A Case of Megacolon: An Adult Presentation of Hirschsprung's Disease?

1Tufts University School of Medicine, Boston, MA

Meeting: Hospital Medicine 2011, May 10-13, Dallas, Texas.

Abstract number: 239

Case Presentation:

C.M. is a 38‐year‐old man who presented to the hospital with severe abdominal distention that developed over 1 month. Four days prior to admission, he had some diarrhea. Past medical history included constipation requiring laxatives since age 6. There was no recent antibiotic use. There was no family history of gastrointestinal disorders. Physical examination revealed the patient was afebrile with normal vital signs. He was a thin man with a markedly distended abdomen, which was nontender to palpation, and bowels sounds were present. Laboratory data included a normal white blood cell count with a normal differential. Chemistries were unremarkable except for a potassium of 3.5. A CT scan demonstrated a marked dilatation of the colon, with the sigmoid colon and rectum measuring 22 cm in diameter, and the descending colon measuring 6.6 cm. There was no evidence of sigmoid volvulus, obstructive mass, or transition point in the bowel. C.M. was admitted to the hospitalist service. GI performed a colonoscopy, which did not demonstrate evidence of pseudomembranes, ischemic colitis, or mechanical obstruction. His distention failed to improve after colonoscopy. The hospitalist requested a Clostridium difficile toxin assay, which came back positive on the second hospital day. Vancomycin retention enemas, oral vancomycin, and intravenous metronidazole were started, with marked improvement of his distention by the fourth day. He was discharged in a stable condition on oral vancomycin on the fifth day.

Scout film from the patient's CT scan demonstrating the megacolon and megarectum.

Discussion:

It is well known that C. difficile colitis can cause a wide spectrum of disease, including toxic megacolon. However, a literature review suggests it is highly unusual to see a 22‐cm sigmoid colon without systemic signs of fulminant colitis. The history of constipation since age 6 suggests a possible underlying disorder of intestinal motility. Hirschsprung's disease (HD) is characterized by a lack of ganglion cells in the rectum, which can extend into the sigmoid colon. HD is usually diagnosed in infancy but can sometimes present in young adulthood. C. difficile colitis superimposed on congenital HD could possibly cause this amount of sigmoid–rectal dilatation. A repeat colonoscopy was recommended as an outpatient to obtain a full‐thickness rectal biopsy.

Cross‐section CT scan demonstrating the 22‐cm sigmoid colon.

Conclusions:

The purpose of reporting this case is to promote awareness that C. difficile colitis can present as a megacolon without any signs of toxic disease. A literature review does not demonstrate a similar presentation; thus, it is possible that an underlying rare disorder such as HD is also complicating the picture.

Disclosures:

J. Benedetto ‐ none

To cite this abstract:

Benedetto J. A Case of Megacolon: An Adult Presentation of Hirschsprung's Disease?. Abstract published at Hospital Medicine 2011, May 10-13, Dallas, Texas. Abstract 239. https://www.shmabstracts.com/abstract/a-case-of-megacolon-an-adult-presentation-of-hirschsprungs-disease/. Accessed December 10, 2018.

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