Ann Dermatol.  2020 Feb;32(1):57-63. 10.5021/ad.2020.32.1.57.

Eccrine Syringofibroadenoma Associated with Bowen's Disease: A Case Report and Review of the Literature

• Lee JS
• Park H
• Yoon HS
• Cho S

• KMID: 2467202
• DOI: http://doi.org/10.5021/ad.2020.32.1.57

Abstract

Eccrine syringofibroadenoma (ESFA) is a rare, benign adnexal neoplasm which usually manifests as a solitary nodule on the extremities of elderly patients. Few case reports have described an association between ESFA and carcinomas including squamous cell carcinoma, porocarcinoma, and basal cell carcinoma. A 66-year-old male presented with a pruritic, erythematous brownish solitary plaque with crusted and verrucous surface on the extensor side of the right thigh. The lesion developed 6 to 7 years ago, and had been growing slowly. Biopsy revealed anastomosing epithelial strands which were composed of 2 areas: the upper area consisting of dysplastic cells with prominent nucleoli and abundant mitoses, and the lower area consisting of oval and round cells, and occasionally small luminal ducts. Dysplastic cells comprised almost the entire epidermis but did not invade into the dermis. Benign syringofibroadenomatous lesion surrounded the dysplastic cells in the lowermost portion of the epidermis. Although it is still unclear whether ESFA undergoes malignant transformation or it is a reactive change to carcinoma, complete excision should be performed to prevent malignant transformation with unknown risk.

Keyword

Basal cell carcinoma; Bowen's disease; Eccrine syringofibroadenoma; Porocarcinoma; Squamous cell carcinoma

MeSH Terms

Aged
Biopsy
Bowen's Disease*
Carcinoma, Basal Cell
Carcinoma, Squamous Cell
Dermis
Epidermis
Extremities
Humans
Male
Mitosis
Phenobarbital
Thigh
Phenobarbital

Figure

• 

  Fig. 1 Pruritic solitary mass on the extensor surface of the right thigh. A 3×2 cm sized, well-circumscribed, erythematous brownish plaque with verrucous and partially crusted surface.

• 

  Fig. 2 Histopathology of the solitary mass. (A) Anastomosing epithelial strands extended down to the dermis and were composed of 2 different areas, i.e., dysplastic cells comprising almost the entire epidermis but not invading into the dermis and benign adenomatous lesion immediately surrounding the dysplastic keratinocytes in the lowermost portion of the epidermis (hematoxylin and eosin [H&E], ×40). (B) Epithelial strands were surrounded by mucinous fibrous stroma, and small luminal ducts were embedded within the strands (H&E, ×100). (C) Dysplastic area was composed of atypical cells with prominent nucleoli and abundant mitoses. Adjacent benign adenomatous portion was composed of oval and round cells (H&E, ×200). (D) Epithelial membrane antigen (EMA) was positive in the cytoplasm of dysplastic cells and weakly positive in the ductal structures in the adenomatous lesion (arrowheads) (EMA, ×40). (E) A p63 was positive in the nucleus of dysplastic cells and weakly positive in the adenoma lesion (p63, ×40). (F) Carcinoembryonic antigen (CEA) stain was weakly positive in the ductal structures in the adenoma lesion (arrowhead and inset on the right upper corner) (CEA, ×40).

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  Fig. 3 Histopathology of excised tissues. (A) Epithelial strands formed anastomoses and ductal structures were observed within the epithelial strands extending down into the dermis (hematoxylin and eosin [H&E], ×40). (B, C) Immediately above and adjacent to the syringofibroadenoma lesion, almost the entire epidermis displayed dysplasia with atypical keratinocytes and numerous mitotic figures (H&E, ×100).

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