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A rare presentation of Rapunzel syndrome with multiple small bowel intussusceptions

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초록

Rapunzel syndrome is caused by gastric trichobezoar with extended tail and small bowel obstruction. Patients with gastric trichobezoar can be asymptomatic until the bezoar increases in size. We report a case of a girl who visited the emergency department with abdominal pain. She was finally diagnosed with Rapunzel syndrome that causes multiple small bowel intussusceptions associated with trichophagia. Surgery was needed to reduce the multiple intussusceptions, and to remove the large trichobezoar. This case highlights to consider the possibility of Rapunzel syndrome when diagnosing the main cause of intussusceptions.

Abstract

Rapunzel syndrome is caused by gastric trichobezoar with extended tail and small bowel obstruction. Patients with gastric trichobezoar can be asymptomatic until the bezoar increases in size. We report a case of a girl who visited the emergency department with abdominal pain. She was finally diagnosed with Rapunzel syndrome that causes multiple small bowel intussusceptions associated with trichophagia. Surgery was needed to reduce the multiple intussusceptions, and to remove the large trichobezoar. This case highlights to consider the possibility of Rapunzel syndrome when diagnosing the main cause of intussusceptions.

참고문헌 (10)

  1. Pandey A, "Simultaneous occurrence of jejuno-jejunal and ileo-ileal intussusception in a child: a rare occurrence", BMJ Case Rep, 2011, pp.bcr0820103~ (2011)
  2. Vaughan ED Jr, "The Rapunzel syndrome. An unusual complication of intestinal bezoar", Surgery, 63, pp.339~343 (1968)
  3. Naik S, "Rapunzel syndrome reviewed and redefined", Dig Surg, 24, pp.157~161 (2007)
  4. Gupta R, "Rapunzel syndrome and its variants in pediatric patients: our experience", Arch Int Surg, 4, pp.152~157 (2014)
  5. Prasanna BK, "Rapunzel syndrome: a rare presentation with multiple small intestinal intussusceptions", World J Gastrointest Surg, 5, pp.282~284 (2013)
  6. Ha SC, "A rare case of Rapunzel syndrome with multiple small bowel intussusceptions and bowel obstruction", Formos J Surg, 49, pp.67~69 (2016)
  7. Faria AP, "The Rapunzel syndrome: a case report: trichobezoar as a cause of intestinal perforation", J Pediatr (Rio J), 76, pp.83~86 (2000)
  8. Anantha RV, "Trichobezoar causing intussusception: youngest case of Rapunzel syndrome in a boy in North America", J Pediatr Surg Case Rep, 1, pp.e11~e13 (2013)
  9. Benatta MA., "Endoscopic retrieval of gastric trichobezoar after fragmentation with electrocautery using polypectomy snare and argon plasma coagulation in a pediatric patient", Gastroenterol Rep (Oxf), 4, pp.251~253 (2016)
  10. Kim SC, "A case report: large trichobezoar causing Rapunzel syndrome", Medicine (Baltimore), 95, pp.e3745~ (2016)

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