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Rare case of adult intestinal hypoganglionosis and review of the literature

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Abstract

Intestinal hypoganglionosis is a rare condition in adults. We report a case of intestinal hypoganglionosis in the mid-distal transverse colon to splenic flexure in a 65-year-old female patient presenting with altered bowel habit and abdominal distension, and reviewed the current literature on this topic. Our patient had a medical history of neurofibromatosis type 1. A preoperative computed tomography (CT) scan demonstrated a grossly dilated transverse colon without obstruction. A laparotomy for subtotal colectomy was performed, with histopathology demonstrating intestinal hypoganglionosis.

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Authors and Affiliations

  1. Colorectal Surgery Unit, Dandenong Hospital, Monash Health, 135 David Street, Dandenong, VIC, Australia

    Alice Lee, Thomas Surya Suhardja & James Tow-Hing Lim

  2. Department of Surgery, School of Clinical Sciences at Monash Health, Monash University, Clayton, VIC, Australia

    Thomas Surya Suhardja

  3. Department of Anatomical Pathology, Monash Medical Centre, Monash Health, Clayton, VIC, Australia

    Ian Simpson

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  1. Alice Lee
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Correspondence to Thomas Surya Suhardja.

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Lee, A., Suhardja, T.S., Simpson, I. et al. Rare case of adult intestinal hypoganglionosis and review of the literature. Clin J Gastroenterol 14, 599–607 (2021). https://doi.org/10.1007/s12328-021-01342-5

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  • DOI: https://doi.org/10.1007/s12328-021-01342-5

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