (CFC) in children. We do not know how well different treatments and models of support work, or the best ways to combine them. In this project we will look for information that has been published about the various treatments and find out the best treatments and treatment combinations. This will provide important information for health professionals, and parents, on how best to manage CFC, and where more research is needed. _x000D_ _x000D_ A stakeholder group (SG) will help deliver the project. It will include parents of children with CFC, those who have experienced CFC as a child, and health professionals and charities involved in providing care and advice. The SG will meet regularly, using telephone and email and will work with the research team to make sure the project covers the most important treatments, treatment combinations, and outcomes. The research team includes experts in finding and reporting what has been published about different treatments, and health professionals who work with children with CFC._x000D_ _x000D_ First, we will get a broad overview of the treatments commonly used by bringing together information from a wide range of sources. For example, the results of research and information from social media. We will build on reviews that have already been done. _x000D_ Second, we will carry out two focussed systematic reviews. A 'systematic review' is a method of carefully bringing together all existing studies, assessing how good each one is, comparing their findings and - if possible - making overall conclusions about what works best. One of these reviews will look at how effective treatments for childhood CFC are and their costs. The other will look at the things that make it easier for treatments to be provided. _x000D_ _x000D_ Finally, we will bring the findings from our broad overview and the two systematic reviews together. This will show what is known about treatments for childhood CFC, including what has been shown to work best, and the best ways to make these treatments available. It will show results for children with and without special needs and where there are gaps in knowledge and more research is needed. To make all this information useful and easy to understand for everyone, we will present it as an ‘evidence map’. This uses a computer software package to create an interactive picture which can be used to explore the findings at different levels of detail. _x000D_ _x000D_ We will share our findings widely, using a plan developed with the SG. The SG will discuss the implications of this project, and will contribute to project reports and summaries. An easy to access summary will be distributed widely to paediatric, community, school and voluntary sector organisations.

Lay abstract: RESEARCH QUESTION_x000D_ What are the most effective interventions, and combinations and sequences of interventions, for childhood chronic functional constipation (CFC), and how can they best be implemented?_x000D_ _x000D_ BACKGROUND_x000D_ Constipation affects 30% of children, becoming chronic in 1/3 cases. There are a range of interventions available, but the optimal strategy for combining and implementing interventions remains unclear._x000D_ _x000D_ METHODS_x000D_ A pragmatic mixed-method design, with input from a stakeholder group (SG):_x000D_ _x000D_ 1. SCOPING REVIEW using Arksey & O’Malley framework. _x000D_ Eligibility criteria: any research including participants <18years with clinical diagnosis of CFC; any intervention(s) for CFC._x000D_ Search methods: comprehensive electronic database search (Medline, CENTRAL, Embase, CINAHL, PsycINFO, AMED, NHS EED, Epistemonikos, Dare, HTA, CDSR), plus others; from October 2011. Two independent reviewers will apply selection criteria._x000D_ Data charting: pre-defined data extracted, checked by 2nd reviewer. Study type, aim, outcomes, and interventions categorised by 2 reviewers._x000D_ Data synthesis: data summarised in tables, visual maps/figures, logic model._x000D_ _x000D_ 2. SYSTEMATIC REVIEW OF EFFECTIVENESS_x000D_ Eligibility criteria: as above, plus evidence of interventions/intervention combinations as prioritised by SG._x000D_ Study selection: a hierarchical approach, for each intervention including: any high quality systematic reviews (updating searches if >12 months previous); if no eligible review - randomised controlled trials (RCTs); and if no RCTs – other primary research studies. All studies focussed on cost-effectiveness will be included._x000D_ Data extraction: will include– aim, methods, results. Interventions described using TIDieR framework._x000D_ Quality appraisal: risk of bias assessed by 2 independent reviewers using ROBIS, ROB-2, or ROBINS-I._x000D_ Data analysis: pair-wise meta-analyses to calculate effect sizes, with exploration of heterogeneity. Network plots to summarise direct evidence; network meta-analysis to combine direct and indirect evidence if transitivity assumption is not violated. Sensitivity analyses to examine intervention components._x000D_ Data synthesis: key findings summarised and GRADE approach used to judge confidence in findings. Synthesis of economic evaluations presented separately. Logic model further defined._x000D_ _x000D_ 3. SYSTEMATIC REVIEW OF FACTORS AFFECTING IMPLEMENTATION_x000D_ Search methods: studies identified from the scoping review._x000D_ Data extraction: barriers and facilitators coded using inductive and deductive coding. PROGRESS-plus framework applied for equity data. _x000D_ Quality appraisal: as above._x000D_ Data synthesis: narrative synthesis, with tables, figures and logic model._x000D_ _x000D_ PATIENT & PUBLIC INVOLVEMENT_x000D_ A SG, comprising people with lived experience of childhood CFC, informal carers, health professionals and charity representatives, will co-produce an intervention taxonomy and logic model; agree most important interventions, outcomes, clinical implications; and guide knowledge translation. _x000D_ _x000D_ OUTPUTS & ANTICIPATED IMPACT _x000D_ Findings from review components will be integrated to produce an interactive digital map, summarising evidence and highlighting gaps. A SG co-produced dissemination strategy will agree impact goals. Impact will include optimal treatment of children with CFC, reducing chronicity and long-term implications._x000D_ _x000D_ TIMELINE_x000D_ 17-months: scoping review (months 1-8), focussed systematic reviews (months 3-14), integration of findings (months 13-17)

Research activity codes: 6.1 PHARMACEUTICALS;6.6 PSYCHOLOGICAL AND BEHAVIOURAL
Health categories: ORAL AND GASTROINTESTINAL