Lin C K, Hakakha M J, Nakamoto J M, Englund A T, Brickman A S, Scott M L, Van Dop C
Division of Pediatric Endocrinology, UCLA School of Medicine 90024.
Biochem Biophys Res Commun. 1992 Nov 30;189(1):343-9. doi: 10.1016/0006-291x(92)91564-7.
Pseudohypoparathyroidism type Ia (PHP-Ia), an inherited multi-hormone resistance syndrome, is associated with deficient cellular activity of the alpha-subunit of the guanine nucleotide-binding protein (Gs alpha) that stimulates adenylyl cyclase. We determined prevalence of three recently described mutations in exons 1 and 10 of the Gs alpha gene among 24 unrelated patients with PHP-Ia. Restriction analysis was used to detect two mutations that produce unique RFLPs, and allele-specific oligonucleotide hybridization was used to detect the other mutation. As none of these mutations were not found, genomic DNA was analyzed with denaturing gradient gel electrophoresis to screen for other mutations in exon 10. Mutations of the initiation codon and exon 10 in the Gs alpha gene thus rarely (< or = 4% each) cause PHP-Ia and the Gs alpha gene mutations causing PHP-Ia are heterogeneous and unique to each pedigree.
I型假性甲状旁腺功能减退症(PHP-Ia)是一种遗传性多激素抵抗综合征,与刺激腺苷酸环化酶的鸟嘌呤核苷酸结合蛋白(Gsα)α亚基的细胞活性不足有关。我们在24例无亲缘关系的PHP-Ia患者中确定了Gsα基因第1外显子和第10外显子中最近描述的三种突变的发生率。采用限制性分析检测产生独特限制性片段长度多态性(RFLP)的两种突变,采用等位基因特异性寡核苷酸杂交检测另一种突变。由于未发现这些突变中的任何一种,因此用变性梯度凝胶电泳分析基因组DNA,以筛选第10外显子中的其他突变。因此,Gsα基因起始密码子和第10外显子的突变很少(每种<或=4%)导致PHP-Ia,并且导致PHP-Ia的Gsα基因突变是异质性的,且每个家系都有其独特性。