Multiple familial trichoepitheliomas: a case report in Thailand and literature review

Authors

  • Sirima Sawatwarakul DIVISION OF DERMATOLOGY, DEPARTMENT OF MEDICINE, FACULTY OF MEDICINE, RAMATHIBODI HOSPITAL, MAHIDOL UNIVERSITY, BANGKOK, THAILAND
  • Pranee Svetvilas DIVISION OF DERMATOLOGY, DEPARTMENT OF MEDICINE, FACULTY OF MEDICINE, RAMATHIBODI HOSPITAL, MAHIDOL UNIVERSITY, BANGKOK, THAILAND
  • Kunlawat Thadanipon DIVISION OF DERMATOLOGY, DEPARTMENT OF MEDICINE, FACULTY OF MEDICINE, RAMATHIBODI HOSPITAL, MAHIDOL UNIVERSITY, BANGKOK, THAILAND

Keywords:

Multiple familial trichoepitheliomas, trichoepitheliomas, MFT

Abstract

Trichoepithelioma is a rare benign tumor of the pilosebaceous follicles. Presenting as multiple small papules and nodules which  usually appear predominantly on face and scalp. Many distinctive patterns were described. Here we report a case of multiple familial trichoepitheliomas (MFT) occurring in one family of a 29-year-old Thai male patient presenting with multiple discrete asymptomatic skin-colored shiny papules on central face for 14 years. We also include literature review of MFT cases established  to date.

References

Ying ZX, Ma HQ, Liu Y, Xiao SX, Wang YX , Wang GX. A novel mutation of CYLD in a Chinese family with multiple familial trichoepithelioma. Journal of the European Academy of Dermatology and Venereology : JEADV 2012;26:1420-3.

Khelifa E, Masouye I, Kaya G , Le Gal FA. Dermoscopy of desmoplastic trichoepithelioma reveals other criteria to distinguish it from Basal cell carcinoma. Dermatology (Basel, Switzerland) 2013;226:101-4.

Harada H, Hashimoto K , Ko MS. The gene for multiple familial trichoepithelioma maps to chromosome 9p21. The Journal of investigative dermatology 1996;107:41-3.

Zhang XJ, Liang YH, He PP, Yang S, Wang HY, Chen JJ et al. Identification of the cylindromatosis tumor-suppressor gene responsible for multiple familial trichoepithelioma. J Invest Dermatol 2004;122:658-64.

Lv H, Li C, Li Y, Tang J , Zhang Z. Three Mutations of CYLD Gene in Chinese Families With Multiple Familial Trichoepithelioma. Am J Dermatopathol 2013.

Yoshida H, Jono H, Kai H , Li JD. The tumor suppressor cylindromatosis (CYLD) acts as a negative regulator for toll-like receptor 2 signaling via negative cross-talk with TRAF6 AND TRAF7. J Biol Chem 2005;280:41111-21.

Reiley W, Zhang M , Sun SC. Negative regulation of JNK signaling by the tumor suppressor CYLD. J Biol Chem 2004;279:55161-7.

Massoumi R, Chmielarska K, Hennecke K, Pfeifer A , Fassler R. Cyld inhibits tumor cell proliferation by blocking Bcl-3-dependent NF-kappaB signaling. Cell 2006;125:665-77.

Stegmeier F, Sowa ME, Nalepa G, Gygi SP, Harper JW , Elledge SJ. The tumor suppressor CYLD regulates entry into mitosis. Proc Natl Acad Sci U S A 2007;104:8869-74.

Yung A, Newton-Bishop JA. A case of Bazex-Dupre-Christol syndrome associated with multiple genital trichoepitheliomas. The British journal of dermatology 2005;153:682-4.

Lee KH, Kim JE, Cho BK, Kim YC , Park CJ. Malignant transformation of multiple familial trichoepithelioma: case report and literature review. Acta Derm Venereol 2008;88:43-6.

Kirby JS, Siebert Lucking SM , Billingsley EM. Trichoblastic carcinoma associated with multiple familial trichoepithelioma. Dermatol Surg 2012;38:2018-21.

Pincus LB, McCalmont TH, Neuhaus IM, Kasper R , Oh DH. Basal cell carcinomas arising within multiple trichoepitheliomas. Journal of cutaneous pathology 2008;35 Suppl 1:59-64.

Misago N , Narisawa Y. Basal cell carcinoma in association with multiple trichoepitheliomas. Dermatology (Basel, Switzerland) 2001;202:261-5.

Allende I, Truchuelo MT, Alcantara J , Boixeda P. [Carbon dioxide-laser treatment of trichoepitheliomas in Brooke-Spiegler syndrome]. Actas dermo-sifiliograficas 2011;102:76-7.

Rallan D , Harland CC. Brooke-Spiegler syndrome: treatment with laser ablation. Clinical and experimental dermatology 2005;30:355-7.

Sajben FP , Ross EV. The use of the 1.0 mm handpiece in high energy, pulsed CO2 laser destruction of facial adnexal tumors. Dermatologic surgery : official publication for American Society for Dermatologic Surgery [et al] 1999;25:41-4.

Raghuveer C, Murthy C, Siddalingappa K, Shivanand DR. Multiple trichoepitheliomas: cosmetic improvement with dermabrasion. Journal of cutaneous and aesthetic surgery 2011;4:68-9.

Alessi SS, Sanches JA, Oliveira WR, Messina MC, Pimentel ER , Festa Neto C. Treatment of cutaneous tumors with topical 5% imiquimod cream. Clinics (Sao Paulo, Brazil) 2009;64:961-6.

Urquhart JL, Weston WL. Treatment of multiple trichoepitheliomas with topical imiquimod and tretinoin. Pediatric dermatology 2005;22:67-70.

Downloads

Published

2026-06-05

How to Cite

Sawatwarakul, S., Svetvilas, P., & Thadanipon, K. (2026). Multiple familial trichoepitheliomas: a case report in Thailand and literature review. Thai Journal of Dermatology, 29(4), 266–271. retrieved from https://he02.tci-thaijo.org/index.php/TJD/article/view/282753