{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,4]],"date-time":"2026-09-04T00:16:24Z","timestamp":1788480984636,"version":"build-2803163510"},"reference-count":84,"publisher":"Oxford University Press (OUP)","issue":"6","license":[{"start":{"date-parts":[[2025,11,4]],"date-time":"2025-11-04T00:00:00Z","timestamp":1762214400000},"content-version":"vor","delay-in-days":5,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2025,10,30]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>There has been an increasing number of single-centre studies describing the long-term outcomes of patients with Hirschsprung disease. This study aimed to systematically review the literature on long-term bowel, urological, and sexual functional outcomes, fertility and quality of life in adults with Hirschsprung disease.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>A PROSPERO-registered systematic review of the English literature was conducted for studies published up to July 2025 that reported functional outcomes beyond childhood (\u226516 years) for patients who had undergone surgery for Hirschsprung disease. Centres were contacted individually for secondary analyses of patient-level data on bowel function score, Gastrointestinal Quality of Life Index, and Short Form 36 questionnaire. Data were analysed and compared with those from healthy controls in the studies retrieved and from a reference healthy population. Hirschsprung disease clinical and surgical variables were correlated with these outcomes of interest in a patient-level analysis.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Fifty-three manuscripts fulfilled the inclusion criteria of 4277 papers retrieved. Patients with Hirschsprung disease had a greater likelihood of constipation (odds ratio 9.27, 95% confidence interval (c.i.) 4.78 to 18.06) and soiling (odds ratio 2.76, 1.96 to 3.89) compared with healthy controls. They scored lower on the Gastrointestinal Quality of Life Index (mean difference \u22125.21, 95% c.i. \u22129.53 to \u22120.89; P = 0.020). There were no significant differences in Short Form 36 domain scores except for physical functioning (mean difference \u22126.30, \u22128.74 to \u22123.87; P &amp;lt; 0.001). At a patient level, longer-segment disease (P &amp;lt; 0.001) and redo pull-through surgery (P = 0.002) were associated with a poorer bowel function score. Short form 36 scores were lower in women across six of eight domains; similarly, Gastrointestinal Quality of Life Index scores were lower in women (P &amp;lt; 0.001) and in patients with longer-segment disease (P &amp;lt; 0.001).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>Among patients with Hirschsprung disease, women, those with longer-segment disease, and patients who underwent redo surgery may be at risk of poorer quality of life.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1093\/bjsopen\/zraf107","type":"journal-article","created":{"date-parts":[[2025,8,5]],"date-time":"2025-08-05T11:33:35Z","timestamp":1754393615000},"source":"Crossref","is-referenced-by-count":7,"title":["Long-term effects of Hirschsprung disease in adults: meta-analysis and patient-level regression study"],"prefix":"10.1093","volume":"9","author":[{"given":"Marta","family":"de Andres Crespo","sequence":"first","affiliation":[{"name":"Institute of Child Health, University College London , London ,","place":["UK"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Cornelia","family":"Bystr\u00f6m","sequence":"additional","affiliation":[{"name":"Department of Pediatric Surgery, Karolinska Institutet , Stockholm ,","place":["Sweden"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Athanasios","family":"Tyraskis","sequence":"additional","affiliation":[{"name":"Specialist Neonatal and Paediatric Surgery, Great Ormond Street Hospital , London ,","place":["UK"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Annika","family":"Mutanen","sequence":"additional","affiliation":[{"name":"Department of Pediatric Surgery, New Children\u2019s Hospital , Helsinki ,","place":["Finland"]},{"name":"Department of Pediatric Surgery, University of Helsinki , Helsinki ,","place":["Finland"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5052-9944","authenticated-orcid":false,"given":"Pernilla","family":"Stenstr\u00f6m","sequence":"additional","affiliation":[{"name":"Department of Pediatric Surgery, Lund University , Lund ,","place":["Sweden"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Esther","family":"Hartman","sequence":"additional","affiliation":[{"name":"Department of Medical and Clinical Psychology, Tilburg University , Tilburg ,","place":["the Netherlands"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Johan","family":"Danielsson","sequence":"additional","affiliation":[{"name":"Institution of Women\u2019s and Children\u2019s Health, Uppsala University , Uppsala ,","place":["Sweden"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0892-9204","authenticated-orcid":false,"given":"Simon","family":"Eaton","sequence":"additional","affiliation":[{"name":"Institute of Child Health, University College London , London ,","place":["UK"]},{"name":"Department of Paediatric Surgery, Institute of Child Health , London ,","place":["UK"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paolo","family":"De Coppi","sequence":"additional","affiliation":[{"name":"Institute of Child Health, University College London , London ,","place":["UK"]},{"name":"Specialist Neonatal and Paediatric Surgery, Great Ormond Street Hospital , London ,","place":["UK"]},{"name":"Department of Paediatric Surgery, Institute of Child Health , London ,","place":["UK"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anna","family":"L\u00f6f Granstr\u00f6m","sequence":"additional","affiliation":[{"name":"Department of Pediatric Surgery, Karolinska Institutet , Stockholm ,","place":["Sweden"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tomas","family":"Wester","sequence":"additional","affiliation":[{"name":"Department of Pediatric Surgery, Karolinska Institutet , Stockholm ,","place":["Sweden"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6016-6334","authenticated-orcid":false,"given":"Mikko","family":"Pakarinen","sequence":"additional","affiliation":[{"name":"Department of Pediatric Surgery, New Children\u2019s Hospital , Helsinki ,","place":["Finland"]},{"name":"Department of Pediatric Surgery, University of Helsinki , Helsinki ,","place":["Finland"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joe","family":"Curry","sequence":"additional","affiliation":[{"name":"Specialist Neonatal and Paediatric Surgery, Great Ormond Street Hospital , London ,","place":["UK"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Stavros","family":"Loukogeorgakis","sequence":"additional","affiliation":[{"name":"Institute of Child Health, University College London , London ,","place":["UK"]},{"name":"Specialist Neonatal and Paediatric Surgery, Great Ormond Street Hospital , London ,","place":["UK"]},{"name":"Department of Paediatric Surgery, Institute of Child Health , London ,","place":["UK"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3303-6320","authenticated-orcid":false,"given":"Joseph","family":"Davidson","sequence":"additional","affiliation":[{"name":"Institute of Child Health, University College London , London ,","place":["UK"]},{"name":"Department of Paediatric Surgery, Institute of Child Health , London ,","place":["UK"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2025,11,4]]},"reference":[{"key":"2025110501265754300_zraf107-B1","doi-asserted-by":"crossref","first-page":"347","DOI":"10.1016\/j.anndiagpath.2006.03.017","article-title":"Hirschsprung\u2019s disease in a young adult: report of a case and review of the literature","volume":"10","author":"Chen","year":"2006","journal-title":"Ann Diagn Pathol"},{"key":"2025110501265754300_zraf107-B2","doi-asserted-by":"crossref","first-page":"8","DOI":"10.1177\/1093526619892351","article-title":"Diagnosis of Hirschsprung disease","volume":"23","author":"Ambartsumyan","year":"2020","journal-title":"Pediatr Dev Pathol"},{"key":"2025110501265754300_zraf107-B3","doi-asserted-by":"crossref","first-page":"1136","DOI":"10.1016\/S0022-3468(99)90585-7","article-title":"Repeat pull-through surgery for complicated Hirschsprung,s disease: indications, techniques, and 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