Boroujerdi Razieh, Shariati Mohsen, Naddafnia Hosein, Rezaei Hojatolah
Counselor in Welfare Organization of Qom, Iran.
Technical Corresponding in Pouya, Genetic Counseling Clinic, Qom, Iran.
Iran J Child Neurol. 2015 Winter;9(1):103-6.
Deficiency of hypoxanthine-guanine phosphoribosyltransferase (HGPRT) is a rare inborn error of purine metabolism and is characterized by uric acid overproduction along with a variety of neurological manifestations that depend on a degree of the enzymatic deficiency. Inheritance of HPRT deficiency is X-linked recessive; thus, males are generally more affected and heterozygous females are carriers (usually asymptomatic). Human HPRT is encoded by a single structural gene on the long arm of the X chromosome at Xq26. More than 300 mutations in the HPRT1 gene have been detected. Diagnosis can be based on clinical and biochemical findings as well as enzymatic and molecular testing. Molecular diagnosis is the best way as it allows for faster and more accurate carrier and prenatal diagnosis. In this report, a new small duplication in the HPRT1 gene was found by sequencing, which has yet to be reported.
次黄嘌呤 - 鸟嘌呤磷酸核糖转移酶(HGPRT)缺乏症是一种罕见的嘌呤代谢先天性缺陷疾病,其特征是尿酸产生过多,并伴有多种神经系统表现,这些表现取决于酶缺乏的程度。HPRT缺乏症的遗传方式为X连锁隐性遗传;因此,男性通常受影响更大,而杂合子女性为携带者(通常无症状)。人类HPRT由位于X染色体长臂Xq26的单个结构基因编码。已检测到HPRT1基因有300多种突变。诊断可基于临床和生化检查结果以及酶学和分子检测。分子诊断是最佳方法,因为它能实现更快、更准确的携带者诊断和产前诊断。在本报告中,通过测序发现了HPRT1基因中的一种新的小重复,此前尚未见报道。