Trichophagia along with trichobezoar in the absence of trichotillomania

Abstract

Trichobezoars are rarely described in the absence of trichotillomania. In this report we present a case of trichobezoar associated with trichophagia 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 stomach. In view of trichobezoar, psychiatry consultation was sought. Exploration of history revealed that the patient was eating hairs and clay since early childhood. As per patient she would eat hairs thrown by others. She would like the taste of hair 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 scalp and other body parts did not show any evidence of alopecia or pulling of hair/short 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.

Introduction

Trichophagia is understood as a compulsive behavior involving eating hairs, usually of own. Trichobezoars are concretions of mostly hair, which accumulate in the gastrointestinal tract. Trichobezoars are often associated with psychiatry disorder like trichotillomania, mental retardation, emotional disturbances and/or pica.[1] Most of the cases of trichobezoars are described in adolescent girls/young females.[2,3]

However, trichophagia and trichobezoars are rarely described in the absence of trichotillomania.[4] In this case report we describe a case of trichobezoar associated with trichophagia in the absence of trichotillomania.

Case Report

A 16-year-old single girl, with 10 years of formal education, from rural background presented to surgery outpatient with complaints of pain in abdomen and vomiting for the last 6 months. She would have frequent bouts of vomiting, frequency of which increased in the last few days prior to presentation. On physical examination, she was found to have a mobile mass of 14 × 16 cm size in the upper abdomen, which was tender, with smooth surface and irregular margins. 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 a large mobile mass made of hairs was seen in the stomach. Based on the findings of ultrasound and upper gastrointestinal endoscopy, a diagnosis of trichobezoar was considered and psychiatry consultation was sought. Psychiatric evaluation revealed that the girl was eating hairs and clay since early childhood. She would eat the hairs thrown here and there by her mother, other family members and neighbors. The patient would like the taste of hairs and had a strong and persistent desire to eat hairs. She would often go out searching for the same. Additionally, at times she would also eat clay. The description of the patient was corroborated by the mother too; who described that despite persistence reprimands, the patient persisted to eat hairs and occasionally would eat clay. However, the patient denied pulling out her own hairs. Physical examination of scalp and other body parts did not reveal any evidence of alopecia or pulling of hair/short hair. Past history did not reveal any evidence of depressive disorders, obsessive compulsive disorder, other anxiety disorders, eating disorders, emotional disturbances, mental sub-normality and other impulse control disorders. Also, there was no family history of any mental illness. Investigations in the form hemoglobin level, complete blood counts, serum electrolytes, liver function test, renal function test, pancreatic enzymes and routine stool examination did not reveal any abnormality. In view of large size of the mass, open laparotomy was done and the mass was removed. On examination, the mass comprised of entangled hairs. A psychiatric diagnosis of other behavioral and emotional disorders with onset usually occurring in childhood and adolescence (Pica of infancy and childhood) as per the ICD-10 was considered. The patient was counseled against eating hairs and clays and was informed about the consequences of the same. Behavioral analysis revealed that the patient would have urge to eat hair more so, when under stress. Additionally, behavioral principles with regards to management of urge and impulse control were discussed. Additionally, the psychotherapeutic intervention focused on teaching adaptive coping skills to deal with the stressful situations. Family members were also educated about the illness and were asked to be vigilant to note the patient behavior of eating hairs and other non-eatable items. The patient improved after the surgery and follow-up at 6 months did not reveal any behavior suggestive of trichophagia or pica.

Discussion

The term “Bezoar” is understood to be derived from the Arabic word “Bazehr” or the Persian word “Padzhar,” which are used for antidote, because traditionally stones from the stomach or intestine of various animals were considered to have medicinal value.[5] Different types of bezoars are described in humans and according to the composition are named as phytobezoar (containing vegetable fibres), lactobezoar (milk products), pharmacobezoar (medications) and trichobezoar (hairs).[1,5] Additionally bezoars composed of cotton and other materials have also been described in the literature.[3] Usually the bezoars are found in the stomach, however, when the tail of the bezoar extends beyond the pylorus and involves the duodenum or further lower gastrointestional tract, it is known as Rapunzel syndrome.[6] In terms of pathogenesis of trichobezoar, it is believed that because of its smooth surface the hairs do not move forward with the peristaltic movement and resultantly get accumulated.[7]

Trichophagia and trichobezoar are abundantly described in surgical literature.[1,2,3,4,5,6,7,8,9,10,11,12,13,14,15] In few cases, no evidence of trichotillomania was found in patients presenting with trichobezoar.[4,9] Previous reports of trichobezoar have also reported co-occurrence of eating hairs and other non-eatable items amounting to pica.[8] Pica is understood as a clinical condition in which the subjects often eats non-nutritive materials like soil, dirt, hairballs, ice, paint and sand, etc., It is usually seen in the setting of low intelligence;[16] however, it can also occur in children with normal intelligence.[16]

As trichobezoar is often associated with trichotillomania and eating of other non-eatable items which can amount to pica, a psychiatric evaluation of such patients is recommended. Although trichotillomania itself is considered to be an impulse control disorder, it may also manifest as a symptom of many other mental disorders. Accordingly detail psychiatric evaluation to focus on ruling trichotillomania and other psychiatric disorders. Detail physical examination should involve looking for patches of hair loss in various parts of the body. A patient may have pica and also trichotillomania as a manifestation of low intelligence. Accordingly if low intelligence is suspected, the patient intelligence quotient must be assessed. In view of the associated pica, these patients must be evaluated for iron deficiency. Management of trichotillomania and pica involves psychoeducation, behavior therapy (habit reversal therapy in case patient has trichotillomania) and teaching adaptive coping skills. Patients with trichobezoar must be followed up regularly because of the risk of recurrence.[11,12]

Most of the cases of trichophagia and trichobezoar are described in patients with trichotillomania, which is considered to be an impulsive control disorder or other psychiatric disorders. Index case had no psychiatric history, any evidence of psychological distress or specific personality traits which could be considered to be associated with trichophagia. The present case exemplifies that patients of trichobezoar can have isolated trichophagia. Hence, the absence of trichotillomania should not be considered to be equivalent of absence of trichophagia.