{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,1]],"date-time":"2026-02-01T05:17:42Z","timestamp":1769923062106,"version":"3.49.0"},"reference-count":16,"publisher":"Oxford University Press (OUP)","issue":"2","license":[{"start":{"date-parts":[[2026,1,31]],"date-time":"2026-01-31T00:00:00Z","timestamp":1769817600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2026,1,31]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Chronic idiopathic megacolon is a rare disorder characterized by persistent colonic dilatation without obstruction or secondary causes. It is usually diagnosed in childhood, often linked to Hirschsprung\u2019s disease; adult-onset cases are exceedingly rare, especially with familial predisposition. We report a man in his 30s with progressive abdominal distension, prior adolescent colectomy, and family history suggesting genetic susceptibility. After excluding secondary causes, chronic idiopathic megacolon was diagnosed. Conservative management failed, necessitating subtotal colectomy with ileorectal anastomosis, leading to complete symptom resolution. Histopathology confirmed normal ganglion cells. Adult-onset idiopathic megacolon poses diagnostic and therapeutic challenges and may have a genetic basis. Subtotal colectomy is safe and effective in refractory cases.<\/jats:p>","DOI":"10.1093\/jscr\/rjaf1037","type":"journal-article","created":{"date-parts":[[2026,1,20]],"date-time":"2026-01-20T12:25:34Z","timestamp":1768911934000},"source":"Crossref","is-referenced-by-count":0,"title":["Familial adult-onset chronic idiopathic megacolon: diagnostic and surgical challenges\u2014a case report"],"prefix":"10.1093","volume":"2026","author":[{"ORCID":"https:\/\/orcid.org\/0009-0002-0544-4797","authenticated-orcid":false,"given":"Mohamad","family":"Shbaro","sequence":"first","affiliation":[{"name":"Faculty of Medicine, American University of Beirut , PO Box 11-0236, Riad El Solh, Beirut 1107 2020 ,","place":["Lebanon"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Bassel","family":"Hafez","sequence":"additional","affiliation":[{"name":"Division of General Surgery, Department of Surgery, American University of Beirut Medical Center , PO Box 11-0236, Riad El Solh, Beirut 1107 2020 ,","place":["Lebanon"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Samer","family":"Doughan","sequence":"additional","affiliation":[{"name":"Division of General Surgery, Department of Surgery, American University of Beirut Medical Center , PO Box 11-0236, Riad El Solh, Beirut 1107 2020 ,","place":["Lebanon"]}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"286","published-online":{"date-parts":[[2026,1,31]]},"reference":[{"key":"2026013111470555000_ref1","doi-asserted-by":"publisher","first-page":"2398","DOI":"10.1007\/s10620-015-3645-5","article-title":"Clinical features and colonic motor disturbances in chronic megacolon in adults","volume":"60","author":"O'Dwyer","year":"2015","journal-title":"Dig Dis Sci"},{"key":"2026013111470555000_ref2","doi-asserted-by":"publisher","first-page":"2112","DOI":"10.3390\/diagnostics11112112","article-title":"Idiopathic megacolon\u2014short review","volume":"11","author":"Constantin","year":"2021","journal-title":"Diagnostics"},{"key":"2026013111470555000_ref3","doi-asserted-by":"publisher","first-page":"e13550","DOI":"10.1111\/nmo.13550","article-title":"Familial chronic megacolon presenting in childhood or adulthood: seeking the presumed gene association","volume":"31","author":"Camilleri","year":"2019","journal-title":"Neurogastroenterol Motil"},{"key":"2026013111470555000_ref4","doi-asserted-by":"publisher","first-page":"5634","DOI":"10.3748\/wjg.v23.i30.5634","article-title":"Acute colonic pseudo-obstruction: a systematic review of aetiology and mechanisms","volume":"23","author":"Wells","year":"2017","journal-title":"World J Gastroenterol"},{"key":"2026013111470555000_ref5","doi-asserted-by":"publisher","first-page":"203","DOI":"10.2147\/ceg.S200760","article-title":"Toxic megacolon: background, pathophysiology, management challenges and solutions","volume":"13","author":"Desai","year":"2020","journal-title":"Clin Exp Gastroenterol"},{"key":"2026013111470555000_ref6","doi-asserted-by":"publisher","first-page":"2750","DOI":"10.1007\/s10620-019-05605-7","article-title":"Chronic megacolon presenting in adolescents or adults: clinical manifestations, diagnosis, and genetic associations","volume":"64","author":"Wang","year":"2019","journal-title":"Dig Dis Sci"},{"key":"2026013111470555000_ref7","doi-asserted-by":"publisher","first-page":"G919","DOI":"10.1152\/ajpgi.00066.2021","article-title":"Visceral myopathy: clinical syndromes, genetics, pathophysiology, and fall of the cytoskeleton","volume":"320","author":"Hashmi","year":"2021","journal-title":"Am J Physiol Gastrointest Liver Physiol"},{"key":"2026013111470555000_ref8","doi-asserted-by":"publisher","first-page":"571","DOI":"10.1111\/nmo.12303","article-title":"Histopathologic findings in patients with idiopathic megacolon: a comparison between dilated and non-dilated loops","volume":"26","author":"Ohkubo","year":"2014","journal-title":"Neurogastroenterol Motil"},{"key":"2026013111470555000_ref9","doi-asserted-by":"publisher","first-page":"25","DOI":"10.1186\/s12876-018-0753-7","article-title":"Symptoms and diagnostic criteria of acquired megacolon - a systematic literature review","volume":"18","author":"Cuda","year":"2018","journal-title":"BMC Gastroenterol"},{"key":"2026013111470555000_ref10","doi-asserted-by":"publisher","first-page":"bcr2018225406","DOI":"10.1136\/bcr-2018-225406","article-title":"Bowel perforation in chronic idiopathic megarectum and megacolon","volume":"2018","author":"Anyaegbuna","year":"2018","journal-title":"BMJ Case Rep"},{"key":"2026013111470555000_ref11","doi-asserted-by":"publisher","first-page":"1136","DOI":"10.1097\/js9.0000000000000373","article-title":"The SCARE 2023 guideline: updating consensus Surgical CAse REport (SCARE) guidelines","volume":"109","author":"Sohrabi","year":"2023","journal-title":"Int J Surg"},{"key":"2026013111470555000_ref12","doi-asserted-by":"publisher","first-page":"5742","DOI":"10.3748\/wjg.v11.i36.5742","article-title":"Megacolon in adulthood after surgical treatment of Hirschsprung's disease in early childhood","volume":"11","author":"Werner","year":"2005","journal-title":"World J Gastroenterol"},{"key":"2026013111470555000_ref13","doi-asserted-by":"publisher","first-page":"9235","DOI":"10.3748\/wjg.v22.i41.9235","article-title":"Reoperation in an adult female with \u201cright-sided\u201d Hirschsprung's disease complicated by refractory hypertension and cough","volume":"22","author":"Wei","year":"2016","journal-title":"World J Gastroenterol"},{"key":"2026013111470555000_ref14","doi-asserted-by":"publisher","first-page":"e14715","DOI":"10.1111\/nmo.14715","article-title":"Chronic intestinal pseudo-obstruction in adults: a practical guide to identify patient subgroups that are suitable for more specific treatments","volume":"36","author":"Basilisco","year":"2024","journal-title":"Neurogastroenterol Motil"},{"key":"2026013111470555000_ref15","doi-asserted-by":"publisher","first-page":"1244","DOI":"10.1097\/dcr.0000000000003430","article-title":"The American Society of Colon and Rectal Surgeons clinical practice guidelines for the evaluation and management of chronic constipation","volume":"67","author":"Alavi","year":"2024","journal-title":"Dis Colon Rectum"},{"key":"2026013111470555000_ref16","doi-asserted-by":"publisher","first-page":"42","DOI":"10.1097\/mog.0000000000000492","article-title":"Eosinophilic colitis and colonic eosinophilia","volume":"35","author":"Walker","year":"2019","journal-title":"Curr Opin Gastroenterol"}],"container-title":["Journal of Surgical Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/jscr\/article-pdf\/2026\/2\/rjaf1037\/66676160\/rjaf1037.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/jscr\/article-pdf\/2026\/2\/rjaf1037\/66676160\/rjaf1037.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,1,31]],"date-time":"2026-01-31T16:47:15Z","timestamp":1769878035000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/jscr\/article\/doi\/10.1093\/jscr\/rjaf1037\/8450255"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,1,31]]},"references-count":16,"journal-issue":{"issue":"2","published-print":{"date-parts":[[2026,1,31]]}},"URL":"https:\/\/doi.org\/10.1093\/jscr\/rjaf1037","relation":{},"ISSN":["2042-8812"],"issn-type":[{"value":"2042-8812","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2026,2]]},"published":{"date-parts":[[2026,1,31]]},"article-number":"rjaf1037"}}