Hahn H, Wicking C, Zaphiropoulous P G, Gailani M R, Shanley S, Chidambaram A, Vorechovsky I, Holmberg E, Unden A B, Gillies S, Negus K, Smyth I, Pressman C, Leffell D J, Gerrard B, Goldstein A M, Dean M, Toftgard R, Chenevix-Trench G, Wainwright B, Bale A E
Centre for Molecular and Cell Biology, University of Queensland, St. Lucia, Australia.
Cell. 1996 Jun 14;85(6):841-51. doi: 10.1016/s0092-8674(00)81268-4.
The nevoid basal cell carcinoma syndrome (NBCCS) is an autosomal dominant disorder characterized by multiple basal cell carcinomas (BCCs), pits of the palms and soles, jaw keratocysts, a variety of other tumors, and developmental abnormalities. NBCCS maps to chromosome 9q22.3. Familial and sporadic BCCs display loss of heterozygosity in this region, consistent with the gene being a tumor suppressor. A human sequence (PTC) with strong homology to the Drosophila segment polarity gene, patched, was isolated from a YAC and cosmid contig of the NBCCS region. Mutation analysis revealed alterations of PTC in NBCCS patients and in related tumors. We propose that a reduction in expression of the patched gene can lead to the developmental abnormalities observed in the syndrome and that complete loss of patched function contributes to transformation of certain cell types.
痣样基底细胞癌综合征(NBCCS)是一种常染色体显性疾病,其特征为多发性基底细胞癌(BCC)、手掌和足底凹陷、颌角角化囊肿、多种其他肿瘤以及发育异常。NBCCS定位于9号染色体长臂22.3区。家族性和散发性BCC在该区域显示杂合性缺失,这与该基因作为肿瘤抑制基因相符。从NBCCS区域的一个酵母人工染色体(YAC)和黏粒重叠群中分离出了一个与果蝇节段极性基因patched具有高度同源性的人类序列(PTC)。突变分析显示NBCCS患者及其相关肿瘤中PTC发生了改变。我们提出,patched基因表达的降低可导致该综合征中所观察到的发育异常,而patched功能的完全丧失则促使某些细胞类型发生转化。