Chidambaram A, Goldstein A M, Gailani M R, Gerrard B, Bale S J, DiGiovanna J J, Bale A E, Dean M
Intramural Research Support Program, Scientific Application International Corp., Frederick, Maryland 21702, USA.
Cancer Res. 1996 Oct 15;56(20):4599-601.
The nevoid basal cell carcinoma syndrome (NBCCS), or Gorlin syndrome, is a multisystem autosomal dominant disorder. The salient features of this syndrome include multiple basal cell carcinomas, palmar and/or plantar pits, odontogenic keratocysts, skeletal and developmental anomalies, and ectopic calcification. Other features include such tumors as ovarian fibromas and medulloblastomas. There is extensive interfamilial as well as intrafamilial variability with respect to the manifestation and severity of the phenotype. Alterations in the human homologue (PTCH) of the Drosophila segment polarity gene patched have been identified in NBCCS patients as well as tumors associated with this syndrome. We report several mutations in this gene in NBCCS patients and present the clinical phenotypes of the individuals in whom these mutations were identified.
痣样基底细胞癌综合征(NBCCS),又称戈林综合征,是一种多系统常染色体显性遗传病。该综合征的显著特征包括多发性基底细胞癌、掌跖凹、牙源性角化囊肿、骨骼和发育异常以及异位钙化。其他特征包括卵巢纤维瘤和髓母细胞瘤等肿瘤。关于该综合征表型的表现和严重程度,家族间以及家族内均存在广泛的变异性。在NBCCS患者以及与该综合征相关的肿瘤中,已发现果蝇节段极性基因patched的人类同源基因(PTCH)发生改变。我们报告了NBCCS患者中该基因的几种突变,并展示了鉴定出这些突变的个体的临床表型。