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鲁宾斯坦-泰比综合征中的多发性毛母质瘤:一例报告

Multiple pilomatricomas in Rubinstein-Taybi syndrome: a case report.

作者信息

Cambiaghi S, Ermacora E, Brusasco A, Canzi L, Caputo R

机构信息

1st Dept. of Dermatology and Pediatric Dermatology, IRCCS-University of Milan, Italy.

出版信息

Pediatr Dermatol. 1994 Mar;11(1):21-5. doi: 10.1111/j.1525-1470.1994.tb00067.x.

Abstract

Rubinstein-Taybi syndrome is a multisystem developmental disorder due to an autosomal dominant mutation. It is clinically defined by the presence of peculiar facies, mental retardation, and broad thumbs and first toes. Important dermatologic findings include hirsutism, keloids, hemangiomas, and dermatoglyphic abnormalities. We report a 12-year-old girl with the typical phenotype of Rubinstein-Taybi syndrome, associated with numerous pilomatricomas. These are benign epithelial neoplasms with hair cell differentiation that may have a familial transmission. Pilomatricomas have not been reported in patients with Rubinstein-Taybi syndrome, although their association with myotonic dystrophy, another autosomal dominant disorder, is well known. Possibilities to explain the association include contiguous gene syndrome, the action of a pleiotropic gene, predisposition to malformations, and mere coincidence.

摘要

鲁宾斯坦-泰比综合征是一种由常染色体显性突变引起的多系统发育障碍。其临床特征为特殊面容、智力迟钝以及宽拇指和宽第一趾。重要的皮肤学表现包括多毛症、瘢痕疙瘩、血管瘤和皮纹异常。我们报告了一名12岁女孩,具有鲁宾斯坦-泰比综合征的典型表型,并伴有大量毛母质瘤。毛母质瘤是一种具有毛发细胞分化的良性上皮性肿瘤,可能有家族性遗传。尽管毛母质瘤与另一种常染色体显性疾病——强直性肌营养不良的关联已为人熟知,但在鲁宾斯坦-泰比综合征患者中尚未有相关报道。解释这种关联的可能性包括邻接基因综合征、多效基因的作用、畸形易感性以及单纯巧合。

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