Spectrum of PTCH1 mutations in French patients with Gorlin syndrome

被引:77
作者
Boutet, N
Bignon, YJ
Drouin-Garraud, V
Sarda, P
Longy, M
Lacombe, D
Gorry, P [1 ]
机构
[1] Inst Bergonie, Lab Genet Oncol, F-33076 Bordeaux, France
[2] Ctr Jean Perrin, Unite Oncogenet, Clermont Ferrand, France
[3] CHU Charles Nicolle, Unite Genet Clin, Rouen, France
[4] CHU Arnaud Villeneuve, Unite Genet Med, Montpellier, France
[5] CHU Pellegrin Enfants, Gen Med Serv, Bordeaux, France
关键词
basal cell; carcinoma; genetic; germline mutation; skin disease;
D O I
10.1046/j.1523-1747.2003.12423.x
中图分类号
R75 [皮肤病学与性病学];
学科分类号
100206 ;
摘要
Gorlin syndrome or nevoid basal cell carcinoma syndrome is an autosomal dominant disease characterized by developmental abnormalities and a predisposition to cancers. The responsible gene for this syndrome is the PTCH tumor suppressor gene encoding for the Sonic Hedgehog receptor. We screened for PTCH mutations in 65 French Gorlin syndrome families or sporadic cases for the first time. Nineteen novel mutations and five new polymorphisms were identified in this group of patients. One microdeletion without frameshift underlines the importance of one amino acid for Ptc receptor function. Although no mutation hot-spot was described, we identified a recurrent mutation.
引用
收藏
页码:478 / 481
页数:4
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