Intraoral Cutaneous Hamartomas - Clinicopathologic and Immunohistochemical Characteristics of 3 Cases

Peter E. Wilkinson, Rajaram Gopalakrishnan, Prokopios P. Argyris

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Intraoral cutaneous hamartomas (ICHs) are uncommon mucosal lesions characterized microscopically by a combination of cutaneous structures, including various stages of follicular and sebaceous elements. Due to their rarity, the clinicopathologic and immunohistochemical attributes of ICHs have not been thoroughly delineated. Three cases of ICH were identified from our records, and formalin-fixed paraffin-embedded sections were immunohistochemically stained with antibodies against androgen receptor, estrogen receptor, and progesterone receptor, p63, factor XIIIα, and CD34. All 3 ICHs involved the buccal mucosa with an M:F ratio = 2:1 and mean age = 42.3 years (age range: 27-61 years). ICHs presented as thickened, painless, white and yellow plaques or nodules of long duration, measuring 0.6-1.5 cm. No history of skin graft in the area of the lesions was reported. Histopathologically, the lesions showed aggregates of rudimentary folliculosebaceous structures. Although well-defined piloerector muscles were present in all cases of ICH, bona fide hair follicles and isolated hair shafts were identified only in 1 case. The overlying oral epithelium exhibited epidermis-like morphological features, while inflammation was generally absent. Immunohistochemically, strong and diffuse nuclear staining for androgen receptor and factor XIIIα was observed in the sebaceous glands, and estrogen receptor and p63 reactivity were confined exclusively to the peripheral basal cells, while progesterone receptor staining was negative in ICHs. CD34 diffusely decorated the lesional stroma. In conclusion, ICH is a rare lesion composed of cutaneous elements in an abnormal location. A predilection for the buccal mucosa is reported in the current study.

Original languageEnglish (US)
Pages (from-to)794-798
Number of pages5
JournalAmerican Journal of Dermatopathology
Issue number11
StatePublished - Nov 1 2019

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  • folliculosebaceous cystic hamartoma
  • immunohistochemistry
  • intraoral cutaneous hamartoma
  • oral cavity
  • skin graft


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