JMCR
Considers any original case report or case series that expands the field of general medical knowledge, and original research relating to case reports.
Submit a manuscript

Email updates

Keep up to date with the latest news and content from Cases Journal and BioMed Central.

Open Access Case Report

Trichobezoar presenting with the 'comma sign' in Rapunzel Syndrome: a case report and literature review

S Dindyal1, NJ Bhuva2*, S Dindyal3, MJ Ramdass4 and V Narayansingh5

Author Affiliations

1 Royal Cornwall Hospital NHS Trust, South West England, UK

2 Imperial College Healthcare NHS Trust, London, UK

3 Plymouth Hospital NHS Trust, South West England, UK

4 South East Thames NHS Trust, London, UK

5 General Hospital, Port-of-Spain, Trinidad, West Indies

For all author emails, please log on.

Cases Journal 2008, 1:286  doi:10.1186/1757-1626-1-286

Published: 30 October 2008

Abstract

Introduction

Rapunzel 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 Presentation

A 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.

Conclusion

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.