Post-Graduation Program in Health Sciences, Universidade Católica de Pelotas, Pelotas, RS, Brazil.
Post-Graduation Program in Health Sciences, Universidade Católica de Pelotas, Pelotas, RS, Brazil; Department of Dermatology, Universidade Federal de Pelotas and Universidade Católica de Pelotas, Pelotas, RS, Brazil.
An Bras Dermatol. 2022 Jan-Feb;97(1):58-62. doi: 10.1016/j.abd.2021.09.002. Epub 2021 Nov 25.
McCune - Albright syndrome is a genetic disease with cutaneous mosaicism caused by post-zygotic activating mutations in GNAS locus, it has a triad of fibrous bone dysplasia, café-au-lait macules and precocious puberty. We examined a 22-year-old female patient with café au lait spot in right side of the abdomen, with a chessboard - like distribution, extending to right thigh with geographical contours, she has also an ovarian cyst, scoliosis and truncal obesity. Biopsies were taken from the hyperpigmented area and processed for light microscopy and for transmission electron microscopy. Light microscopy showed increased melanin pigment with HE staining. Immunohistochemistry with melanocytic markers (HMB-45 and Melan-A) revealed a normal number of melanocytes. Transmission electron microscopy demonstrated normal epidermal structures, such as desmosomes, cytokeratin filaments and hemidesmosomes. With high magnifications an irregular melanossomal contour was seen, with some indentations in their outline.
McCune-Albright 综合征是一种具有皮肤镶嵌性的遗传性疾病,由 GNAS 基因座的合子后激活突变引起,其三联征为纤维性骨发育不良、咖啡斑和性早熟。我们检查了一位 22 岁的女性患者,其右侧腹部有咖啡斑,呈棋盘状分布,延伸至右大腿,呈地理轮廓状,她还患有卵巢囊肿、脊柱侧凸和躯干性肥胖。从色素沉着区域取活检进行光镜和透射电镜检查。光镜下 HE 染色显示黑色素增多。用黑色素细胞标志物(HMB-45 和 Melan-A)进行免疫组化显示黑色素细胞数量正常。透射电镜显示正常的表皮结构,如桥粒、细胞角蛋白丝和半桥粒。高倍放大时可见不规则的黑色素体轮廓,其轮廓有些凹陷。