A Novel Movement Disorder in Related Male Labrador Retrievers Characterized by Extreme Generalized Muscular Stiffness
Identifieur interne : 001807 ( Main/Curation ); précédent : 001806; suivant : 001808A Novel Movement Disorder in Related Male Labrador Retrievers Characterized by Extreme Generalized Muscular Stiffness
Auteurs : A. E. Vanhaesebrouck [Royaume-Uni] ; G. D. Shelton [États-Unis] ; L. Garosi [Royaume-Uni] ; T. R. Harcourt-Brown [Royaume-Uni] ; J. Couturier [Royaume-Uni] ; S. Behr [Royaume-Uni] ; R. J. Harvey [Royaume-Uni] ; N. D. Jeffery [Royaume-Uni, États-Unis] ; K. Matiasek [Allemagne] ; W. F. Blakemore [Royaume-Uni] ; N. Granger [Royaume-Uni]Source :
- Journal of Veterinary Internal Medicine [ 0891-6640 ] ; 2011-09.
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Abstract
Objectives: To describe the clinical phenotype of a new motor disorder in Labrador Retrievers. Animals and Methods: Case series study. Seven young male Labrador Retrievers presented for evaluation of stiff gait. Results: All affected dogs had generalized muscular stiffness, persistent at rest and resulting in restricted joint movements. They showed a forward flexed posture, festinating gait, and bradykinesia. Signs developed between 2 and 16 months of age and tended to stabilize in adulthood. Needle electromyogram in the conscious state showed continuous motor unit activity in resting epaxial and proximal limb muscles. This activity was abolished by general anesthesia. Muscle and nerve histopathology was normal. In 2 dogs necropsied, astrocytosis was evident throughout the spinal cord gray matter, reticular formation and caudate nuclei. Decreased neuronal counts were selectively found in the spinal cord Rexed's lamina VII, but not in VIII and IX. Pedigree analysis showed that the affected dogs were from 5 related litters. Conclusions and Clinical Importance: This new hypertonicity syndrome in Labrador Retrievers is unique because of the selective distribution of the histological lesions, the lack of progression in adulthood, and its exclusive occurrence in male dogs. Pedigree analysis suggests an X‐linked hereditary disease, although other modes of inheritance cannot be ruled out with certainty. We hypothesize that altered output from basal nuclei and reticular formation together with motor neuron disinhibition caused by a decreased number of spinal cord interneurons leads to the muscular stiffness.
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DOI: 10.1111/j.1939-1676.2011.0757.x
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Blakemore</name><affiliation wicri:level="1"><mods:affiliation>Department of Veterinary Medicine, The Queen's Veterinary School Hospital, University of Cambridge, Cambridge, UK</mods:affiliation><country xml:lang="fr">Royaume-Uni</country><wicri:regionArea>Department of Veterinary Medicine, The Queen's Veterinary School Hospital, University of Cambridge, Cambridge</wicri:regionArea></affiliation></author><author><name sortKey="Granger, N" sort="Granger, N" uniqKey="Granger N" first="N." last="Granger">N. Granger</name><affiliation wicri:level="1"><mods:affiliation>Department of Veterinary Medicine, The Queen's Veterinary School Hospital, University of Cambridge, Cambridge, UK</mods:affiliation><country xml:lang="fr">Royaume-Uni</country><wicri:regionArea>Department of Veterinary Medicine, The Queen's Veterinary School Hospital, University of Cambridge, Cambridge</wicri:regionArea></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">ISTEX</idno><idno type="RBID">ISTEX:0B7A63FA6360ADB56CBDE3564F0E103C0237A7D3</idno><date when="2011" year="2011">2011</date><idno type="doi">10.1111/j.1939-1676.2011.0757.x</idno><idno type="url">https://api.istex.fr/document/0B7A63FA6360ADB56CBDE3564F0E103C0237A7D3/fulltext/pdf</idno><idno type="wicri:Area/Main/Corpus">001A66</idno><idno type="wicri:Area/Main/Curation">001807</idno></publicationStmt><sourceDesc><biblStruct><analytic><title level="a" type="main" xml:lang="en">A Novel Movement Disorder in Related Male Labrador Retrievers Characterized by Extreme Generalized Muscular Stiffness</title><author><name sortKey="Vanhaesebrouck, A E" sort="Vanhaesebrouck, A E" uniqKey="Vanhaesebrouck A" first="A. E." last="Vanhaesebrouck">A. E. Vanhaesebrouck</name><affiliation wicri:level="1"><mods:affiliation>Department of Veterinary Medicine, The Queen's Veterinary School Hospital, University of Cambridge, Cambridge, UK</mods:affiliation><country xml:lang="fr">Royaume-Uni</country><wicri:regionArea>Department of Veterinary Medicine, The Queen's Veterinary School Hospital, University of Cambridge, Cambridge</wicri:regionArea></affiliation></author><author><name sortKey="Shelton, G D" sort="Shelton, G D" uniqKey="Shelton G" first="G. D." last="Shelton">G. D. Shelton</name><affiliation wicri:level="2"><mods:affiliation>Comparative Neuromuscular Laboratory, Department of Pathology, School of Medicine, University of California, San Diego, CA</mods:affiliation><country xml:lang="fr">États-Unis</country><placeName><region type="state">Californie</region></placeName><wicri:cityArea>Comparative Neuromuscular Laboratory, Department of Pathology, School of Medicine, University of California, San Diego</wicri:cityArea></affiliation></author><author><name sortKey="Garosi, L" sort="Garosi, L" uniqKey="Garosi L" first="L." last="Garosi">L. Garosi</name><affiliation wicri:level="1"><mods:affiliation>Davies Veterinary Specialists, Higham Gobion, UK</mods:affiliation><country xml:lang="fr">Royaume-Uni</country><wicri:regionArea>Davies Veterinary Specialists, Higham Gobion</wicri:regionArea></affiliation></author><author><name sortKey="Harcourt Rown, T R" sort="Harcourt Rown, T R" uniqKey="Harcourt Rown T" first="T. R." last="Harcourt-Brown">T. R. Harcourt-Brown</name><affiliation wicri:level="1"><mods:affiliation>Department of Veterinary Medicine, The Queen's Veterinary School Hospital, University of Cambridge, Cambridge, UK</mods:affiliation><country xml:lang="fr">Royaume-Uni</country><wicri:regionArea>Department of Veterinary Medicine, The Queen's Veterinary School Hospital, University of Cambridge, Cambridge</wicri:regionArea></affiliation></author><author><name sortKey="Couturier, J" sort="Couturier, J" uniqKey="Couturier J" first="J." last="Couturier">J. Couturier</name><affiliation wicri:level="1"><mods:affiliation>Davies Veterinary Specialists, Higham Gobion, UK</mods:affiliation><country xml:lang="fr">Royaume-Uni</country><wicri:regionArea>Davies Veterinary Specialists, Higham Gobion</wicri:regionArea></affiliation></author><author><name sortKey="Behr, S" sort="Behr, S" uniqKey="Behr S" first="S." last="Behr">S. Behr</name><affiliation wicri:level="1"><mods:affiliation>Davies Veterinary Specialists, Higham Gobion, UK</mods:affiliation><country xml:lang="fr">Royaume-Uni</country><wicri:regionArea>Davies Veterinary Specialists, Higham Gobion</wicri:regionArea></affiliation><affiliation wicri:level="1"><mods:affiliation>Willows Referral Service, Solihull, UK</mods:affiliation><country xml:lang="fr">Royaume-Uni</country><wicri:regionArea>Willows Referral Service, Solihull</wicri:regionArea></affiliation></author><author><name sortKey="Harvey, R J" sort="Harvey, R J" uniqKey="Harvey R" first="R. J." last="Harvey">R. J. Harvey</name><affiliation wicri:level="1"><mods:affiliation>Department of Pharmacology, The School of Pharmacy, London, UK</mods:affiliation><country xml:lang="fr">Royaume-Uni</country><wicri:regionArea>Department of Pharmacology, The School of Pharmacy, London</wicri:regionArea></affiliation></author><author><name sortKey="Jeffery, N D" sort="Jeffery, N D" uniqKey="Jeffery N" first="N. D." last="Jeffery">N. D. Jeffery</name><affiliation wicri:level="1"><mods:affiliation>Department of Veterinary Medicine, The Queen's Veterinary School Hospital, University of Cambridge, Cambridge, UK</mods:affiliation><country xml:lang="fr">Royaume-Uni</country><wicri:regionArea>Department of Veterinary Medicine, The Queen's Veterinary School Hospital, University of Cambridge, Cambridge</wicri:regionArea></affiliation><affiliation wicri:level="2"><mods:affiliation>Lloyd Veterinary Medical Center, College of Veterinary Medicine, Iowa State University, Ames, OH</mods:affiliation><country xml:lang="fr">États-Unis</country><placeName><region type="state">Ohio</region></placeName><wicri:cityArea>Lloyd Veterinary Medical Center, College of Veterinary Medicine, Iowa State University, Ames</wicri:cityArea></affiliation></author><author><name sortKey="Matiasek, K" sort="Matiasek, K" uniqKey="Matiasek K" first="K." last="Matiasek">K. Matiasek</name><affiliation wicri:level="1"><mods:affiliation>Section of Clinical & Comparative Neuropathology, Institute of Veterinary Pathology, Ludwig Maximilians University of Munich, Munich, Germany</mods:affiliation><country xml:lang="fr">Allemagne</country><wicri:regionArea>Section of Clinical & Comparative Neuropathology, Institute of Veterinary Pathology, Ludwig Maximilians University of Munich, Munich</wicri:regionArea></affiliation></author><author><name sortKey="Blakemore, W F" sort="Blakemore, W F" uniqKey="Blakemore W" first="W. F." last="Blakemore">W. F. Blakemore</name><affiliation wicri:level="1"><mods:affiliation>Department of Veterinary Medicine, The Queen's Veterinary School Hospital, University of Cambridge, Cambridge, UK</mods:affiliation><country xml:lang="fr">Royaume-Uni</country><wicri:regionArea>Department of Veterinary Medicine, The Queen's Veterinary School Hospital, University of Cambridge, Cambridge</wicri:regionArea></affiliation></author><author><name sortKey="Granger, N" sort="Granger, N" uniqKey="Granger N" first="N." last="Granger">N. Granger</name><affiliation wicri:level="1"><mods:affiliation>Department of Veterinary Medicine, The Queen's Veterinary School Hospital, University of Cambridge, Cambridge, UK</mods:affiliation><country xml:lang="fr">Royaume-Uni</country><wicri:regionArea>Department of Veterinary Medicine, The Queen's Veterinary School Hospital, University of Cambridge, Cambridge</wicri:regionArea></affiliation></author></analytic><monogr></monogr><series><title level="j">Journal of Veterinary Internal Medicine</title><idno type="ISSN">0891-6640</idno><idno type="eISSN">1939-1676</idno><imprint><publisher>Blackwell Publishing Inc</publisher><pubPlace>Malden, USA</pubPlace><date type="published" when="2011-09">2011-09</date><biblScope unit="volume">25</biblScope><biblScope unit="issue">5</biblScope><biblScope unit="page" from="1089">1089</biblScope><biblScope unit="page" to="1096">1096</biblScope></imprint><idno type="ISSN">0891-6640</idno></series><idno type="istex">0B7A63FA6360ADB56CBDE3564F0E103C0237A7D3</idno><idno type="DOI">10.1111/j.1939-1676.2011.0757.x</idno><idno type="ArticleID">JVIM757</idno></biblStruct></sourceDesc><seriesStmt><idno type="ISSN">0891-6640</idno></seriesStmt></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Canine</term><term>Central nervous system</term><term>Muscle stiffness</term><term>Rigidity</term></keywords></textClass><langUsage><language ident="en">en</language></langUsage></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">Objectives: To describe the clinical phenotype of a new motor disorder in Labrador Retrievers. Animals and Methods: Case series study. Seven young male Labrador Retrievers presented for evaluation of stiff gait. Results: All affected dogs had generalized muscular stiffness, persistent at rest and resulting in restricted joint movements. They showed a forward flexed posture, festinating gait, and bradykinesia. Signs developed between 2 and 16 months of age and tended to stabilize in adulthood. Needle electromyogram in the conscious state showed continuous motor unit activity in resting epaxial and proximal limb muscles. This activity was abolished by general anesthesia. Muscle and nerve histopathology was normal. In 2 dogs necropsied, astrocytosis was evident throughout the spinal cord gray matter, reticular formation and caudate nuclei. Decreased neuronal counts were selectively found in the spinal cord Rexed's lamina VII, but not in VIII and IX. Pedigree analysis showed that the affected dogs were from 5 related litters. Conclusions and Clinical Importance: This new hypertonicity syndrome in Labrador Retrievers is unique because of the selective distribution of the histological lesions, the lack of progression in adulthood, and its exclusive occurrence in male dogs. Pedigree analysis suggests an X‐linked hereditary disease, although other modes of inheritance cannot be ruled out with certainty. We hypothesize that altered output from basal nuclei and reticular formation together with motor neuron disinhibition caused by a decreased number of spinal cord interneurons leads to the muscular stiffness.</div></front></TEI></record>Pour manipuler ce document sous Unix (Dilib)
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