Trichobezoar: an unusual cause of pancreatitis in a teenager M.H. Jonkers, M.S. Oudijn, R. van Baren, D.C. Aronson Pediatric Surgical Center, Emma Kinderziekenhuis AMC, Amsterdam Case report A 14-year-old Negroid girl presented with a 3-day history of progressive nausea, vomiting, anorexia, and spasmodic pain in the upper abdomen. Her medical history showed a surgically corrected bilateral inguinal hernia at the age of 8 years. Physical examination revealed an ill girl; her body temperature was 38.6û C. The abdomen was distended with very little peristalsis; signs of peritonitis were observed. Rectal examination was normal. The initial laboratory results showed an erythrocyte sedimentation rate of 9 mm/hour, leukocytes 27.1 x 109 /L with 93 percent neutrophils, and amylase 1290 U/L. The sickle cell test was negative. All other laboratory results were normal. The abdominal X-ray was suggestive for coprostasis. Ultrasonography of the abdomen only showed a small collection of free fluid in the Douglas space. The pancreas could not be visualized. At laparoscopy, a large amount of sanguineous fluid was found in the abdomen. On the greater omentum, some calcifications were visible. The appendix, both adnexa, liver, and gallbladder were normal and no Meckels diverticulum was found. The duodenum was distended. A large firm tumor of 10 cm was distinguished in the gastric region. The precise origin of this mass could not be established. A laparotomy was performed. The mass appeared to be located in the stomach. Gastrotomy revealed a huge trichobezoar. Also in the jejunum, a long trichobezoar was found (length 40 cm). The amylase level of the abdominal fluid collection was 16000 U/L. After removal of the masses from the stomach and from the jejunum, normal gastric emptying was possible. A thorough wash-out of the peritoneal cavity was performed to remove all hair rests. After the diagnosis was established, it became evident that she was known for trichophagia years ago, but she claimed not to have eaten her hair in the last few years. The postoperative course was first complicated by a wound abscess, followed by an intra-abdominal abscess. At re-laparotomy for abscess drainage, still hairs were found intra-abdominally. She completely recovered and psychiatric care was introduced. Comment The trichobezoar had probably obstructed the pancreatic duct, which must have caused the pancreatitis. The combination of a gastric hairball and a hairtail in the jejunum is known as the Rapunzel syndrome. Rapunzel was the long haired maiden, described by the Grimm brothers in 1812, who lowered her tresses to allow Prince Charming to climb up to her prison tower to rescue her. Trichobezoar was first described in 1968 by Vaughan et al. Ever since, eleven case reports have been described in the literature.1 Not in all children who eat their hair, a bezoar will develop. The reason for bezoar formation remains unclear. The quantity and length of hair, together with altered mucosal crypts, are probably responsible for bezoar formation. Bezoars have been known to occur in animals and men. The word bezoar is probably derived from the Arabic "badzehr'which means antidote. This was the reason why bezoars were collected for medicinal use.2 Over 90 percent of the trichobezoars are found in girls younger than 20 years. Only half of the patients have a history of trichophagia.1 Trichobezoars are generally found in emotionally disturbed, mentally retarded or depressed patients. In these patients, bezoars can reoccur. Symptoms of trichobezoar are very unspecific. The patients may present with symptoms varying from mild abdominal pain, constipation, to total gastro-intestinal obstruction with pancreatitis or obstructive jaundice. The treatment is primary surgical. Pancreatitis is a rare diagnosis in children. As far as we are aware, this is the second child described in literature with an obstructive pancreatitis caused by a trichobezoar.3 References 1. G.B. Dalshaug, S.. Wainer et al. The Rapunzel Syndrome (Trichobezoar) Causing Atypical Intussusception in a Child: a Case Report. Journal of Pediatric Surgery Vol 34, No 3 1999: pp479-80. 2. E. Balik, I. Ulman. The Rapunzel Syndrome: A case report and review of the literature Eur J pediatr Surg 1993(3) 171-3. 3. Shawis R.N. , Doig C.M. Gastric bezoar associated with transient pancreatitis. Arch of Dis Child 1984; 59(10): 994-5.   TOP