Very Early Onset Trichotillomania Presenting with Recurrent Trichobezoar: Conventional Wisdom Questioned

Abstract

Trichotillomania (TTM), a disorder characterized by compulsive hair pulling, is undergoing a conceptual and nosological re-evaluation. Little long-term information is available about this condition when it strikes in early childhood. Trichobezoar, an ingestional foreign body lodged in the gastrointestinal tract composed of swallowed hair, is usually associated with underlying psychiatric and emotional disturbances. In this report, we describe a young girl who had her symptom onset at the age of 3, but presented again after more than a decade with recurrent symptomatic large trichobezoars needing surgical removal both times. Her clinical course and presentation challenged contemporary understanding of TTM.

INTRODUCTION

Trichotillomania (TTM) is a disabling clinical condition characterized by the essential criteria of recurrent hair pulling resulting in noticeable hair loss. It is now classified under obsessive-compulsive and related disorders in Diagnostic and Statistical Manual of Mental Disorders-5.[1] Individuals who have their symptom onset before 6 years are uncommon and have been classified as very early onset TTM.[2] The literature on very early onset TTM is sparse contributing to a poor understanding of this variant. In this report, we aim to present the case of a young girl with very early onset TTM who presented with recurrent large gastric trichobezoar, necessitating surgical intervention.

CASE REPORT

A 14-year-old developmentally normal girl was referred to the psychiatry outpatient department with complaints of repeated pulling and ingestion of hair for a year. Physical complaints included abdominal pain, repeated episodes of vomiting, and significant weight loss. Her practice of pulling and ingesting her hair had led to significant tensions with her parents with whom she had stopped communicating. During mental status examination, she described the urge to pull out her hair as repugnant and detrimental to her health and appearance. She had similar symptoms at the age of 3 when she was diagnosed with a large gastric trichobezoar and underwent gastrostomy. No symptoms were reported in the intervening period. Detailed psychiatric evaluation revealed no other co-morbid conditions. Clinical examination at intake revealed a poorly nourished individual. A mobile hard mass was felt in the epigastric region measuring roughly 15 cm × 10 cm. Computed tomographic study with contrast revealed a large ovoid heterogeneous mass measuring 16 cm × 8 cm × 5 cm and extending up to the pylorus [Figure 1]. In view of the large size of bezoar, she was advised exploratory laparotomy with gastrostomy. Currently, 12 weeks have passed since the surgery, and her symptoms have spontaneously remitted.

Figure 1.

Computed tomography abdomen showing the trichobezoar occupying nearly the whole lumen of the stomach

DISCUSSION

The present case offers some unique insights into the understanding of TTM. Previously, hair pulling with onset in preschool age has been noted to have a benign and time-limited course.[3,4,5] But our patient presented with recurrent symptomatic large trichobezoars more than a decade apart. The symptom of hair pulling was clearly ego-dystonic, and this is at variance with contemporary understanding. On both occasions, the surgical removal of the bezoars was followed by complete remission of symptoms. This coupled with a long gap between her recurrences led us to question the wisdom of initiating maintenance pharmacological treatment. We were unable to find any similar reports where hair pulling went into remission following surgery for trichobezoar. Recently, Kirpinar et al. have reported a case of recurrent trichobezoar but the patient required a combination of drugs and behavior therapy to induce remission upon recurrence.[6] There is a paucity of long-term data on TTM with onset in very early childhood. The present case aims to illuminate this aspect. To conclude, clinicians should consider the possibility of a trichobezoar in children presenting with a mobile upper abdominal mass and unexplained weight loss. Although our patient did not require focused management, such instances are probably rare and need to be observed closely and reliably. Psychotherapeutic techniques like Habit Reversal Training and Stimulus Control (HRT Plus). have been found to be effective in treating drug-resistant presentations of TTM and preventing recurrences.[7] The variant phenomenology of hair pulling noted in our case may have classificatory implications for International Classification of Disease-11.