Pubfacts - Scientific Publication Data
  • Categories
  • |
  • Journals
  • |
  • Authors
  • Login
  • Categories
  • Journals

Search Our Scientific Publications & Authors

Publications
  • Publications
  • Authors
find publications by category +
Translate page:

Deficiency of WFS1 leads to the impairment of AVP secretion under dehydration in male mice.

Authors:
Junki Kurimoto Hiroshi Takagi Takashi Miyata Yuichi Hodai Yohei Kawaguchi Daisuke Hagiwara Hidetaka Suga Tomoko Kobayashi Mariko Sugiyama Takeshi Onoue Yoshihiro Ito Shintaro Iwama Ryoichi Banno Katsuya Tanabe Yukio Tanizawa Hiroshi Arima

Pituitary 2021 Mar 5. Epub 2021 Mar 5.

Department of Endocrinology and Diabetes, Nagoya University Graduate School of Medicine, 65 Tsurumai-cho, Showa-ku, Nagoya, 466-8550, Japan.

Wolfram syndrome (WS) is mainly caused by mutations in the WFS1 gene and characterized by diabetes mellitus, optic atrophy, hearing loss, and central diabetes insipidus (CDI). WFS1 is an endoplasmic reticulum (ER)-resident transmembrane protein, and Wfs1 knockout (Wfs1-/-) mice, which have been used as a mouse model for WS, reportedly manifested impairment of glucose tolerance due to pancreatic β-cell loss. In the present study, we examined water balance, arginine vasopressin (AVP) secretion, and ER stress in AVP neurons of the hypothalamus in Wfs1-/- mice. There were no differences in urine volumes between Wfs1-/- and wild-type mice with free access to water. Conversely, when mice were subjected to intermittent water deprivation (WD) for 20 weeks, during which water was unavailable for 2 days a week, urine volumes were larger in Wfs1-/- mice, accompanied by lower urine AVP concentrations and urine osmolality, compared to wild-type mice. The mRNA expression of immunoglobulin heavy chain binding protein, a marker of ER stress, was significantly increased in the supraoptic nucleus and paraventricular nuclei in Wfs1-/- mice compared to wild-type mice after WD. Our results thus showed that Wfs1 knockout leads to a decrease in AVP secretion during dehydration, which could explain in part the mechanisms by which Wfs1 mutations cause CDI in humans.

Download full-text PDF

Source
http://dx.doi.org/10.1007/s11102-021-01135-6DOI Listing
March 2021

Publication Analysis

Top Keywords

wfs1-/- mice
16
avp secretion
12
wild-type mice
12
mice
9
secretion dehydration
8
urine volumes
8
compared wild-type
8
wfs1 knockout
8
avp
5
wfs1-/-
5
wfs1
5
access water
4
mice free
4
wfs1-/- wild-type
4
free access
4
conversely mice
4
deprivation 20 weeks
4
20 weeks water
4
water unavailable
4
unavailable 2 days
4

Keyword Occurance

Similar Publications

Deficiency of WFS1 leads to the impairment of AVP secretion under dehydration in male mice.

Authors:
Junki Kurimoto Hiroshi Takagi Takashi Miyata Yuichi Hodai Yohei Kawaguchi Daisuke Hagiwara Hidetaka Suga Tomoko Kobayashi Mariko Sugiyama Takeshi Onoue Yoshihiro Ito Shintaro Iwama Ryoichi Banno Katsuya Tanabe Yukio Tanizawa Hiroshi Arima

Pituitary 2021 Mar 5. Epub 2021 Mar 5.

Department of Endocrinology and Diabetes, Nagoya University Graduate School of Medicine, 65 Tsurumai-cho, Showa-ku, Nagoya, 466-8550, Japan.

Wolfram syndrome (WS) is mainly caused by mutations in the WFS1 gene and characterized by diabetes mellitus, optic atrophy, hearing loss, and central diabetes insipidus (CDI). WFS1 is an endoplasmic reticulum (ER)-resident transmembrane protein, and Wfs1 knockout (Wfs1-/-) mice, which have been used as a mouse model for WS, reportedly manifested impairment of glucose tolerance due to pancreatic β-cell loss. In the present study, we examined water balance, arginine vasopressin (AVP) secretion, and ER stress in AVP neurons of the hypothalamus in Wfs1-/- mice. Read More

View Article and Full-Text PDF
March 2021
Similar Publications

Behavioral characterization of a novel Cisd2 mutant mouse.

Authors:
Christiann H Gaines Angela E Snyder Robin B Ervin Joseph Farrington Kenneth Walsh Sarah A Schoenrock Lisa M Tarantino

Behav Brain Res 2021 May 18;405:113187. Epub 2021 Feb 18.

Department of Genetics, University of North Carolina at Chapel Hill, NC, United States; Division of Pharmacotherapy and Experimental Therapeutics, Eshelman School of Pharmacy, University of North Carolina at Chapel Hill, NC, United States. Electronic address:

Wolfram syndrome (WFS) is a rare autosomal recessive disorder characterized by diabetes mellitus and insipidus, progressive optic atrophy and sensorineural deafness. An increased incidence of psychiatric disorders has also been reported in WFS patients. There are two subtypes of WFS. Read More

View Article and Full-Text PDF
May 2021
Similar Publications

Wfs1 and Related Molecules as Key Candidate Genes in the Hippocampus of Depression.

Authors:
Jing Yang Chaoqin Chen Xiaoyuan Jin Lu Liu Jiajia Lin Xianhui Kang Shengmei Zhu

Front Genet 2020 22;11:589370. Epub 2021 Jan 22.

Department of Anesthesiology, The First Affiliated Hospital, Zhejiang University School of Medicine, Hangzhou, China.

Background: Depression is a prevalent mental disorder, which is difficult to diagnose and treat due to its unclear pathogenic mechanisms. The discovery of novel and effective therapeutic targets for depression is urgently needed. The hippocampus is a crucial region involved in depression and has been a therapeutic target for many antidepressants. Read More

View Article and Full-Text PDF
January 2021
Similar Publications

Postnatal maturation of calcium signaling in islets of Langerhans from neonatal mice.

Authors:
Hannah L West Kathryn L Corbin Cathleen V D'Angelo Lauren M Donovan Ishrat Jahan Guoqiang Gu Craig S Nunemaker

Cell Calcium 2021 03 28;94:102339. Epub 2020 Dec 28.

Dept. of Biomedical Sciences, Heritage College of Osteopathic Medicine, Ohio University, Athens, OH, USA; Diabetes Institute, Heritage College of Osteopathic Medicine, Ohio University, Athens, OH, USA. Electronic address:

Pancreatic islet cells develop mature physiological responses to glucose and other fuels postnatally. In this study, we used fluorescence imaging techniques to measure changes in intracellular calcium ([Ca]) to compare islets isolated from mice on postnatal days 0, 4, and 12 with islets from adult CD-1 mice. In addition, we used publicly available RNA-sequencing data to compare expression levels of key genes in β-cell physiology with [Ca] data across these ages. Read More

View Article and Full-Text PDF
March 2021
Similar Publications

Multiple Retinal Anomalies in Wfs1-Deficient Mice.

Authors:
Arleta Waszczykowska Agnieszka Zmysłowska Marcin Braun Marilin Ivask Sulev Koks Piotr Jurowski Wojciech Młynarski

Diagnostics (Basel) 2020 Aug 19;10(9). Epub 2020 Aug 19.

Department of Pediatrics, Oncology and Hematology, Medical University of Łódź, Sporna 36/50 Street, 91-738 Łódź, Poland.

Background: Wolfram syndrome (WFS, OMIM: #222300) is an ultrarare autosomal recessive disorder characterized by diabetes insipidus, diabetes mellitus, optic nerve atrophy and deafness. It has been reported that the average retinal thickness in WFS patients decreases with the progression of the disease.

Aim: To investigate retinal thickness and wolframin expression disorders in Wolfram syndrome 1 gene knockout (Wfs1KO) mice compared to their wild-type (WT) littermates. Read More

View Article and Full-Text PDF
August 2020
Similar Publications
© 2021 PubFacts.
  • About PubFacts
  • Privacy Policy
  • Sitemap