Al-Zubaidi Nabaa, Sayna Sabertehrani, Kani Zahra Asadi, Gheisari Mehdi, Zahedi Khatere
Skin Research Center Shahid Beheshti University of Medical Sciences Tehran Iran.
Department of Dermatology, Loghman Hakim Hopsital Shahid Beheshti University of Medical Sciences Tehran Iran.
Clin Case Rep. 2025 Jul 29;13(8):e70700. doi: 10.1002/ccr3.70700. eCollection 2025 Aug.
Congenital melanocytic nevi (CMN), arising from the proliferation of melanocytes, manifest at birth and are colloquially termed as bathing trunk, coat-sleeve, or stocking nevi. These lesions, occurring in approximately 1 to 2% of neonates, predominantly localize over the trunk and thigh regions. Giant congenital nevi, characterized by a size exceeding 20 cm, often present with pigmentation and hairiness. Herein, we present the case of a 14-year-old patient diagnosed with a giant congenital nevus (bathing trunk nevus) accompanied by lipomatosis, while concurrently reviewing the literature to delineate this rare association. Our investigation identified a total of six cases, comprising five females and one male. Notably, half of the cases (3/6) exhibited multiple lipomatous lesions, with the back region emerging as the most prevalent site. In our presented case, progressive enlargement of a soft tissue mass in the right flank prompted excision, revealing a well-circumscribed lipomatous lesion infiltrated diffusely by benign-appearing melanocytes within fat lobules, as confirmed by immunohistochemistry. Specifically, positivity for HMB45 and S100 markers was observed. Beyond the rarity of such occurrences, our report underscores the infrequent concomitance of CMN with lipomatosis.
先天性黑素细胞痣(CMN)由黑素细胞增殖引起,出生时即出现,通俗地称为躯干、衣袖或长袜痣。这些病变约在1%至2%的新生儿中出现,主要分布在躯干和大腿区域。巨大先天性痣的特征是大小超过20厘米,常伴有色素沉着和多毛。在此,我们报告一例14岁患者,诊断为巨大先天性痣(躯干痣)并伴有脂肪瘤病,同时回顾文献以阐述这种罕见的关联。我们的调查共发现6例,包括5名女性和1名男性。值得注意的是,半数病例(3/6)表现为多发脂肪瘤性病变,背部是最常见的部位。在我们报告的病例中,右侧腹软组织肿块逐渐增大促使进行切除,切除后发现一个边界清晰的脂肪瘤性病变,脂肪小叶内有外观良性的黑素细胞弥漫浸润,免疫组化证实了这一点。具体而言,观察到HMB45和S100标记呈阳性。除了这种情况的罕见性之外,我们的报告强调了CMN与脂肪瘤病同时存在的情况很少见。