Activation of vasopressin neurons leads to phenotype progression in a mouse model for familial neurohypophysial diabetes insipidus

Maiko Hiroi, Yoshiaki Morishita, Masayuki Hayashi, Nobuaki Ozaki, Yoshihisa Sugimura, Hiroshi Nagasaki, Akira Shiota, Yutaka Oiso, Hiroshi Arima

Research output: Contribution to journalArticle

14 Citations (Scopus)

Abstract

Familial neurohypophysial diabetes insipidus (FNDI) is a rare disease that is inherited in an autosomal dominant manner. In a previous study, we made a mouse model for FNDI, which showed progressive polyuria accompanied by inclusion bodies in the arginine vasopressin (AVP) neurons formed by aggregates in the endoplasmic reticulum. The present study was conducted to determine whether the activities of AVP neurons are related to the phenotype progression in the FNDI model. In the first experiment, female heterozygous mice were administered either desmopressin (dDAVP) or a vehicle (control) subcutaneously with osmotic minipumps for 30 days. The dDAVP treatment significantly decreased the urine volume, AVP mRNA expression, and inclusion bodies in the AVP neurons. Urine volume in the dDAVP group remained significantly less than the control for 14 days even after the minipumps were removed. In the second experiment, the males were fed either a 0.2% Na or 2.0% Na diet for 6 mo. Urine AVP excretion was significantly increased in the 2.0% Na group compared with the 0.2% Na group for the first 2 mo but gradually decreased thereafter. Throughout the experiments, urine volume increased progressively in the 2.0% Na group but not in the 0.2% Na group. Immunohistochemical analyses revealed that inclusion bodies in the AVP cells had signifi-cantly increased in the 2.0% Na compared with the 0.2% Na group. These data demonstrated that activation of AVP neurons could accelerate the aggregate formation as well as the progression of the polyuria in the FNDI model mice.

Original languageEnglish
JournalAmerican Journal of Physiology - Regulatory Integrative and Comparative Physiology
Volume298
Issue number2
DOIs
Publication statusPublished - 01-02-2010

Fingerprint

Neurogenic Diabetes Insipidus
Arginine Vasopressin
Vasopressins
Phenotype
Neurons
Inclusion Bodies
Urine
Polyuria
Deamino Arginine Vasopressin
Rare Diseases
Endoplasmic Reticulum
Diet
Messenger RNA

All Science Journal Classification (ASJC) codes

  • Physiology
  • Physiology (medical)

Cite this

Hiroi, Maiko ; Morishita, Yoshiaki ; Hayashi, Masayuki ; Ozaki, Nobuaki ; Sugimura, Yoshihisa ; Nagasaki, Hiroshi ; Shiota, Akira ; Oiso, Yutaka ; Arima, Hiroshi. / Activation of vasopressin neurons leads to phenotype progression in a mouse model for familial neurohypophysial diabetes insipidus. In: American Journal of Physiology - Regulatory Integrative and Comparative Physiology. 2010 ; Vol. 298, No. 2.
@article{10b7315b46bb43a78c032e34c1d372c8,
title = "Activation of vasopressin neurons leads to phenotype progression in a mouse model for familial neurohypophysial diabetes insipidus",
abstract = "Familial neurohypophysial diabetes insipidus (FNDI) is a rare disease that is inherited in an autosomal dominant manner. In a previous study, we made a mouse model for FNDI, which showed progressive polyuria accompanied by inclusion bodies in the arginine vasopressin (AVP) neurons formed by aggregates in the endoplasmic reticulum. The present study was conducted to determine whether the activities of AVP neurons are related to the phenotype progression in the FNDI model. In the first experiment, female heterozygous mice were administered either desmopressin (dDAVP) or a vehicle (control) subcutaneously with osmotic minipumps for 30 days. The dDAVP treatment significantly decreased the urine volume, AVP mRNA expression, and inclusion bodies in the AVP neurons. Urine volume in the dDAVP group remained significantly less than the control for 14 days even after the minipumps were removed. In the second experiment, the males were fed either a 0.2{\%} Na or 2.0{\%} Na diet for 6 mo. Urine AVP excretion was significantly increased in the 2.0{\%} Na group compared with the 0.2{\%} Na group for the first 2 mo but gradually decreased thereafter. Throughout the experiments, urine volume increased progressively in the 2.0{\%} Na group but not in the 0.2{\%} Na group. Immunohistochemical analyses revealed that inclusion bodies in the AVP cells had signifi-cantly increased in the 2.0{\%} Na compared with the 0.2{\%} Na group. These data demonstrated that activation of AVP neurons could accelerate the aggregate formation as well as the progression of the polyuria in the FNDI model mice.",
author = "Maiko Hiroi and Yoshiaki Morishita and Masayuki Hayashi and Nobuaki Ozaki and Yoshihisa Sugimura and Hiroshi Nagasaki and Akira Shiota and Yutaka Oiso and Hiroshi Arima",
year = "2010",
month = "2",
day = "1",
doi = "10.1152/ajpregu.00529.2009",
language = "English",
volume = "298",
journal = "American Journal of Physiology - Regulatory Integrative and Comparative Physiology",
issn = "0363-6119",
publisher = "American Physiological Society",
number = "2",

}

Activation of vasopressin neurons leads to phenotype progression in a mouse model for familial neurohypophysial diabetes insipidus. / Hiroi, Maiko; Morishita, Yoshiaki; Hayashi, Masayuki; Ozaki, Nobuaki; Sugimura, Yoshihisa; Nagasaki, Hiroshi; Shiota, Akira; Oiso, Yutaka; Arima, Hiroshi.

In: American Journal of Physiology - Regulatory Integrative and Comparative Physiology, Vol. 298, No. 2, 01.02.2010.

Research output: Contribution to journalArticle

TY - JOUR

T1 - Activation of vasopressin neurons leads to phenotype progression in a mouse model for familial neurohypophysial diabetes insipidus

AU - Hiroi, Maiko

AU - Morishita, Yoshiaki

AU - Hayashi, Masayuki

AU - Ozaki, Nobuaki

AU - Sugimura, Yoshihisa

AU - Nagasaki, Hiroshi

AU - Shiota, Akira

AU - Oiso, Yutaka

AU - Arima, Hiroshi

PY - 2010/2/1

Y1 - 2010/2/1

N2 - Familial neurohypophysial diabetes insipidus (FNDI) is a rare disease that is inherited in an autosomal dominant manner. In a previous study, we made a mouse model for FNDI, which showed progressive polyuria accompanied by inclusion bodies in the arginine vasopressin (AVP) neurons formed by aggregates in the endoplasmic reticulum. The present study was conducted to determine whether the activities of AVP neurons are related to the phenotype progression in the FNDI model. In the first experiment, female heterozygous mice were administered either desmopressin (dDAVP) or a vehicle (control) subcutaneously with osmotic minipumps for 30 days. The dDAVP treatment significantly decreased the urine volume, AVP mRNA expression, and inclusion bodies in the AVP neurons. Urine volume in the dDAVP group remained significantly less than the control for 14 days even after the minipumps were removed. In the second experiment, the males were fed either a 0.2% Na or 2.0% Na diet for 6 mo. Urine AVP excretion was significantly increased in the 2.0% Na group compared with the 0.2% Na group for the first 2 mo but gradually decreased thereafter. Throughout the experiments, urine volume increased progressively in the 2.0% Na group but not in the 0.2% Na group. Immunohistochemical analyses revealed that inclusion bodies in the AVP cells had signifi-cantly increased in the 2.0% Na compared with the 0.2% Na group. These data demonstrated that activation of AVP neurons could accelerate the aggregate formation as well as the progression of the polyuria in the FNDI model mice.

AB - Familial neurohypophysial diabetes insipidus (FNDI) is a rare disease that is inherited in an autosomal dominant manner. In a previous study, we made a mouse model for FNDI, which showed progressive polyuria accompanied by inclusion bodies in the arginine vasopressin (AVP) neurons formed by aggregates in the endoplasmic reticulum. The present study was conducted to determine whether the activities of AVP neurons are related to the phenotype progression in the FNDI model. In the first experiment, female heterozygous mice were administered either desmopressin (dDAVP) or a vehicle (control) subcutaneously with osmotic minipumps for 30 days. The dDAVP treatment significantly decreased the urine volume, AVP mRNA expression, and inclusion bodies in the AVP neurons. Urine volume in the dDAVP group remained significantly less than the control for 14 days even after the minipumps were removed. In the second experiment, the males were fed either a 0.2% Na or 2.0% Na diet for 6 mo. Urine AVP excretion was significantly increased in the 2.0% Na group compared with the 0.2% Na group for the first 2 mo but gradually decreased thereafter. Throughout the experiments, urine volume increased progressively in the 2.0% Na group but not in the 0.2% Na group. Immunohistochemical analyses revealed that inclusion bodies in the AVP cells had signifi-cantly increased in the 2.0% Na compared with the 0.2% Na group. These data demonstrated that activation of AVP neurons could accelerate the aggregate formation as well as the progression of the polyuria in the FNDI model mice.

UR - http://www.scopus.com/inward/record.url?scp=75449096144&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=75449096144&partnerID=8YFLogxK

U2 - 10.1152/ajpregu.00529.2009

DO - 10.1152/ajpregu.00529.2009

M3 - Article

VL - 298

JO - American Journal of Physiology - Regulatory Integrative and Comparative Physiology

JF - American Journal of Physiology - Regulatory Integrative and Comparative Physiology

SN - 0363-6119

IS - 2

ER -