Published online: 2019-09-26

Case Report

Trichophagia along with trichobezoar in the absence of Aseem Mehra, Ajit Avasthi, Vikas Gupta1, Sandeep Grover Department of and Surgery1, Post Graduate Institute of Medical Education and Research (PGIMER), Chandigarh, India

ABSTRACT Trichobezoars are rarely described in the absence of trichotillomania. In this report we present a case of trichobezoar associated with in the absence of trichotillomania. A 16‑year‑old girl presented to surgery outpatient with complaints of pain in abdomen and vomiting for the last 6 months. Physical examination revealed a 14 × 16 cm firm, tender, mass with smooth surface, irregular margins, which was mobile with respiration. Ultrasound abdomen revealed a bizarre lesion in the right upper and middle abdomen suggestive of gastric bezoars. Upper gastrointestinal endoscopy did not reveal any abnormality in the esophagus and showed a large mobile mass in the . In view of trichobezoar, psychiatry consultation was sought. Exploration of history revealed that the patient was eating and clay since early childhood. As per patient she would eat hairs thrown by others. She would like the taste of and had strong persistent desire to eat hair and would go out searching for the same. At times she would also eat clay. However, she denied of pulling her own hairs. Physical examination of and other body parts did not show any evidence of alopecia or pulling of hair/. She was managed surgically and was counseled about the consequences of eating hairs and clays and was encouraged not to eat hair. To conclude our case suggests that patients can have trichobezoar and trichophagia even in the absence of trichotillomania. Key words: Trichophagia, trichotillomania, trichobezoar

Introduction Case Report

Trichophagia is understood as a A 16‑year‑old single girl, with 10 years of formal involving eating hairs, usually of own. Trichobezoars education, from rural background presented to surgery are concretions of mostly hair, which accumulate in the outpatient with complaints of pain in abdomen and gastrointestinal tract. Trichobezoars are often associated vomiting for the last 6 months. She would have frequent with psychiatry disorder like trichotillomania, mental bouts of vomiting, frequency of which increased in retardation, emotional disturbances and/or .[1] Most the last few days prior to presentation. On physical of the cases of trichobezoars are described in adolescent examination, she was found to have a mobile mass girls/young females.[2,3] of 14 × 16 cm size in the upper abdomen, which was tender, with smooth surface and irregular margins. However, trichophagia and trichobezoars are rarely Ultrasound abdomen revealed a bizarre lesion in the described in the absence of trichotillomania.[4] In this case right upper and middle abdomen suggestive of gastric report we describe a case of trichobezoar associated with bezoars. Upper gastrointestinal endoscopy did not reveal trichophagia in the absence of trichotillomania. any abnormality in the esophagus and a large mobile mass made of hairs was seen in the stomach. Based on Access this article online the findings of ultrasound and upper gastrointestinal Quick Response Code: endoscopy, a diagnosis of trichobezoar was considered Website: and psychiatry consultation was sought. Psychiatric www.ruralneuropractice.com evaluation revealed that the girl was eating hairs and clay since early childhood. She would eat the hairs thrown

DOI: here and there by her mother, other family members and 10.4103/0976-3147.145204 neighbors. The patient would like the taste of hairs and had a strong and persistent desire to eat hairs. She would

Address for correspondence: Dr. Sandeep Grover, Department of Psychiatry, Post Graduate Institute of Medical Education and Research, Chandigarh ‑ 160 012, Punjab and Haryana, India. E‑mail: [email protected]

Journal of Neurosciences in Rural Practice | 2014 | Vol 5 | Supplement 1 S55 Mehra, et al.: Trichobezoar in the absence of trichotillomania often go out searching for the same. Additionally, at times pylorus and involves the duodenum or further lower she would also eat clay. The description of the patient gastrointestional tract, it is known as Rapunzel was corroborated by the mother too; who described that syndrome.[6] In terms of pathogenesis of trichobezoar, it despite persistence reprimands, the patient persisted is believed that because of its smooth surface the hairs to eat hairs and occasionally would eat clay. However, do not move forward with the peristaltic movement and the patient denied pulling out her own hairs. Physical resultantly get accumulated.[7] examination of scalp and other body parts did not reveal any evidence of alopecia or pulling of hair/short hair. Trichophagia and trichobezoar are abundantly described Past history did not reveal any evidence of depressive in surgical literature.[1‑15] In few cases, no evidence disorders, obsessive compulsive disorder, other of trichotillomania was found in patients presenting disorders, eating disorders, emotional disturbances, with trichobezoar.[4,9] Previous reports of trichobezoar mental sub‑normality and other impulse control have also reported co‑occurrence of eating hairs and disorders. Also, there was no family history of any mental other non‑eatable items amounting to pica.[8] Pica is illness. Investigations in the form level, understood as a clinical condition in which the subjects complete blood counts, serum electrolytes, liver function often eats non‑nutritive materials like soil, dirt, hairballs, test, renal function test, pancreatic enzymes and routine ice, paint and sand, etc., It is usually seen in the setting of stool examination did not reveal any abnormality. In low intelligence;[16] however, it can also occur in children view of large size of the mass, open laparotomy was done with normal intelligence.[16] and the mass was removed. On examination, the mass comprised of entangled hairs. A psychiatric diagnosis As trichobezoar is often associated with trichotillomania of other behavioral and emotional disorders with onset and eating of other non‑eatable items which can amount usually occurring in childhood and adolescence (Pica of to pica, a psychiatric evaluation of such patients is infancy and childhood) as per the ICD‑10 was considered. recommended. Although trichotillomania itself is The patient was counseled against eating hairs and clays and was informed about the consequences of the same. considered to be an impulse control disorder, it may Behavioral analysis revealed that the patient would have also manifest as a symptom of many other mental disorders. Accordingly detail psychiatric evaluation to urge to eat hair more so, when under . Additionally, behavioral principles with regards to management of focus on ruling trichotillomania and other psychiatric urge and impulse control were discussed. Additionally, disorders. Detail physical examination should involve the psychotherapeutic intervention focused on teaching looking for patches of in various parts of the adaptive coping skills to deal with the stressful situations. body. A patient may have pica and also trichotillomania Family members were also educated about the illness and as a manifestation of low intelligence. Accordingly if were asked to be vigilant to note the patient behavior low intelligence is suspected, the patient intelligence of eating hairs and other non‑eatable items. The patient quotient must be assessed. In view of the associated improved after the surgery and follow‑up at 6 months pica, these patients must be evaluated for . did not reveal any behavior suggestive of trichophagia Management of trichotillomania and pica involves or pica. psychoeducation, behavior therapy (habit reversal therapy in case patient has trichotillomania) and teaching Discussion adaptive coping skills. Patients with trichobezoar must be followed up regularly because of the risk of [11,12] The term “Bezoar” is understood to be derived from the recurrence. Arabic word “Bazehr” or the Persian word “Padzhar,” which are used for antidote, because traditionally stones Most of the cases of trichophagia and trichobezoar are from the stomach or intestine of various animals were described in patients with trichotillomania, which is considered to have medicinal value.[5] Different types considered to be an impulsive control disorder or other of bezoars are described in humans and according to psychiatric disorders. Index case had no psychiatric the composition are named as phytobezoar (containing history, any evidence of psychological distress or vegetable fibres), lactobezoar (milk products), specific personality traits which could be considered pharmacobezoar () and trichobezoar (hairs).[1,5] to be associated with trichophagia. The present case Additionally bezoars composed of cotton and other exemplifies that patients of trichobezoar can have isolated materials have also been described in the literature.[3] trichophagia. Hence, the absence of trichotillomania Usually the bezoars are found in the stomach, however, should not be considered to be equivalent of absence of when the tail of the bezoar extends beyond the trichophagia.

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References iron deficiency and pica: in a 5‑year‑old child iron deficiency and pica. Turk J Gastroenterol 2014;25:100‑2. 10. Gurzu S, Jung I. Gastric tricho‑wool bezoar in an 18‑year‑old girl. S Afr J 1. Gonuguntla V, Joshi DD. Rapunzel syndrome: A comprehensive review Surg 2013;51:33‑4. of an unusual case of trichobezoar. Clin Med Res 2009;7:99‑102. 11. Kırpınar I, Kocacenk T, Koçer E, Memmi N. Recurrent trichobezoar due 2. De Bakey M, Ochsner W. Bezoars and concretions: A comprehensive to trichophagia: A case report. Gen Hosp Psychiatry 2013;35:439‑41. review of the literature with an analysis of 303 collected cases and a 12. Tiwary SK, Kumar S, Khanna R, Khanna AK. Recurrent Rapunzel presentation of 8 additional cases. Surgery 1938;4:934‑63. syndrome. Singapore Med J 2011;52:e128‑30. 3. Chintamani, Durkhure R, Singh JP, Singhal V. Cotton bezoar–a rare cause 13. Jain M, Solanki SL, Bhatnagar A, Jain PK. An unusual case report of Rapunzel of intestinal obstruction: Case report. BMC Surg 2003;3:5. syndrome trichobezoar in a 3‑year‑old boy. Int J Trichology 2011;3:102‑4. 4. Tiago S, Nuno M, João A, Carla V, Gonçalo M, Joana N. Trichophagia 14. Kim JS, Nam CW. A case of Rapunzel syndrome. Pediatr Gastroenterol and trichobezoar: Case report. Clin Pract Epidemiol Ment Health Hepatol Nutr 2013;16:127‑30. 2012;8:43‑5. 15. Prasanna BK, Sasikumar K, Gurunandan U, Sreenath GS, Kate V. 5. Williams RS. The fascinating history of bezoars. Med J Aust Rapunzel syndrome: A rare presentation with multiple small intestinal 1986;145:613‑4. intussusceptions. World J Gastrointest Surg 2013;5:282‑4. 6. Memon SA, Mandhan P, Qureshi JN, Shairani AJ. Recurrent Rapunzel 16. World Health Organization. The ICD‑10 Classification of Mental and syndrome‑a case report. Med Sci Monit 2003;9:CS92‑4. Behavioural Disorders. Geneva: World Health Organization; 1992. 7. Frey AS, McKee M, King RA, Martin A. Hair apparent: Rapunzel syndrome. Am J Psychiatry 2005;162:242‑8. How to cite this article: Mehra A, Avasthi A, Gupta V, Grover S. 8. Western C, Bokhari S, Gould S. Rapunzel syndrome: A case report and Trichophagia along with trichobezoar in the absence of trichotillomania. review. J Gastrointest Surg 2008;12:1612‑4. J Neurosci Rural Pract 2014;5:55-7. 9. Islek A, Sayar E, Yılmaz A, Boneval C, Artan R. A rare outcome of Source of Support: Nil. Conflict of Interest: None declared.

Commentary

Independently by their location, bezoars are defined but Aspergillus fumigatus and other types of fungi

as concretions of undigestible materials. In most such as Rhizopus were also reported as a cause for of the cases they occur in stomach or intestines, these renal concretions, with or without involvement being composed by hair (trichobezoar), indigestible of renal parenchyma.[4‑7] Independently by the type vegetables or fruits (phytobezoar-general term, of fungus, fungal balls of the kidney have especially diospyrobezoar-tannin‑containing persimmon fibers), been reported in neonates and premature infants, they milk products (lactobezoar), drugs (pharmacobezoar), being sometimes associated with bilateral pelviureteric cotton, sand, metal, plastic, chemically‑transformed obstruction and consecutive anuria and urinary ascite.[4,6] gloves, and even wool, as in one of our previous These fungal concretions were especially related to reported cases in a post‑partum young girl.[1] In the immunosuppression, treatment with antibiotics, and gastrointestinal tract, most of the cases occurred in using of intravenous or urinary catheters.[4] Pelviureteral patients with diagnosed or even unknown psychiatric aspergilloma is an opportunistic lesion that can occur disorders such as trichophagia, trichotillomania, pica, in diabetics or other patients with impaired immunity, emotional disorders, , etc.[1] leading also to uni‑or bilateral ureteric obstruction[5] and renal abscesses.[6] Aspergilloma of the urinary Compared with the bezoars of the gastrointestinal tract is usually a secondary spread from pulmonary tract, few cases have been reported in field of urology. aspergillosis while candida bezoars occur in cases with Obstruction of the pelviureteric tract can be even systemic candidiasis and Rhizopus is a manifestation produced by foreign materials, such as an indwelling of zygomycosis.[4‑7] Primary renal aspergilloma and ureteral stent forgotten in a renal transplant patient,[2] or xanthogranulomatous pyelonephritis have also been by concretions of endogen undigestible materials.[3‑7] To reported in immunocompetent patients.[6] Till July 2014, our best knowledge, bezoars of the kidney and urinary about 65 cases of fungal bezoars of the urinary tract tract can occur as a result of at least three main causes. were reported, one third of them being related to First of all, a severe renal failure can to synchronous aspergillosis.[5,6] renal and intestinal deposits of drugs (renal and intestinal pharmacobezoars), first case being reported in 1973.[3] A third and more rare cause of obstruction of the renal tubes with or without involvement of the pelviureteric Second cause and the most frequent one is the fungal structures were reported in association with poisonings. bezoar, it being especially related to Candida albicans A strange case, for example, led to death of an adult

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