ArticleThe American journal of case reports2018
A Rare Case of Adult-Onset Rectosigmoid Hypoganglionosis.
Article in The American journal of case reports, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 10 papers.
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Who cites it
10 citing papers in PubMed, 16 citations in OpenAlex.
- Abdominal compartment syndrome with colonic hypoganglionosis and massive colonic distension in a young adult: A case report.World journal of clinical cases · 2025Article
- Pain management in a patient with intestinal failure in the palliative care setting: case report.BMC palliative care · 2025Article
- Suspicion diagnostic of Hirschsprung's disease in an adult intraoperatively: A case report.Trauma case reports · 2024Article
- Chronic intestinal pseudo-obstruction due to adult-onset acquired isolated hypoganglionosis with muscular atrophy in the small intestine: a case report and review of literature.Clinical journal of gastroenterology · 2024Review
- Sigmoid Volvulus as a Clinical Manifestation of Acquired Colonic Hypoganglionosis: A Case Report.Cureus · 2023Article
- Adult patients with allied disorders of Hirschsprung's disease in emergency department: An 11-year retrospective study.World journal of gastrointestinal surgery · 2022Article
- Rare case of adult intestinal hypoganglionosis and review of the literature.Clinical journal of gastroenterology · 2021Review
- Unusual case of cholecystocolonic fistula secondary to megabowel.BMJ case reports · 2020Article
- A case of severe megacolon due to acquired isolated hypoganglionosis after low anterior resection for lower rectal cancer.Clinical journal of gastroenterology · 2020Article
- Atypical Presentation of Acquired Hypoganglionosis in a Patient Taking Clozapine.Case reports in gastrointestinal medicine · 2020Article
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Authors and funding
5 authors at 1 institution in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
BACKGROUND Intestinal hypoganglionosis is very rare and accounts for 3% to 5% of all classified congenital intestinal innervation disorders. Isolated hypoganglionosis of the colon is a particularly rare form of the disease, and differential diagnosis includes association with Hirschsprung's disease and chronic intestinal pseudo-obstruction (CIPO) related to visceral myopathies. Most cases are diagnosed at an early age or in childhood with only a few cases reported in adults. CASE REPORT We report a case of isolated hypoganglionosis of the rectum and sigmoid presenting as an emergency with acute intestinal obstruction in a 20-year-old male patient. A history of chronic constipation was reported since childhood, but this condition had never been investigated. A preoperative CT scan showed a megasigmoid and megarectum. A Hartmann's procedure was performed. The patient made a slow recovery and was discharged on the 12th postoperative day in good condition. Histology showed features consistent with isolated hypoganglionosis, and a full thickness rectal biopsies taken 2 months later confirmed the diagnosis. CONCLUSIONS Isolated hypoganglionosis in an adult is very rare, and a high index of suspicion is warranted in young patients with a history of chronic constipation to avoid delayed presentation as an emergency.
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