Trichobezoar presenting with the comma signin Rapunzel Syndrome: a case report and literature review
IntroductionRapunzel Syndrome is an uncommon presentation of trichobezoar, involving strands of swallowed hair extending as a tail through the small intestine, beyond the stomach. This was first described in 1968 and until 2003 only thirteen cases have been reported.Case PresentationA 55-year old man of East Indian descent presented to the surgical team via the emergency department in Trinidad, West Indies, with an acute abdomen and small bowel obstruction.
He had a recent psychiatric history and patchy alopecia as well as a family history of schizophrenia. A supine abdominal radiograph revealed a small bowel obstruction as well as an unusual air shadow in the left hypochondrium in the region of the duodeno-jejunal flexure associated with the stomach bubble, which resembled a 'comma'.
At laparotomy, the stomach and third part of the duodenum were distended and contained a large mass of dark, foul-smelling hair that occupied the stomach, crossed the pylorus and extended into the small bowel. A retroperitoneal perforation of the third part of duodenum was found and repaired.
The large trichobezoar was removed via a gastro-enterotomy and the patient made a slow, but complete recovery.
Conclusions: We wish to report another case of Rapunzel Syndrome and describe an unusual radiologic sign associated with a retroperitoneal perforation of the third part of duodenum - the comma sign.
To the best of our knowledge this is the first reported case of a spontaneous retroperitoneal perforation of the third part of duodenum associated with Rapunzel Syndrome.
Author: Shiva Dindyal, Neel Bhuva, Sanjay Dindyal, Jimmy Ramdass and Vijay Narayansingh
Credits/Source: Cases Journal 2008, 1:286
Published on: 2008-10-31
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