The youngest male breast clear cell hidradenoma case: a literature review

Clear cell hidradenoma (CCH) of the breast is an exceedingly rare benign adnexal tumor. Its clinical and radiological presentation can closely mimic malignancy, posing a significant diagnostic challenge. We report a case of a 6-year-old boy presenting with a non-healing, ulcerated nodule on the left nipple-areolar complex. Ultrasonography revealed a lobulated, cystic nodule. An ultrasound-guided core needle biopsy was performed, revealing epithelial cells with clear cytoplasm, warranting excision. The lesion was completely excised. Histopathology confirmed CCH, but an involved lateral margin necessitated a successful re-excision. Immunohistochemistry (IHC) on the excised specimen showed tumor cells positive for p63, Cytokeratin, and EMA, and negative for SMA, CD10, ER, and PR. The patient remains disease-free at 12 months. To our knowledge, this is the youngest male reported with breast CCH. This case highlights the diagnostic pitfall where CCH mimics malignancy and underscores the critical role of histopathology and IHC, particularly p63 positivity and myoepithelial marker negativity, in reaching an accurate diagnosis. Complete surgical excision with clear margins is the definitive treatment.

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Publication Details

Journal
BMC Pediatrics
Published
2026-03-13
DOI
https://doi.org/10.1186/s12887-026-06566-3
Primary Topic
Cancer and Skin Lesions
Type
article
Field-Weighted Citation Impact
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article

The youngest male breast clear cell hidradenoma case: a literature review

Şükrüye Demirkaya, ESRA KARAKUS, SEDA SAHIN, Yavuz Yilmaz
BMC Pediatrics
Cancer and Skin Lesions
article

The youngest male breast clear cell hidradenoma case: a literature review

Şükrüye Demirkaya, ESRA KARAKUS, SEDA SAHIN, Yavuz Yilmaz
article en

Abstract

Clear cell hidradenoma (CCH) of the breast is an exceedingly rare benign adnexal tumor. Its clinical and radiological presentation can closely mimic malignancy, posing a significant diagnostic challenge. We report a case of a 6-year-old boy presenting with a non-healing, ulcerated nodule on the left nipple-areolar complex. Ultrasonography revealed a lobulated, cystic nodule. An ultrasound-guided core needle biopsy was performed, revealing epithelial cells with clear cytoplasm, warranting excision. The lesion was completely excised. Histopathology confirmed CCH, but an involved lateral margin necessitated a successful re-excision. Immunohistochemistry (IHC) on the excised specimen showed tumor cells positive for p63, Cytokeratin, and EMA, and negative for SMA, CD10, ER, and PR. The patient remains disease-free at 12 months. To our knowledge, this is the youngest male reported with breast CCH. This case highlights the diagnostic pitfall where CCH mimics malignancy and underscores the critical role of histopathology and IHC, particularly p63 positivity and myoepithelial marker negativity, in reaching an accurate diagnosis. Complete surgical excision with clear margins is the definitive treatment.

BMC Pediatrics
Ministry of Health (TR), Bilkent University (TR), University of Health Science (KH), Başkent University Hospital (TR), Sağlık Bilimleri Üniversitesi (TR), Yüksek İhtisas Üniversitesi (TR)
Good health and well-being
Openalex Percentile: Top 22%
Cancer and Skin Lesions
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