Garrido Maria C, Maroñas-Jiménez Lidia, Morales-Raya Carlos, Ruano Yolanda, Rodriguez-Peralto Jose L
Departments of Pathology, and.
Dermatology, Hospital Universitario 12 de Octubre, Universidad Complutense, Madrid, Spain.
Am J Dermatopathol. 2018 Jul;40(7):506-510. doi: 10.1097/DAD.0000000000001039.
Congenital melanocytic nevi (CMN) are benign melanocytic proliferations that are usually present at birth. A somatic mosaicism for an NRAS point mutation is responsible for the several phenotypic abnormalities that may be associated with congenital nevi. We report the case of a 7-year-old boy with a proliferative nodule (PN) arising in a Giant CMN completely excised and with several visceral and intraspinal melanoma metastases with no evidence of primary cutaneous melanoma. The careful analysis of the clinical, morphologic, and molecular features allowed the distinction of between the benign PN (BPN) and the melanoma. The BPN showed a characteristic comparative genomic hybridization pattern with gains or losses of whole chromosomes, whereas the melanoma displayed gains or losses involving complex partial chromosomal copy number gains or losses. Leptomeningeal melanocytes are more susceptible to transformation by oncogenic NRAS than cutaneous melanocytes, and central nervous system melanomas are more common than cutaneous melanomas in the setting of CMN. Thus, it has been recommended to characterize the congenital disease in patients with 2 CMN at birth, independently of size and site, with a single magnetic resonance imaging screening younger than the age of 1 year.
先天性黑素细胞痣(CMN)是通常在出生时就存在的良性黑素细胞增殖性病变。NRAS点突变的体细胞镶嵌现象是导致先天性痣可能伴有的多种表型异常的原因。我们报告了一例7岁男孩的病例,其巨大先天性黑素细胞痣中出现增殖性结节(PN),该结节已被完全切除,且存在多处内脏和脊柱内黑色素瘤转移,未发现原发性皮肤黑色素瘤。对临床、形态学和分子特征的仔细分析有助于区分良性PN(BPN)和黑色素瘤。BPN显示出具有全染色体增减的特征性比较基因组杂交模式,而黑色素瘤则表现出涉及复杂部分染色体拷贝数增减的增减情况。软脑膜黑素细胞比皮肤黑素细胞更容易被致癌NRAS转化,在CMN的情况下,中枢神经系统黑色素瘤比皮肤黑色素瘤更常见。因此,建议对出生时患有2个CMN的患者进行先天性疾病特征描述,无论其大小和部位如何,在1岁之前进行单次磁共振成像筛查。