Ponti Giovanni, Pellacani Giovanni, Tomasi Aldo, Sammaria Giuliano, Manfredini Marco
Department of Surgical, Medical, Dental and Morphological Sciences with Interest transplant, Oncological and Regenerative Medicine, Dermatology Unit, University of Modena and Reggio Emilia, Italy.
Department of Surgical, Medical, Dental and Morphological Sciences with Interest transplant, Oncological and Regenerative Medicine, Dermatology Unit, University of Modena and Reggio Emilia, Italy.
Gene. 2016 Sep 10;589(2):104-11. doi: 10.1016/j.gene.2016.01.012. Epub 2016 Jan 12.
There are several genetic diseases with a wide spectrum of congenital bone stigmata in association to cutaneous and visceral benign and malignant neoplasms. Gorlin-Goltz syndrome, also named nevoid basal cell carcinoma syndrome, is an autosomal dominant systemic disease with almost complete penetrance and high intra-familial phenotypic variability, caused by germline mutations of the gene PTCH1. The syndrome is characterized by unusual skeletal changes and high predisposition to the development of multiple basal cell carcinomas, odontogenic keratocysts tumors and other visceral tumors. The Gorlin syndrome, clinically defined as distinct syndrome in 1963, existed during Dynastic Egyptian times, as revealed by a costellation of skeletal findings compatible with the syndrome in mummies dating back to 3000years ago and, most likely, in the ancient population of Pompeii. These paleogenetic and historical evidences, together with the clinical and biomolecular modern evidences, confirm the quite benign behavior of the syndrome and the critical value of the multiple and synchronous skeletal anomalies in the recognition of these rare and complex genetic disease.
有几种遗传性疾病,其先天性骨骼特征广泛,与皮肤和内脏的良性及恶性肿瘤相关。戈林-戈尔茨综合征,也称为痣样基底细胞癌综合征,是一种常染色体显性全身性疾病,几乎完全显性且家族内表型变异度高,由基因PTCH1的种系突变引起。该综合征的特征是骨骼异常以及极易发生多发性基底细胞癌、牙源性角化囊肿肿瘤和其他内脏肿瘤。1963年临床上将戈林综合征定义为一种独特的综合征,它在古埃及王朝时期就已存在,这一点可从追溯到3000年前的木乃伊以及很可能在庞贝古城的古代人群中发现的一系列与该综合征相符的骨骼发现得到证实。这些古遗传学和历史证据,连同临床和生物分子方面的现代证据,证实了该综合征相当良性的表现以及多种同步骨骼异常在识别这些罕见且复杂的遗传性疾病中的关键价值。