A Case of Zosteriform Spiradenoma Following Varicose Veins

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Spiradenoma is a benign skin adnexal tumor, traditionally hypothesized to occur in the intradermal portion of the eccrine duct, although its exact origin is not clear. Recently, however, immunochemical examination of these tumors revealed follicular stem cell markers, which are considered to be derived from the folliculosebaceous-apocrine unit1.

A 46-year-old woman presented with multiple erythematous nodules on the right posterior thigh and calf that first occurred 15 years ago (Fig. 1). She complained of the skin lesions being tender and a heavy feeling and numbness in her legs. The patient had a history of varicose veins diagnosed by a cardiovascular surgeon. Upon physical examination, multiple erythematous nodules were observed, reaching 0.5 to 2 cm in diameter, showing a zosteriform distribution following the adjacent varicose veins.

Fig. 1
(A) Multiple erythematous nodules formed a zosteriform distribution on the right posterior thigh and calf. (B) Each skin lesion was observed to run with varicose veins in the lower extremities. We received the patient’s consent form about publishing all photographic materials.

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After a skin biopsy was performed on one of the tender lesions of the thigh, a large, well-circumscribed, encapsulated tumor was detected in the dermis. The tumor consisted of multiple lobules (Fig. 2A), which formed cords, bands, or pseudoglandular rosettes (Fig. 2B). The lobules were composed of two cell types; small, darkly staining basaloid cells and larger cells with pale nuclei in the center (Fig. 2C). Based on the biopsy results, spiradenoma was diagnosed. In addition, the patient underwent an excisional biopsy of another tender calf lesion, which revealed the same pathologic findings. She is currently undergoing regular follow-up care without any change in the number or size of the skin lesions.

Fig. 2
(A) A large, well-circumscribed, encapsulated tumor in the dermis (H&E, ×10). (B) The tumor consisted of multiple lobules, which formed cords, bands, or pseudoglandular rosettes (H&E, ×40). (C) The lobules were composed of two cell types; small, darkly staining basaloid cells and larger cells with pale nuclei in the center (H&E, ×200). (D) β-catenin expression in reactive nuclei of cells in the tumor (Immunohistochemistry, ×400; insert, ×40).

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Eccrine spiradenoma, a disease first reported by Kersting and Helwig in 1956, usually manifests clinically as a solitary tender nodule. However, this condition rarely occurs as multifocal tumors with a localized pattern of linear or zosteriform distribution. So far, there have been about 30 of these cases reported in the literature, but only 3 have been observed in the lower extremities2. Furthermore, this is the first report of zosteriform spiradenoma following the existing varicose veins.

Chronic venous insufficiency, with a clinical manifestation of varicose veins, leads to venous hypertension. Venous hypertension induces the upregulation of matrix metalloproteinase-93, which not only promotes inflammation and remodeling of the extracellular matrix, but also regulatesβ-catenin signaling via the modulation of N-cadherin4. Im et al.5 reported that the upregulation of β-catenin and the downregulation of both axin and glycogen synthase kinase 3β are signaling alternations that are important in the development of spiradenoma. They also found that the expression of β-catenin in nuclei can occur in varying degrees. Authors additionally conducted immunohistochemistry for other skin lesions of patients and observed the immunoreactivity of the nuclei for β-catenin (Fig. 2D).

Further research is needed to clarify the relationship between chronic venous insufficiency and a multiple zosteriform distribution of spiradenoma. However, it is reasonable to consider that a link may exist between the two diseases because of their temporal relation and important shared signaling pathway. In conclusion, we herein report a case of spiradenoma with a unique clinical manifestation and suggest considering the possibility that chronic venous insufficiency may contribute to the development of this condition.