Clinical features and molecular analysis of arginine-vasopressin neurophysin II gene in long-term follow-up patients with idiopathic central diabetes insipidus

Arq Bras Endocrinol Metabol. 2010 Mar;54(3):269-73. doi: 10.1590/s0004-27302010000300004.

Abstract

Introduction: Central diabetes insipidus (DI) characterized by polyuria, polydipsia and inability to concentrate urine, has different etiologies including genetic, autoimmune, post-traumatic, among other causes. Autosomal dominant central DI presents the clinical feature of a progressive decline of arginine-vasopressin (AVP) secretion.

Objective: In this study, we characterized the clinical features and sequenced the AVP-NPII gene of seven long-term follow-up patients with idiopathic central DI in an attempt to determine whether a genetic cause would be involved.

Methods: The diagnosis of central DI was established by fluid deprivation test and hyper-tonic saline infusion. For molecular analysis, genomic DNA was extracted and the AVP-NPII gene was amplified by polymerase chain reaction and sequenced.

Results: Sequencing analysis revealed a homozygous guanine insertion in the intron 2 (IVS2 +28 InsG) of the AVP-NPII gene in four patients, which represents an alternative gene assembly. No mutation in the code region of the AVP-NPII gene was found.

Conclusions: The homozygous guanine insertion in intron 2 (IVS2 +28 InsG) is unlikely to contribute to the AVP-NPII gene modulation in DI. In addition, the etiology of idiopathic central DI in children may not be apparent even after long-term follow-up, and requires continuous etiological surveillance.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Child
  • Child, Preschool
  • Diabetes Insipidus, Neurogenic / diagnosis*
  • Diabetes Insipidus, Neurogenic / genetics*
  • Female
  • Follow-Up Studies
  • Humans
  • Infant
  • Introns / genetics
  • Male
  • Mutagenesis, Insertional / genetics
  • Neurophysins / genetics*
  • Protein Precursors / genetics*
  • Vasopressins / genetics*

Substances

  • AVP protein, human
  • Neurophysins
  • Protein Precursors
  • Vasopressins