Familial neurohypophyseal diabetes insipidus due to a novel mutation in the arginine vasopressin-neurophysin II gene

Eur J Endocrinol. 2011 Jul;165(1):161-5. doi: 10.1530/EJE-11-0048. Epub 2011 Apr 15.

Abstract

Background: Familial neurohypophyseal (central) diabetes insipidus (DI) is caused by mutations in the arginine vasopressin-neurophysin II (AVP-NPII) gene. The majority of cases is inherited in an autosomal dominant way. In this study, we present the clinical features of a mother and her son with autosomal dominant neurohypophyseal DI caused by a novel mutation.

Case: A thirty-four-year-old woman and her three-year-old son were evaluated because of polyuria and polydipsia since the age of 1.5 years onwards. Both patients were subjected to a water deprivation test confirming the diagnosis of central DI. Magnetic resonance imaging of the brain of the mother showed a hypothalamus without apparent abnormalities and a relatively small neurohypophysis without a hyperintense signal. Mutation analysis showed a c.322G>T (p.?/p.Glu108X) in Exon 2 of the AVP-NPII gene in both mother and son.

Discussion: This study reports neurohypophyseal DI in a mother and her son due to a novel mutation in Exon 2 of the AVP-NPII gene. Clinical and pathophysiological aspects of this disease are shortly reviewed and discussed.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Arginine Vasopressin / genetics*
  • Child, Preschool
  • Deamino Arginine Vasopressin / therapeutic use
  • Diabetes Insipidus, Neurogenic / drug therapy
  • Diabetes Insipidus, Neurogenic / genetics*
  • Female
  • Humans
  • Male
  • Neurophysins / genetics*

Substances

  • Neurophysins
  • Arginine Vasopressin
  • Deamino Arginine Vasopressin