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PMID: 11818550 Published · ppublish English Journal Article Research Support, Non-U.S. Gov't Research Support, U.S. Gov't, P.H.S.

Focal loss of the glutamate transporter EAAT2 in a transgenic rat model of SOD1 mutant-mediated amyotrophic lateral sclerosis (ALS).

Howland DS, Liu J, She Y, Goad B, Maragakis NJ, Kim B, Erickson J, Kulik J, DeVito L, Psaltis G, DeGennaro LJ, Cleveland DW, Rothstein JD

Abstract

Transgenic overexpression of Cu(+2)/Zn(+2) superoxide dismutase 1 (SOD1) harboring an amyotrophic lateral sclerosis (ALS)-linked familial genetic mutation (SOD1(G93A)) in a Sprague-Dawley rat results in ALS-like motor neuron disease. Motor neuron disease in these rats depended on high levels of mutant SOD1 expression, increasing from 8-fold over endogenous SOD1 in the spinal cord of young presymptomatic rats to 16-fold in end-stage animals. Disease onset in these rats was early, approximately 115 days, and disease progression was very rapid thereafter with affected rats reaching end stage on average within 11 days. Pathological abnormalities included vacuoles initially in the lumbar spinal cord and subsequently in more cervical areas, along with inclusion bodies that stained for SOD1, Hsp70, neurofilaments, and ubiquitin. Vacuolization and gliosis were evident before clinical onset of disease and before motor neuron death in the spinal cord and brainstem. Focal loss of the EAAT2 glutamate transporter in the ventral horn of the spinal cord coincided with gliosis, but appeared before motor neuron/axon degeneration. At end-stage disease, gliosis increased and EAAT2 loss in the ventral horn exceeded 90%, suggesting a role for this protein in the events leading to cell death in ALS. These transgenic rats provide a valuable resource to pursue experimentation and therapeutic development, currently difficult or impossible to perform with existing ALS transgenic mice.

MeSH Terms
Amino Acid Substitution Animals Animals, Genetically Modified Brain/metabolism,pathology Disease Models, Animal Excitatory Amino Acid Transporter 2/blood,deficiency,genetics,physiology Humans Immunohistochemistry Motor Neuron Disease/enzymology,genetics,metabolism,pathology Muscle, Skeletal/metabolism,pathology Mutagenesis, Site-Directed Rats Rats, Sprague-Dawley Spinal Cord/metabolism,pathology Superoxide Dismutase/blood,genetics Superoxide Dismutase-1 Time Factors
Chemicals
Excitatory Amino Acid Transporter 2 SOD1 protein, human Sod1 protein, mouse Sod1 protein, rat Superoxide Dismutase Superoxide Dismutase-1
Authors & Affiliations
13 authors, click to expand affiliations / ORCID
Howland David S
Department of Molecular Genetics, Wyeth Research, CN8000, Princeton, NJ 08543, USA. howland@war.wyeth.com
Liu Jian
She Yijin
Goad Beth
Maragakis Nicholas J
Kim Benjamin
Erickson Jamie
Kulik John
DeVito Lisa
Psaltis George
DeGennaro Louis J
Cleveland Don W
Rothstein Jeffrey D
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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
2002-02-05
Epub
2002-00-29
Pages
1604-9
Language
English
Region
United States
NLM ID
7505876
PMCID
PMC122237
Subset
IM
Grants
NINDS NIH HHS · R37 NS027036 · United States
NINDS NIH HHS · NS 27036 · United States
NINDS NIH HHS · NS33958 · United States
NINDS NIH HHS · R01 NS027036 · United States
NIA NIH HHS · P01 AG012992 · United States
NINDS NIH HHS · R01 NS033958 · United States
NIA NIH HHS · AG 12992 · United States
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