دورية أكاديمية
أعرض في EDS

Genetic essential tremor in gamma-aminobutyric acidA receptor alpha1 subunit knockout mice.

التفاصيل البيبلوغرافية
العنوان: Genetic essential tremor in gamma-aminobutyric acidA receptor alpha1 subunit knockout mice.
المؤلفون: Kralic JE; Department of Pharmacology, University of North Carolina School of Medicine, Chapel Hill, North Carolina 27599-7178, USA., Criswell HE, Osterman JL, O'Buckley TK, Wilkie ME, Matthews DB, Hamre K, Breese GR, Homanics GE, Morrow AL
المصدر: The Journal of clinical investigation [J Clin Invest] 2005 Mar; Vol. 115 (3), pp. 774-9.
نوع المنشور: Journal Article; Research Support, U.S. Gov't, P.H.S.
اللغة: English
بيانات الدورية: Publisher: American Society for Clinical Investigation Country of Publication: United States NLM ID: 7802877 Publication Model: Print Cited Medium: Print ISSN: 0021-9738 (Print) Linking ISSN: 00219738 NLM ISO Abbreviation: J Clin Invest Subsets: MEDLINE
أسماء مطبوعة: Publication: 1999- : Ann Arbor, MI : American Society for Clinical Investigation
Original Publication: New Haven [etc.] American Society for Clinical Investigation.
مواضيع طبية MeSH: Essential Tremor/*genetics , Essential Tremor/*metabolism , Protein Subunits/*metabolism , Receptors, GABA-A/*metabolism, Adrenergic beta-Antagonists/pharmacology ; Animals ; Anticonvulsants/pharmacology ; Disease Models, Animal ; Dizocilpine Maleate/pharmacology ; Essential Tremor/drug therapy ; Ethanol/therapeutic use ; Excitatory Amino Acid Antagonists/pharmacology ; Humans ; Mice ; Mice, Knockout ; Motor Activity/physiology ; Patch-Clamp Techniques ; Primidone/pharmacology ; Propranolol/pharmacology ; Protein Subunits/genetics ; Receptors, GABA-A/genetics
مستخلص: Essential tremor is the most common movement disorder and has an unknown etiology. Here we report that gamma-aminobutyric acidA (GABA(A)) receptor alpha1-/- mice exhibit postural and kinetic tremor and motor incoordination that is characteristic of essential tremor disease. We tested mice with essential-like tremor using current drug therapies that alleviate symptoms in essential tremor patients (primidone, propranolol, and gabapentin) and several candidates hypothesized to reduce tremor, including ethanol; the noncompetitive N-methyl-D-aspartate receptor antagonist MK-801; the adenosine A1 receptor agonist 2-chloro-N6-cyclopentyladenosine (CCPA); the GABA(A) receptor modulators diazepam, allopregnanolone, and Ro15-4513; and the L-type Ca2+ channel antagonist nitrendipine. Primidone, propranolol, and gabapentin reduced the amplitude (power) of the pathologic tremor. Nonsedative doses of ethanol eliminated tremor in mice. Diazepam, allopregnanolone, Ro15-4513, and nitrendipine had no effect or enhanced tremor, whereas MK-801 and CCPA reduced tremor. To understand the etiology of tremor in these mice, we studied the electrophysiological properties of cerebellar Purkinje cells. Cerebellar Purkinje cells in GABA(A) receptor alpha1-/- mice exhibited a profound loss of all responses to synaptic or exogenous GABA, but no differences in abundance, gross morphology, or spontaneous synaptic activity were observed. This genetic animal model elucidates a mechanism of GABAergic dysfunction in the major motor pathway and potential targets for pharmacotherapy of essential tremor.
التعليقات: Comment in: J Clin Invest. 2005 Mar;115(3):584-6. (PMID: 15765140)
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معلومات مُعتمدة: R37 AA010422 United States AA NIAAA NIH HHS; AA10422 United States AA NIAAA NIH HHS; P60 AA011605 United States AA NIAAA NIH HHS; AA09013 United States AA NIAAA NIH HHS; P50 AA011605 United States AA NIAAA NIH HHS; GM52035 United States GM NIGMS NIH HHS; MH61971 United States MH NIMH NIH HHS; U01 MH061971 United States MH NIMH NIH HHS; AA11605 United States AA NIAAA NIH HHS; R01 AA010422 United States AA NIAAA NIH HHS
المشرفين على المادة: 0 (Adrenergic beta-Antagonists)
0 (Anticonvulsants)
0 (Excitatory Amino Acid Antagonists)
0 (Protein Subunits)
0 (Receptors, GABA-A)
13AFD7670Q (Primidone)
3K9958V90M (Ethanol)
6LR8C1B66Q (Dizocilpine Maleate)
9Y8NXQ24VQ (Propranolol)
تواريخ الأحداث: Date Created: 20050315 Date Completed: 20050509 Latest Revision: 20220410
رمز التحديث: 20231215
مُعرف محوري في PubMed: PMC1052003
DOI: 10.1172/JCI23625
PMID: 15765150
قاعدة البيانات: MEDLINE
الوصف
تدمد:0021-9738
DOI:10.1172/JCI23625