Department of Endocrinology, Shandong Provincial Hospital Affiliated to Shandong University, Jinan, Shandong 250021, P.R. China.
Shandong Clinical Medical Center of Endocrinology and Metabolism, Jinan, Shandong 250021, P.R. China.
Mol Med Rep. 2018 Mar;17(3):4265-4272. doi: 10.3892/mmr.2018.8391. Epub 2018 Jan 8.
21‑Hydroxylase deficiency (21‑OHD) is the most common cause of congenital adrenal hyperplasia. Inherited in an autosomal recessive manner, 21‑OHD is caused by mutations in the cytochrome P450 family 21 subfamily A member 2 (CYP21A2) gene. The present study was designed to investigate the genetic characteristics of one Chinese pedigree and to identify the genotype‑phenotype association, thereby facilitating the precise diagnosis of 21‑OHD at the molecular level. Members of a Chinese family with 21‑OHD were screened for mutations in the CYP21A2 gene. Clinical data and biochemical parameters, including androgen and derivatives, were collected. Complete DNA sequencing and multiplex ligation‑dependent probe amplification (MLPA) were utilized to analyze the genetic variations in the full‑length CYP21A2 gene. A C‑T transition located in exon 8 of the CYP21A2 gene, leading to the predicted amino acid residue change from Arg to Trp at codon 342, was identified in the mother and four sisters. Additionally, heterozygous deletion mutations of exons 1, 3, 4, 6 and 7 of paternal origin were detected in the four sisters by MLPA analysis. During the one‑year follow‑up, the four sisters exhibited symptom improvement following treatment with glucocorticoids, and the proband and one sister successfully conceived. The results of the present study demonstrated that novel compound heterozygous variations in the CYP21A2 gene may be causative agents of 21‑OHD, providing insights into the functions of this gene and a more comprehensive understanding of the disorder.
21-羟化酶缺乏症(21-OHD)是最常见的先天性肾上腺皮质增生症的病因。21-OHD 以常染色体隐性遗传方式遗传,是由于细胞色素 P450 家族 21 亚家族 A 成员 2(CYP21A2)基因的突变引起的。本研究旨在调查一个中国家系的遗传特征,并确定基因型-表型相关性,从而促进 21-OHD 在分子水平上的精确诊断。对一个患有 21-OHD 的中国家庭的成员进行 CYP21A2 基因突变筛查。收集临床数据和生化参数,包括雄激素及其衍生物。利用全基因组测序和多重连接依赖性探针扩增(MLPA)分析 CYP21A2 基因全长的遗传变异。在 CYP21A2 基因的 8 号外显子中发现了一个 C-T 转换,导致预测的氨基酸残基从精氨酸变为色氨酸,位于密码子 342。在母亲和四个姐妹中发现了 CYP21A2 基因的外显子 1、3、4、6 和 7 的杂合性缺失突变。通过 MLPA 分析,发现来自父亲的外显子 1、3、4、6 和 7 的杂合性缺失突变。在一年的随访中,四个姐妹在接受糖皮质激素治疗后症状得到改善,先证者和一个姐妹成功怀孕。本研究结果表明,CYP21A2 基因的新型复合杂合变异可能是 21-OHD 的致病原因,为该基因的功能提供了见解,并对该疾病有了更全面的认识。