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

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.

CT appearance of the rapunzel syndrome: an unusual form of bezoar and gastrointestinal obstruction

The CT appearance oa an unusual type of trichobezoar which cause the Rapunzel syndrome are described. This syndrome is characterized by intestinal obstruction secondary to a bezoar possessing a tail which extends to or beyond the ileocecal valve. Most cases of bezoar are diagnosed with plain films or barium meal, but CT may be requested in patients who present with abdominal masses. The Rapunzel syndrome may be diagnosed with a combination of the CT findings and the history.(AU)

The rapunzel syndrome: report of a case and review of the literature

The Rapunzel Syndrome, a rare manifestation of trichobezoar, occurs when bolus gastrointestinal obstruction is produced by an unusual trichobezoar with a long tail that extends to or beyond the ileocaecal valve. A five-year-old Jamaican girl presented with this abnormality and was found at laparotomy also to have an ileo-ileal intussuception. For the Rapunzel Syndrome, we recommended bezoar extraction at laparotomy via multiple enterotomies. In addition, psychiatric evaluation and therapy is essential due to the commonly associated finding of underlying emotional stress. (AU)

An unusual mixed bezoar in a child

A case report is presented of a mixed bezoar, composed of hair and carpet, occurring in a Negro toddler. The tail of the bezoar had progressed into the small bowel, giving rise to necrosis, perforation and peritonitis before removal of the mass and bowel resection led to a favourable outcome. The unusual features of the case are stressed (AU)