Colomb Evelyne, Kaplan Josseline, Garchon Henri-Jean
INSERM U580, Paris, France.
Hum Mutat. 2003 Dec;22(6):496. doi: 10.1002/humu.9197.
The CYP1B1 gene (GenBank: U56438), a member of the cytochrome P450 gene family, has been shown to be mutated in patients with primary congenital glaucoma (PCG), a rare but severely blinding form of glaucoma. Here, we have investigated CYP1B1 mutations in 31 unrelated French PCG patients. Mutations were found in 15 (48%) patients. Six of these mutations were novel. One, g.3979delA, caused a frameshift followed by a stop codon at residue 59. Two mutations, g.4547C>T (p.Q248X) and g.8167C>T (p.R444X), created a stop codon. Three other mutations, g.4499G>C (p.G232R), g.8033T>G (p.I399S), (p.N423Y), induced a significant amino acid change. Seven patients, who were of French descent, were compound heterozygotes. Six patients, whose families came from North Africa or from Portugal, carried a homozygous mutation reflecting their geographic origin. Intriguingly, one mutation, p.E229K, was present in heterozygous state in two unrelated patients. All together, these findings demonstrate the major role and the diversity of CYP1B1 mutations in French PCG patients.
细胞色素P450基因家族成员CYP1B1基因(基因库:U56438)已被证实在原发性先天性青光眼(PCG)患者中发生突变,PCG是一种罕见但致盲严重的青光眼类型。在此,我们研究了31名无亲缘关系的法国PCG患者中的CYP1B1突变情况。在15名(48%)患者中发现了突变。其中6种突变是新发现的。一种突变,g.3979delA,导致移码,随后在第59位残基处出现终止密码子。两种突变,g.4547C>T(p.Q248X)和g.8167C>T(p.R444X),产生了终止密码子。另外三种突变,g.4499G>C(p.G232R)、g.8033T>G(p.I399S)、(p.N423Y),导致了显著的氨基酸变化。7名具有法国血统的患者为复合杂合子。6名家族来自北非或葡萄牙的患者携带纯合突变,反映了他们的地理起源。有趣的是,一种突变p.E229K在两名无亲缘关系的患者中以杂合状态存在。总之,这些发现证明了CYP1B1突变在法国PCG患者中的主要作用和多样性。