Abstract
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.ConclusionWe 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.
Highlights
Rapunzel Syndrome is an uncommon presentation of trichobezoar, involving strands of swallowed hair extending as a tail through the small intestine, beyond the stomach
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
Summary
We report this further case of Rapunzel Syndrome associated with a small bowel obstruction. We acknowledge the fact that the 'comma sign' may apply to any patient with a retroperitoneal perforation of the third part of the duodenum. This by itself is quite a rare occurrence and when identified on a plain supine abdominal radiograph should alert the clinician to rare pathology such as tumour, foreign body or trichobezoar. We recommend closer attention be paid to the appearance of this sign on a plain supine abdominal radiograph in order to facilitate greater recognition of retroperitoneal perforations in this region and to correlate the findings with the history in order to diagnose bezoars of the Rapunzel Syndrome type pre-operatively.
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