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

Sep 30, 2026

Heterotopic hair in the oral cavity is rare, and reported causes include prior skin graft reconstruction, oral and maxillofacial trauma, or the absence of an identifiable etiology. A case is presented of a patient with trichotillomania showing a persistent sinus track opening between teeth #9 and #10, alongside a well-defined periradicular lesion. Microscopy demonstrated a partially lined cyst-like entity containing terminal hairs and a foreign body-type giant cell reaction, linked to repeated hair pulling and placement in the mouth.

LanguageEnglish

CategoryHealthcare

FormatPDF

Pages6

Reading Time15 min

AccessFree to download

Abstract

Introduction

Case report

Clinical evaluation

Imaging (CBCT)

Excisional biopsy and pathology

Discussion

Oral and Maxillofacial Surgery (2026) 30:13 [https://doi.org/10.1007/s10006-025-01495-4](https://doi.org/10.1007/s10006-025-01495-4) CASE REPORT Intraosseous hair-induced cyst-like lesion of the maxilla associated with trichotillomania: first reported case and review of the literature Jay Saepoo1 · Steven Dorris1 · Kittiphoj Tikkhanarak1 · Sherry Timmons1 · Nidhi Handoo1 · John Hellstein1 · Adam Holton1,2 · Emily Lanzel1 Received: 23 September 2025 / Accepted: 4 December 2025 © The Author(s) 2026 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 periradicularlesion 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. Keywords Trichotillomania · Cyst · Foreign body reaction · Hair · Oral hair 􀀍 Jay Saepoo [jirayu-saepoo@uiowa.edu](jirayu-saepoo@uiowa.edu) Steven Dorris [steven-dorris@uiowa.edu](steven-dorris@uiowa.edu) Kittiphoj Tikkhanarak [kittiphoj-tikkhanarak@uiowa.edu](kittiphoj-tikkhanarak@uiowa.edu) Sherry Timmons [sherry-timmons@uiowa.edu](sherry-timmons@uiowa.edu) Nidhi Handoo [nidhi-handoo@uiowa.edu](nidhi-handoo@uiowa.edu) John Hellstein [john-hellstein@uiowa.edu](john-hellstein@uiowa.edu) Adam Holton [holton.uiowa@gmail.com](holton.uiowa@gmail.com) Emily Lanzel [emily-lanzel@uiowa.edu](emily-lanzel@uiowa.edu) 1 Department of Oral Pathology, Radiology and Medicine, College of Dentistry, The University of Iowa, 801 Newton Rd., Iowa city, IA, USA 2 Holton Oral and Maxillofacial Surgery, 820 Grand Ave, Spencer, IA 51301, USA Introduction In DSM − 5 (Diagnostic and Statistical Manual of Mental Disorders), under Obsessive-Compulsive and Related Disorders category, trichotillomania is a psychological disorder characterized by repetitive hair pulling, with an estimated adult prevalence of approximately 1.7%, affecting men and women equally. This condition is often associated with significant distress and comorbid mental health disorders, including anxiety, depression, obsessive-compulsive disorder, post-traumatic stress disorder, and attention-deficit/ hyperactivity disorder [1, 2] . Heterotopic hair within the oral cavity is a rare phenomenon. Previous reports have documented heterotopic hair growth in the facial gingiva, palatal gingiva, buccal mucosa, and dorsal tongue [3–7], or in the areas of prior skin graft reconstruction [8] . In most of these cases, no clear etiology for intraoral hair growth was identified [3, 4, 7] . Here, we report a case of an intraosseous hair-induced cyst with foreign body reaction in a patient with trichotillomania. To our knowledge, this is the first reported case of such a lesion in the anterior maxilla associated with trichotillomania. 1 3 Case report A 64-year-old female presented to a private oral and maxillofacial surgery clinic for evaluation of a persistent sinus track opening on the facial aspect between teeth \#9 and \#10 . Clinical examination revealed soft tissues within normal limits, with no tenderness on palpation. There was no evidence of purulent, serous or serosanguinous discharge. Percussion testing elicited no pain, and vitality testing of the involved teeth showed normal responses. There was no evidence of cleft lip or cleft palate, and the patient denied

FAQs

What rare oral condition is highlighted in this case report?

The report describes heterotopic hair in the oral cavity, presenting as an intraosseous hair-induced cyst-like lesion in the anterior maxilla associated with trichotillomania.

What clinical and imaging findings were observed?

A persistent sinus track opening was found between teeth #9 and #10, with a well-defined hydraulic-appearing hypodense lesion on CBCT showing interproximal and palatal involvement and mild surrounding sclerosis.

How did the pathology support the final interpretation?

Histopathology showed thin, non-keratinizing stratified squamous epithelium with numerous terminal hairs, focal fibroblast proliferation in a vascular stroma, and numerous multinucleated giant cells consistent with a foreign body-type reaction.

Sep 30, 2026

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