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Intraoral Cutaneous Hamartomas—Clinicopathologic and Immunohistochemical Characteristics of 3 Cases

Wilkinson, Peter E.*; Gopalakrishnan, Rajaram BDS, PhD; Argyris, Prokopios P. DDS, MS, PhD†,‡

The American Journal of Dermatopathology: November 2019 - Volume 41 - Issue 11 - p 794–798
doi: 10.1097/DAD.0000000000001354
Original Study

Abstract: 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.

*School of Dentistry, University of Minnesota, Minneapolis, MN;

Division of Oral and Maxillofacial Pathology, School of Dentistry, University of Minnesota, Minneapolis, MN; and

Department of Biochemistry, Molecular Biology and Biophysics, College of Biological Sciences, University of Minnesota, Minneapolis, MN.

Correspondence: Prokopios P. Argyris, DDS, MS, PhD, Division of Oral and Maxillofacial Pathology, School of Dentistry, University of Minnesota, 515 Delaware Street SE 16-206B, Minneapolis, MN 55455 (e-mail:

The authors declare no conflicts of interest.

Presented at the poster session of the Joint International Academy of Oral Pathology (IAOP) and American Academy of Oral and Maxillofacial Pathology (AAOMP) Meeting; June 23–28, 2018; Vancouver, Canada.

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