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PMID: 9600992 Published · ppublish English Journal Article Research Support, Non-U.S. Gov't Research Support, U.S. Gov't, P.H.S.

Altered brain neurotransmitter receptors in transgenic mice expressing a portion of an abnormal human huntington disease gene.

Cha JH, Kosinski CM, Kerner JA, Alsdorf SA, Mangiarini L, Davies SW, Penney JB, Bates GP, Young AB

Abstract

Loss of neurotransmitter receptors, especially glutamate and dopamine receptors, is one of the pathologic hallmarks of brains of patients with Huntington disease (HD). Transgenic mice that express exon 1 of an abnormal human HD gene (line R6/2) develop neurologic symptoms at 9-11 weeks of age through an unknown mechanism. Analysis of glutamate receptors (GluRs) in symptomatic 12-week-old R6/2 mice revealed decreases compared with age-matched littermate controls in the type 1 metabotropic GluR (mGluR1), mGluR2, mGluR3, but not the mGluR5 subtype of G protein-linked mGluR, as determined by [3H]glutamate receptor binding, protein immunoblotting, and in situ hybridization. Ionotropic alpha-amino-3-hydroxy-5-methyl-4-isoxazolepropionic acid and kainate receptors were also decreased, while N-methyl-D-aspartic acid receptors were not different compared with controls. Other neurotransmitter receptors known to be affected in HD were also decreased in R6/2 mice, including dopamine and muscarinic cholinergic, but not gamma-aminobutyric acid receptors. D1-like and D2-like dopamine receptor binding was drastically reduced to one-third of control in the brains of 8- and 12-week-old R6/2 mice. In situ hybridization indicated that mGluR and D1 dopamine receptor mRNA were altered as early as 4 weeks of age, long prior to the onset of clinical symptoms. Thus, altered expression of neurotransmitter receptors precedes clinical symptoms in R6/2 mice and may contribute to subsequent pathology.

MeSH Terms
Animals Brain/metabolism Dopamine/metabolism Glutamic Acid/metabolism Humans Huntingtin Protein Huntington Disease/genetics,metabolism Mice Mice, Transgenic Nerve Tissue Proteins/biosynthesis,genetics Nuclear Proteins/biosynthesis,genetics Radioligand Assay Receptors, Dopamine/biosynthesis Receptors, Glutamate/biosynthesis Transfection
Chemicals
HTT protein, human Htt protein, mouse Huntingtin Protein Nerve Tissue Proteins Nuclear Proteins Receptors, Dopamine Receptors, Glutamate Glutamic Acid Dopamine
Authors & Affiliations
9 authors, click to expand affiliations / ORCID
Cha J H
Department of Neurology, Massachusetts General Hospital, Boston, MA, 02114, USA.
Kosinski C M
Kerner J A
Alsdorf S A
Mangiarini L
Davies S W
Penney J B
Bates G P
Young A B
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Article Info
Journal
Proceedings of the National Academy of Sciences of the United States of America
Abbr.
Proc Natl Acad Sci U S A
ISSN
0027-8424
Published
1998-05-26
Pages
6480-5
Language
English
Region
United States
NLM ID
7505876
PMCID
PMC27817
Subset
IM
Grants
NIA NIH HHS · AG 11337 · United States
NINDS NIH HHS · NS01916 · United States
NIA NIH HHS · R37 AG013617 · United States
NIA NIH HHS · R01 AG013617 · United States
NINDS NIH HHS · NS31579 · United States
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