Ann Dermatol.  2010 May;22(2):229-231. 10.5021/ad.2010.22.2.229.

A Case of Basaloid Follicular Hamartoma

• Go JW
• Oh HE
• Cho HK
• Kang WH
• Ro BI

• KMID: 2265403
• DOI: http://doi.org/10.5021/ad.2010.22.2.229

Abstract

Basaloid follicular hamartoma (BFH), uncommon rare benign neoplasm connected to the adnexal structures, presents with multiple clinical manifestations that can develop into basal cell carcinoma. BFH may be congenital or acquired, and the congenital form can be further divided into the generalized and unilateral type, and the acquired form may present as localized and solitary lesions. Congenital, generalized BFH is associated with systemic diseases such as alopecia, cystic fibrosis, hypohidrosis, and myasthenia gravis. In contrast, sporadic cases are observed as unilateral or localized lesions. BFH shows thick cords and thin strands of anastomosing basaloid proliferations that arise from hair follicles and are enclosed by loose fibrous stroma. Here, we report a 70-year-old man with an acquired, solitary form of BFH.

Keyword

Basaloid follicular hamartoma

MeSH Terms

Aged
Alopecia
Carcinoma, Basal Cell
Cystic Fibrosis
Hair Follicle
Hamartoma
Humans
Hypohidrosis
Myasthenia Gravis

Figure

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  Fig. 1 The skin lesion is solitary, skin colored, 0.6×0.6 cm, and smooth, with a central depressed papule on the left forehead.

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  Fig. 2 Histopathology shows multiple islands composed of tumor cells situated in the upper dermis with variable epidermal connections (H&E, ×40).

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  Fig. 3 Basal cell proliferation surrounded by a loose fibrous stroma with no cleft between the stroma and tumor cells. No cellular atypia and atypical mitosis (H&E, ×400).

Reference

1. Lee HJ, Lee WJ, Chang SE, Lee MW, Moon KC, Choi JH, et al. A case of familial basaloid follicular hamartoma syndrome. Korean J Dermatol. 2007. 45:1070–1073.

2. Patel AB, Harting MS, Smith-Zagone MJ, Hsu S. Familial basaloid follicular hamartoma: a report of one family. Dermatol Online J. 2008. 14:14.

3. Choi SW, Ryu YS, Kim HO, Kim CW. A case of basaloid follicular hamartoma combined with basal cell epithelioma. Korean J Dermatol. 1998. 36:148–151.

4. Han ES, Seo SJ, Hong CK, Song KY, Ro BI. A case of basaloid follicular hamartoma. Korean J Dermatol. 1997. 35:303–306.

5. Walsh N, Ackerman AB. Basaloid follicular hamartoma: solitary and multiple types. J Am Acad Dermatol. 1993. 29:125–129.

6. Carney RG. Linear unilateral basal-cell nevus with comedones; report of a case. AMA Arch Derm Syphilol. 1952. 65:471–476.

7. Brownstein MH. Basaloid follicular hamartoma: solitary and multiple types. J Am Acad Dermatol. 1992. 27:237–240.

8. El-Darouti MA, Marzouk SA, Abdel-Halim MR, Zidan AZ, Fawzy MM. Basaloid follicular hamartoma. Int J Dermatol. 2005. 44:361–365.

9. Smith KJ, Skelton H. Basaloid follicular hamartomas associated with autoimmune disease: a possible role for retinoids in therapy. J Am Acad Dermatol. 2003. 49:1067–1070.

10. Kaiser A, Spence RJ, Parwani A, Tihan T, Barrett TL. Basaloid follicular hamartoma with trichoblastomatous proliferations. J Cutan Med Surg. 2003. 7:395–398.

11. Nelson BR, Johnson TM, Waldinger T, Gillard M, Lowe L. Basaloid follicular hamartoma: a histologic diagnosis with diverse clinical presentations. Arch Dermatol. 1993. 129:915–917.

12. Jih DM, Shapiro M, James WD, Levin M, Gelfand J, Williams PT, et al. Familial basaloid follicular hamartoma: lesional characterization and review of the literature. Am J Dermatopathol. 2003. 25:130–137.

13. Oseroff AR, Shieh S, Frawley NP, Cheney R, Blumenson LE, Pivnick EK, et al. Treatment of diffuse basal cell carcinomas and basaloid follicular hamartomas in nevoid basal cell carcinoma syndrome by wide-area 5-aminolevulinic acid photodynamic therapy. Arch Dermatol. 2005. 141:60–67.