Kharrat Maher, Tardy Véronique, M'Rad Ridha, Maazoul Faouzi, Jemaa Lamia Ben, Refaï Mohamed, Morel Yves, Chaabouni Habiba
Laboratoire de Génétique Humaine, Faculté de Médecine de Tunis, 1006, Tunisie.
J Clin Endocrinol Metab. 2004 Jan;89(1):368-74. doi: 10.1210/jc.2003-031056.
Congenital adrenal hyperplasia (CAH) is a group of autosomal recessive disorders mainly due to defects in the steroid 21-hydroxylase (CYP21) gene. To determine the mutational spectrum in the Tunisian CAH population, the CYP21 active gene was analyzed in 51 unrelated patients using our cascade strategy (digestion by restriction enzyme, sequencing). All patients had a classical form of 21-hydroxylase deficiency. Mutations were detected in over 94% of the chromosomes examined. The most frequent mutation in the Tunisian CAH population was found to be Q318X, with large prevalence (35.3%), in contrast to 0.5-13.8% described in other series. Incidence of other mutations does not differ, as previously described: large deletions (19.6%), mutation in intron 2 (17.6%), and I172N (10.8%). Four novel mutations were found in four patients with the salt-wasting form. These four novel mutations include three point mutations that have not been reported to occur in the CYP21P pseudogene: R483W, W19X, 2669insC, and one small conversion of DNA sequence from exon 5 to exon 8. Our results have shown a good genotype/phenotype correlation in the case of most mutations. This is the first report of screening for mutations of 21-hydroxylase gene in the Tunisian population and even in the Arab population.
先天性肾上腺皮质增生症(CAH)是一组常染色体隐性疾病,主要由类固醇21-羟化酶(CYP21)基因缺陷引起。为确定突尼斯CAH患者群体中的突变谱,我们采用级联策略(限制性酶切、测序)对51例无亲缘关系的患者的CYP21活性基因进行了分析。所有患者均患有典型的21-羟化酶缺乏症。在所检测的染色体中,超过94%检测到了突变。突尼斯CAH患者群体中最常见的突变是Q318X,其发生率很高(35.3%),而其他研究系列报道的发生率为0.5%-13.8%。其他突变的发生率与之前描述的无差异:大片段缺失(19.6%)、内含子2突变(17.6%)和I172N(10.8%)。在4例失盐型患者中发现了4种新突变。这4种新突变包括3种未报道在CYP21P假基因中出现的点突变:R483W、W19X、2669insC,以及1种从外显子5到外显子8的小DNA序列转换。我们的结果表明,大多数突变情况下基因型与表型具有良好的相关性。这是首次在突尼斯人群甚至阿拉伯人群中筛查21-羟化酶基因突变的报告。