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TRICHOBEZOAR IN YOUNG NEPALESE GIRL WITH RAPUNZEL SYNDROME: A CASE REPORT

Trichobezoar is a gastric foreign body that is composed of the patient’s own hair in the stomach. Rapunzel syndrome is an unusual form of trichobezoar in stomach,which extends into any part of the small intestine.We are reporting the case of gastric trichobezoar with thin tail extending into the duodenum in a twenty year oldgirl. She had a history of trichophagia. She presented with epigastric pain and vomiting. Endoscopic removal was attempted but this was unsuccessful. Subsequently the patient underwent exploratory laparotomy and gastrotomy.She recovered well and was discharged with advice for psychiatric consultation. Key word: Trichobezoar, Rapunzel syndrome Received: 13 October, 2020 Accepted: 25 December, 2020 DOI: https://doi.org/10.3329/bjm.v32i1.51099 noted in the right adnexa, measures 4.7 X 4.2 X 2.5 cm.(25.5 ml.). Chest x-ray was normal. She underwent upper gastrointestinal endoscopy for further evaluation.Therstomachwaswas full of hair (Fig.1). Various accessories like Dormia Basket,Crocodile forceps, snare were used to remove hair. Even Argon plasma coagulation probe (linear) was used to fragment hair (Fig.2,3).A small amount of fragment of hair could be removed (Fig.4). After endoscopic retrieval was unsuccessful, surgical intervention was done with laparotomy. Intraoperatively a firm mass was felt inside the stomach.Approximately ten centimeters of anterior gastrotomy was done, and a 14 X 7cm.of firm consolidated mass of hair was found inside the Fig.-1 : Endoscopic view of a large trichobezoar within the stomach. Bangladesh J Medicine 2021; 32 : 69-72 stomach extending upto first and second part of duodenum(Fig.5 ).The specimen was delivered intact(Fig.5),no other hairy mass was felt upto the second part of duodenum. Patient was allowed oral intake on the third day.Five days later she was discharged.Then patient was referred to psychiatry hospital for further evaluation and management to prevent the recurrence. Fig.-2 : Fragmentation wih Argon plasma coagulation (linear probe) Fig.-3 : Trichobezoar within the stomach. Fig.-4 : Endoscopic removal of fragments of trichobezoar. Fig.-5 : Image of operative specimen of large trichobezoar. Discussion: A bezoar is a conglomeration of food or foreign material which accumulate and remain within the gastrointestinal tract.The word “bezoar”comes from the Arabic word “bedzehr” or the Persian word “Padzhar”meaning “protecting against a poison”.In history bezoars from animal guts were used as precious stones,antidotes to poisons.It is the part of traditional Chinese medicine3,7,8.They have been classified into four groups: phytobezoars(caused by vegetables), trichobezoars (caused by hair),lactobezoars(caused by milk), miscellaneous (caused by medication; pharmacobezoars,tissue papers,sand or fungus).The first reference to a bezoar in a human was in 1779 during an autopsy of a patient who died from gastric perforation and peritonitis3,8. Rapunzel was the name of the maiden in the “Grimm brothers”fairy tale in 1812,whose long hair flowed out of her prison tower allowing her prince to rescue her 9. Rapunzel syndrome was first described in literature by Vaughan et al.in 19688.Fewer than a hundred cases of Rapunzel syndrome have been reported in the literature since the initial description. In Rapunzel syndrome,the trichobezoar extends through the Trichobezoar in young Nepalese girl with Rapunzel syndrome: A case report BJM Vol. 32 No. 1

Her physical examination revealed stable vitals.She was thin built with pallor on examination and she was afebrile. Her hair was short and broken. There was mild tenderness in epigastric region. Cardiorespiratory examination was unremarkable.
She underwent upper gastrointestinal endoscopy for further evaluation.Therstomachwaswas full of hair ( Fig.1). Various accessories like Dormia Basket,Crocodile forceps, snare were used to remove hair. Even Argon plasma coagulation probe (linear) was used to fragment hair (Fig.2,3).A small amount of fragment of hair could be removed (Fig.4).
After endoscopic retrieval was unsuccessful, surgical intervention was done with laparotomy. Intraoperatively a firm mass was felt inside the stomach.Approximately ten centimeters of anterior gastrotomy was done, and a 14 X 7cm.of firm consolidated mass of hair was found inside the stomach extending upto first and second part of duodenum (Fig.5 ).The specimen was delivered intact (Fig.5),no other hairy mass was felt upto the second part of duodenum. Patient was allowed oral intake on the third day.Five days later she was discharged.Then patient was referred to psychiatry hospital for further evaluation and management to prevent the recurrence.

Discussion:
A bezoar is a conglomeration of food or foreign material which accumulate and remain within the gastrointestinal tract.The word "bezoar"comes from the Arabic word "bedzehr" or the Persian word "Padzhar"meaning "protecting against a poison".In history bezoars from animal guts were used as precious stones,antidotes to poisons.It is the part of traditional Chinese medicine 3,7,8 .They have been classified into four groups: phytobezoars(caused by vegetables), trichobezoars (caused by hair),lactobezoars(caused by milk), miscellaneous (caused by medication; pharmacobezoars,tissue papers,sand or fungus).The first reference to a bezoar in a human was in 1779 during an autopsy of a patient who died from gastric perforation and peritonitis 3,8 .
Rapunzel was the name of the maiden in the "Grimm brothers"fairy tale in 1812,whose long hair flowed out of her prison tower allowing her prince to rescue her 9. Rapunzel syndrome was first described in literature by Vaughan et al.in 1968 8 .Fewer than a hundred cases of Rapunzel syndrome have been reported in the literature since the initial description. In Rapunzel syndrome,the trichobezoar extends through the pylorus into the small intestine 8 .Most cases of trichobezoar are reported in females with traditional long hair,one reported male case who ate the hair of his sister 11 .Most patients with trichobezoar suffer from psychiatric disorder including trichotillomania (pulling out their own hair)and trichophagia (eating hair).
The formation of trichobezoar in these patients depends upon the quantity and duration of trichophagia. On an average only 1% patients with trichophagia develop trichobezoar 6,12,13. As more and more hair accumulates,a ball of hair is formed which becomes too large to escape and results in gastric atony and subsequently takes the shape of stomach 14 . The acid contents of the stomach denature hair protein and give the bezoar a jet black color 15. The most common presenting signs are abdominal pain, nausea, vomiting, obstruction, peritonitis and less commonly patients have presented with weight loss, anorexia, hematemesis and intussusception. Complications include gastric ulceration, obstructive jaundice, acute pancreatitis and gastric emphysema 6,15. Diagnostic modalities include ultrasound, computed tomography(CT). CT scan has high accuracy rate but ultrasound in such cases is very sensitive 20. .Upper gastrointestinal endoscopy remains the examination of choice in the diagnosis of intragastric trichobezoar as it allows visualizing the hair threads and trying an extraction 21. Various therapeutic modalities have been proposed to treat trichobezoar.
Gorteret al.in a retrospective review of 108 cases of trichobezoar evaluated the variable treatments tried in these cases.It was noted that 5% of attempted endoscopic removal were successful ( small trichobezoar may respond to endoscopic fragmentation and vigorous lavage), 75% of attempted laparoscopic surgical extractionwere successful. Laparotomy followed by gastrotomy was 99 % successful 22,23. Specialized bezotomes (device that pulverize bezoars ) and bezotriptors ( devices that fragment bezoars by acoustic waves ) have been used to fragment large and solid trichobezoars 21,23. Various other methods like extracorporal shock wave lithotripsy, intra-gastric administration of enzymes ( pancreatic lipase,cellulose) and medications metoclopramide, acetylcysteine, demonstrate have been used with varying success 24.

Conclusion:
Rapunzel syndrome should be considered as differential diagnosis in young girl presenting with abdominalpain,vomiting and anaemia.Early diagnosis can prevent these complications. Psychiatric evaluation,counselling and treatment are helpful in preventing recurrence. Trichobezoar often coexist withpsychiatric illness.