Andrew R Barclay, Susanna Meade, Catherine Richards, Timothy Warlow, Daniel E Lumsden, Charlie Fairhurst, Catherine Paxton, Katharine Forrest, Santosh R Mordekar, David Campbell, Julian Thomas, Michelle Brooks, Gregor M Walker, Osvaldo Borrelli, Helen Wells, Susie Holt, Shoana Quinn, Yi Fan Liang, Mohamed Mutalib, Elena Cernat, Alex C H Lee, Claire Teresa Lundy, Fiona McElligott, Jo Griffiths, Paul Eunson, Haidee Norton, Lisa Whyte, Mark A Samaan, Sue Protheroe
{"title":"Definition, investigation and management of gastrointestinal dystonia in children and young people with neurodisability.","authors":"Andrew R Barclay, Susanna Meade, Catherine Richards, Timothy Warlow, Daniel E Lumsden, Charlie Fairhurst, Catherine Paxton, Katharine Forrest, Santosh R Mordekar, David Campbell, Julian Thomas, Michelle Brooks, Gregor M Walker, Osvaldo Borrelli, Helen Wells, Susie Holt, Shoana Quinn, Yi Fan Liang, Mohamed Mutalib, Elena Cernat, Alex C H Lee, Claire Teresa Lundy, Fiona McElligott, Jo Griffiths, Paul Eunson, Haidee Norton, Lisa Whyte, Mark A Samaan, Sue Protheroe","doi":"10.1136/archdischild-2024-327551","DOIUrl":null,"url":null,"abstract":"<p><strong>Background: </strong>Children and young people with severe neurodisabling conditions (CYPSND)experience severe functional gastrointestinal symptoms and dependence on artificial nutrition. 'Gastrointestinal dystonia' (GID) has been applied by clinicians when symptoms become debilitating and potentially life-limiting. Evidence is lacking regarding the definition and appropriate management of GID.</p><p><strong>Methods: </strong>We therefore assembled a RAND appropriateness panel. We performed a systematic review, created an online survey and distributed this to a panel of 27 experts from five stakeholder groups from 13 UK specialist centres across the British Isles (gastroenterology, neurology/neurodisability, surgery, palliative care and allied health professionals). A Disagreement Index ≥1 indicated disagreement.</p><p><strong>Findings: </strong>The panel rated the appropriateness of 250 statements covering the following in GID: definition, clinical evaluation, nutritional assessment/feeding strategies, investigations, medications and prescribing, surgical interventions, safeguarding, palliative care and ethics. Agreement was reached except in selected statements regarding uncommon diagnostic features. There was uncertainty in specific clinical scenarios regarding: investigation, the use of blenderised diet, certain pharmacological agents and surgical interventions. The only intervention deemed inappropriate was antireflux surgery in the context of GID and gastrointestinal dysmotility without reflux disease. The remaining statements (198) were considered appropriate.</p><p><strong>Interpretation: </strong>We present a comprehensive review, agreement on the definition of GID and recommendations on management pathways agreed by a selected panel of multidisciplinary experts. Clear diagnostic criteria will enable important epidemiological work to record outcomes for this complex patient group. Identifying the associated morbidity, burden of care and mortality will help advocate for appropriate health resources and support to carers and families.</p>","PeriodicalId":8150,"journal":{"name":"Archives of Disease in Childhood","volume":" ","pages":"742-750"},"PeriodicalIF":3.2000,"publicationDate":"2025-08-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Archives of Disease in Childhood","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1136/archdischild-2024-327551","RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q1","JCRName":"PEDIATRICS","Score":null,"Total":0}
引用次数: 0
Abstract
Background: Children and young people with severe neurodisabling conditions (CYPSND)experience severe functional gastrointestinal symptoms and dependence on artificial nutrition. 'Gastrointestinal dystonia' (GID) has been applied by clinicians when symptoms become debilitating and potentially life-limiting. Evidence is lacking regarding the definition and appropriate management of GID.
Methods: We therefore assembled a RAND appropriateness panel. We performed a systematic review, created an online survey and distributed this to a panel of 27 experts from five stakeholder groups from 13 UK specialist centres across the British Isles (gastroenterology, neurology/neurodisability, surgery, palliative care and allied health professionals). A Disagreement Index ≥1 indicated disagreement.
Findings: The panel rated the appropriateness of 250 statements covering the following in GID: definition, clinical evaluation, nutritional assessment/feeding strategies, investigations, medications and prescribing, surgical interventions, safeguarding, palliative care and ethics. Agreement was reached except in selected statements regarding uncommon diagnostic features. There was uncertainty in specific clinical scenarios regarding: investigation, the use of blenderised diet, certain pharmacological agents and surgical interventions. The only intervention deemed inappropriate was antireflux surgery in the context of GID and gastrointestinal dysmotility without reflux disease. The remaining statements (198) were considered appropriate.
Interpretation: We present a comprehensive review, agreement on the definition of GID and recommendations on management pathways agreed by a selected panel of multidisciplinary experts. Clear diagnostic criteria will enable important epidemiological work to record outcomes for this complex patient group. Identifying the associated morbidity, burden of care and mortality will help advocate for appropriate health resources and support to carers and families.
期刊介绍：
Archives of Disease in Childhood is an international peer review journal that aims to keep paediatricians and others up to date with advances in the diagnosis and treatment of childhood diseases as well as advocacy issues such as child protection. It focuses on all aspects of child health and disease from the perinatal period (in the Fetal and Neonatal edition) through to adolescence. ADC includes original research reports, commentaries, reviews of clinical and policy issues, and evidence reports. Areas covered include: community child health, public health, epidemiology, acute paediatrics, advocacy, and ethics.
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