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Multiple Eruptive Epithelioid Hemangiomas on the Right Upper Extremity in an Asian Man

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초록

Epithelioid hemangioma is a rare benign vascular neoplasm. Recently, the term “multiple eruptive epithelioid hemangioma” has been proposed for epithelioid hemangioma with dis- tinct features. It is different from usual epithelioid hemangioma because of the multifocal distributions in various body regions with an eruptive onset. In addition, the histopathology of multiple eruptive epithelioid hemangioma shows increased cellular proliferation, mitosis, and nuclear pleomorphism and positive findings for FOS-B compared to classic epithelioid hemangioma. Herein, we report the case of a 59-year-old man with unusual manifestations suitable for multiple eruptive epithelioid hemangioma. He had multiple erythematous to purple-red dome-shaped nodules on the right hand, arm, and shoulder. The initial lesion was a solitary erythematous nodule on the right hand that abruptly extended to the right arm and shoulder. Microscopically, the tumor was a well-demarcated dermal nodule and showed capillary sized vascular structures. Vascular structures had epithelioid endothelial cells with abundant eosinophilic cytoplasm and vesicular nuclei. The tumor cells showed mild nuclear pleomorphism and a few mitosis and feature of resembling cobble stone was observed. In immunohistochemistry, CD31 and CD34 were positive in the endothelial cells. The endothelial cells showed nuclear positivity in FOS-B. Based on the clinical and histo- pathological findings, the final diagnosis was multiple eruptive epithelioid hemangiomas. This is the first report of multiple eruptive epithelioid hemangiomas in an Asian man after the term had been introduced.

Abstract

Epithelioid hemangioma is a rare benign vascular neoplasm. Recently, the term “multiple eruptive epithelioid hemangioma” has been proposed for epithelioid hemangioma with dis- tinct features. It is different from usual epithelioid hemangioma because of the multifocal distributions in various body regions with an eruptive onset. In addition, the histopathology of multiple eruptive epithelioid hemangioma shows increased cellular proliferation, mitosis, and nuclear pleomorphism and positive findings for FOS-B compared to classic epithelioid hemangioma. Herein, we report the case of a 59-year-old man with unusual manifestations suitable for multiple eruptive epithelioid hemangioma. He had multiple erythematous to purple-red dome-shaped nodules on the right hand, arm, and shoulder. The initial lesion was a solitary erythematous nodule on the right hand that abruptly extended to the right arm and shoulder. Microscopically, the tumor was a well-demarcated dermal nodule and showed capillary sized vascular structures. Vascular structures had epithelioid endothelial cells with abundant eosinophilic cytoplasm and vesicular nuclei. The tumor cells showed mild nuclear pleomorphism and a few mitosis and feature of resembling cobble stone was observed. In immunohistochemistry, CD31 and CD34 were positive in the endothelial cells. The endothelial cells showed nuclear positivity in FOS-B. Based on the clinical and histo- pathological findings, the final diagnosis was multiple eruptive epithelioid hemangiomas. This is the first report of multiple eruptive epithelioid hemangiomas in an Asian man after the term had been introduced.

참고문헌 (6)

  1. Adler BL, "Epidemiology and treatment of angiolymphoid hyperplasia with eosinophilia(ALHE) : a systematic review", J Am Acad Dermatol, 74, pp.506~512 (2016)
  2. Ko JS, "Diagnostically challenging epithelioid vascular tumors", Surg Pathol Clin, 8, pp.331~351 (2015)
  3. Llamas-Velasco M, "Multiple eruptive epithelioid hemangiomas : a subset of cutaneous cellular epithelioid hemangioma with expression of FOS-B", Am J Surg Pathol, 43, pp.26~34 (2019)
  4. Wells GC, "Subcutaneous angiolymphoid hyperplasia with eosinophilia", Br J Dermatol, 81, pp.1~14 (1969)
  5. Kantrow S, "Cutaneous epithelioid angiomatous nodule : report of a case and absence of microsatellite instability", J Cutan Pathol, 34, pp.515~516 (2007)
  6. Lamps LW, "Cat-scratch disease : historic, clinical, and pathologic perspectives", Am J Clin Pathol, 121(Suppl), pp.S71~S80 (2004)

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