2025, Vol. 7, Issue 2, Part D
Adult-onset rectal hypoganglionosis: A rare cause of long-standing constipation in a 27-year-old female following appendectomy - a surgical case report
Author(s): Mohamed G El Madawy
Abstract: Background: Hypoganglionosis is a rare disorder of enteric innervation characterized by a decreased number of myenteric ganglion cells. It typically presents in childhood, while adult-onset cases are uncommon and often delayed in diagnosis.
Case Presentation: A 27-year-old female, no chronic medical illnesses, presented with progressive, refractory constipation that began after an appendectomy at the age of 18. Over the years, her bowel movements became infrequent until she developed complete stool retention for 40 days prior to presentation, though flatus passage was maintained. Imaging revealed dolichocolon and a short, funnel-shaped distal segment on contrast enema. Anorectal manometry showed increased rectal compliance and paradoxical anal contraction. Histopathology confirmed severe hypoganglionosis involving the rectum and sigmoid colon. Surgical resection with colo-anal anastomosis led to full symptomatic resolution.
Conclusion: Adult-onset rectal hypoganglionosis is rare and frequently misdiagnosed. Full-thickness biopsy remains the diagnostic gold standard. Surgical resection of the affected segment results in excellent functional outcomes when the hypoganglionic segment is completely excised.
DOI: 10.22271/27081494.2025.v7.i2d.224Pages: 238-241 | Views: 306 | Downloads: 70Download Full Article: Click Here
How to cite this article:
Mohamed G El Madawy.
Adult-onset rectal hypoganglionosis: A rare cause of long-standing constipation in a 27-year-old female following appendectomy - a surgical case report. Int J Case Rep Surg 2025;7(2):238-241. DOI:
10.22271/27081494.2025.v7.i2d.224
