Literature DB >> 18395613

Increased thirst and drinking in Huntington's disease and the R6/2 mouse.

Nigel I Wood1, Anna O G Goodman, Jorien M M van der Burg, Véronique Gazeau, Patrik Brundin, Maria Björkqvist, Asa Petersén, Sarah J Tabrizi, Roger A Barker, A Jennifer Morton.   

Abstract

While Huntington's disease (HD) is a condition that primarily involves the basal ganglia, there is evidence to suggest that the hypothalamus is also affected. Because the osmoreceptors regulating thirst are situated in the circumventricular region of the hypothalamus, we were interested in whether altered thirst is a part of the HD phenotype. We used the LABORAS behavioural monitoring system and water consumption to show that drinking behaviour was abnormal in R6/2 mice. By 10 weeks of age, R6/2 mice spent significantly more time drinking and drank a greater volume than their wild-type (WT) littermates. The numbers of immunoreactive vasopressin neurons in the paraventricular nucleus (PVN) of the hypothalamus in R6/2 mice were significantly decreased from 8 weeks of age, suggesting that the change in drinking behaviour may be the result of hypothalamic dysfunction. We gave a xerostomia (dry mouth) questionnaire to HD patients and control subjects, and also measured their urine osmolality and serum vasopressin. The mean total xerostomia score was significantly higher in HD patients than in controls, indicating greater thirst in HD patients. Urine osmolality was unaffected in HD patients up to clinical stage III, and none of the patients had diabetes. However, serum vasopressin was increased, suggesting a dysregulation in the control of hypothalamic vasopressin release. A dry mouth can affect taste, mastication and swallowing, all of which may contribute to the significant weight loss seen in both HD patients and R6/2 mice, as can dehydration. We suggest that increased thirst may be an important and clinically relevant biomarker for the study of disease progression in HD.

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Year:  2008        PMID: 18395613     DOI: 10.1016/j.brainresbull.2007.12.007

Source DB:  PubMed          Journal:  Brain Res Bull        ISSN: 0361-9230            Impact factor:   4.077


  24 in total

1.  Dysfunctions in circadian behavior and physiology in mouse models of Huntington's disease.

Authors:  Takashi Kudo; Analyne Schroeder; Dawn H Loh; Dika Kuljis; Maria C Jordan; Kenneth P Roos; Christopher S Colwell
Journal:  Exp Neurol       Date:  2010-12-22       Impact factor: 5.330

2.  Mutant huntingtin fragment selectively suppresses Brn-2 POU domain transcription factor to mediate hypothalamic cell dysfunction.

Authors:  Tomoyuki Yamanaka; Asako Tosaki; Haruko Miyazaki; Masaru Kurosawa; Yoshiaki Furukawa; Mizuki Yamada; Nobuyuki Nukina
Journal:  Hum Mol Genet       Date:  2010-02-25       Impact factor: 6.150

3.  Responses to environmental enrichment differ with sex and genotype in a transgenic mouse model of Huntington's disease.

Authors:  Nigel I Wood; Valentina Carta; Stefan Milde; Elizabeth A Skillings; Catherine J McAllister; Y L Mabel Ang; Alasdair Duguid; Nadeev Wijesuriya; Samira Mohd Afzal; Joe X Fernandes; T W Leong; A Jennifer Morton; Jennifer Morton
Journal:  PLoS One       Date:  2010-02-12       Impact factor: 3.240

4.  Sleep and circadian dysfunction in neurodegenerative disorders: insights from a mouse model of Huntington's disease.

Authors:  Dika Kuljis; Analyne M Schroeder; Takashi Kudo; Dawn H Loh; David L Willison; Christopher S Colwell
Journal:  Minerva Pneumol       Date:  2012-09

5.  Normalizing glucocorticoid levels attenuates metabolic and neuropathological symptoms in the R6/2 mouse model of huntington's disease.

Authors:  Brett D Dufour; Jodi L McBride
Journal:  Neurobiol Dis       Date:  2018-10-05       Impact factor: 5.996

6.  Platelet-derived extracellular vesicles in Huntington's disease.

Authors:  Hélèna L Denis; Jérôme Lamontagne-Proulx; Isabelle St-Amour; Sarah L Mason; Andreas Weiss; Sylvain Chouinard; Roger A Barker; Eric Boilard; Francesca Cicchetti
Journal:  J Neurol       Date:  2018-09-12       Impact factor: 4.849

7.  Sexually dimorphic serotonergic dysfunction in a mouse model of Huntington's disease and depression.

Authors:  Thibault Renoir; Michelle S Zajac; Xin Du; Terence Y Pang; Leah Leang; Caroline Chevarin; Laurence Lanfumey; Anthony J Hannan
Journal:  PLoS One       Date:  2011-07-08       Impact factor: 3.240

8.  Progression of basal ganglia pathology in heterozygous Q175 knock-in Huntington's disease mice.

Authors:  Yunping Deng; Hongbing Wang; Marion Joni; Radhika Sekhri; Anton Reiner
Journal:  J Comp Neurol       Date:  2020-09-20       Impact factor: 3.215

Review 9.  Mouse models of polyglutamine diseases: review and data table. Part I.

Authors:  Maciej Figiel; Wojciech J Szlachcic; Pawel M Switonski; Agnieszka Gabka; Wlodzimierz J Krzyzosiak
Journal:  Mol Neurobiol       Date:  2012-09-07       Impact factor: 5.590

10.  High-Throughput Automated Phenotyping of Two Genetic Mouse Models of Huntington's Disease.

Authors:  Fuat Balci; Stephen Oakeshott; Jul Lea Shamy; Bassem F El-Khodor; Igor Filippov; Richard Mushlin; Russell Port; David Connor; Ahmad Paintdakhi; Liliana Menalled; Sylvie Ramboz; David Howland; Seung Kwak; Dani Brunner
Journal:  PLoS Curr       Date:  2013-07-11
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