Rittig S, Robertson G L, Siggaard C, Kovács L, Gregersen N, Nyborg J, Pedersen E B
Research Laboratory of Nephrology and Hypertension, Skejby Hospital, Aarhus University, Denmark.
Am J Hum Genet. 1996 Jan;58(1):107-17.
Familial neurohypophyseal diabetes insipidus (FNDI) is an autosomal dominant disorder characterized by progressive postnatal deficiency of arginine vasopressin as a result of mutation in the gene that encodes the hormone. To determine the extent of mutations in the coding region that produce the phenotype, we studied members of 17 unrelated kindreds with the disorder. We sequenced all 3 exons of the gene by using a rapid, direct dye-terminator method and found the causative mutation in each kindred. In four kindreds, the mutations were each identical to mutations described in other affected families. In the other 13 kindreds each mutation was unique. There were two missense mutations that altered the cleavage region of the signal peptide, seven missense mutations in exon 2, which codes for the conserved portion of the protein, one nonsense mutation in exon 2, and three nonsense mutations in exon 3. These findings, together with the clinical features of FNDI, suggest that each of the mutations exerts an effect by directing the production of a pre-prohormone that cannot be folded, processed, or degraded properly and eventually destroys vasopressinergic neurons.
家族性神经垂体性尿崩症(FNDI)是一种常染色体显性疾病,其特征为出生后由于编码该激素的基因突变导致精氨酸加压素逐渐缺乏。为了确定产生该表型的编码区突变范围,我们研究了17个患有该疾病的不相关家族的成员。我们采用快速直接染料终止法对该基因的所有3个外显子进行测序,并在每个家族中发现了致病突变。在4个家族中,突变与其他患病家族中描述的突变相同。在其他13个家族中,每个突变都是独特的。有两个错义突变改变了信号肽的切割区域,外显子2中有7个错义突变(该外显子编码蛋白质的保守部分),外显子2中有1个无义突变,外显子3中有3个无义突变。这些发现与FNDI的临床特征一起表明,每个突变通过指导产生一种无法正确折叠、加工或降解的前激素原发挥作用,并最终破坏加压素能神经元。