Intraosseous hair-induced cyst-like lesion of the maxilla associated with trichotillomania: first reported case and review of the literature

TL;DR

A patient with trichotillomania developed a rare cyst-like lesion in the maxilla caused by ingested hair, the first such case reported in medical literature.

Contribution

First reported case of intraosseous hair-induced cyst-like lesion linked to trichotillomania.

Findings

The lesion was partially lined and contained terminal hairs with a foreign body-type giant cell reaction.

The patient's trichotillomania behavior involved placing hair in her mouth and pressing it against the affected area.

This case expands the understanding of heterotopic hair-related oral pathologies.

Abstract

Heterotopic hair in the oral cavity is a rare condition, with possible etiologies including prior skin graft reconstruction, oral and maxillofacial trauma, or, in some clinical context, no identifiable cause. We report a case of a patient with trichotillomania who presented with a persistent sinus track opening between teeth #9 and #10 and a well-defined periradicular lesion in the area. Microscopically, it revealed a partially lined cyst-like entity containing terminal hairs, associated with a foreign body-type giant cell reaction. The patient admitted to a habit of repeatedly pulling and plucking her hair, placing it in her mouth, and occasionally pressing it against the area of concern. To our knowledge, this is the first reported case of an intraosseous hair-induced cyst-like entity with foreign body reaction in the anterior maxilla associated with trichotillomania.

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