Yang Hongbo, Yan Kemin, Wang Linjie, Gong Fengying, Jin Zimeng, Zhu Huijuan
Department of Endocrinology, Peking Union Medical College Hospital, Beijing 100730, P.R. China.
Exp Ther Med. 2019 Aug;18(2):1309-1314. doi: 10.3892/etm.2019.7645. Epub 2019 Jun 4.
Familial neurohypophyseal diabetes insipidus (FNDI) is a rare single-gene disorder caused by mutations of the arginine vasopressin-neurophysin II (AVP-NPII) gene. These changes impair the release of vasopressin from the posterior pituitary gland. In the present study, the AVP-NPII gene of a Chinese adult patient with central diabetes insipidus, the patient's symptomatic mother and an asymptomatic sister of the patient was sequenced. Examination of the family history revealed cases of FNDI across four generations. Gene sequencing analysis revealed a novel heterozygous mutation, c.268A>T (p.Lys90Ter), in exon 2 of the AVP-NPII gene, in the patient and the patient's mother, which led to the loss of 6 cysteine residues and aberrant disulfide bonds, which is predicted to alter the mature protein structure. The present study identified a novel heterozygous nonsense mutation of the AVP-NPII gene associated with FNDI, which broadens the spectrum of known mutations associated with this disorder and contributes to the understanding of its molecular basis.
家族性神经垂体性尿崩症(FNDI)是一种罕见的单基因疾病,由精氨酸加压素 - 神经垂体素II(AVP - NPII)基因突变引起。这些变化会损害垂体后叶释放加压素。在本研究中,对一名患有中枢性尿崩症的中国成年患者、该患者有症状的母亲以及该患者无症状的妹妹的AVP - NPII基因进行了测序。家族史调查显示四代人中均有FNDI病例。基因测序分析在患者及其母亲的AVP - NPII基因第2外显子中发现了一个新的杂合突变,即c.268A>T(p.Lys90Ter),该突变导致6个半胱氨酸残基缺失和异常二硫键形成,预计会改变成熟蛋白的结构。本研究鉴定出一种与FNDI相关的AVP - NPII基因新的杂合无义突变,拓宽了与该疾病相关的已知突变谱,有助于理解其分子基础。