Laminin α2 (merosin)-deficient muscular dystrophy and demyelinating neuropathy in two cats
We report laminin α2 (merosin) deficiency associated with muscular dystrophy and demyelinating neuropathy in two cats. The cats developed progressive muscle weakness, and atrophy. Either hypotonia or contractures resulted in recumbency, necessitating euthanasia. Muscle biopsies showed dystrophic cha...
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| Published in | Journal of the neurological sciences Vol. 189; no. 1; pp. 37 - 43 |
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| Main Authors | , , , , , , |
| Format | Journal Article |
| Language | English |
| Published |
Elsevier B.V
15.08.2001
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| Subjects | |
| Online Access | Get full text |
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| Abstract | We report laminin α2 (merosin) deficiency associated with muscular dystrophy and demyelinating neuropathy in two cats. The cats developed progressive muscle weakness, and atrophy. Either hypotonia or contractures resulted in recumbency, necessitating euthanasia. Muscle biopsies showed dystrophic changes including marked endomysial fibrosis, myofiber necrosis, variability of fiber size, and perimysial lipid accumulation. Immunohistochemistry showed that laminin α2 chain was absent or reduced, while dystrophin and all the components of the dystrophin-associated glycoprotein complex were present and normal. One cat was examined in detail. Motor nerve conduction velocity (MNCV) was decreased, and ultrastructurally the peripheral nerves showed Schwann cell degeneration and demyelination. Brain imaging was not performed, but white matter changes were not apparent in the brain at necropsy. The disease in these cats is similar to primary or secondary merosin (laminin α2)-deficient congenital muscular dystrophy (CMD) in humans and to
dystrophia muscularis in mice. |
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| AbstractList | We report laminin α2 (merosin) deficiency associated with muscular dystrophy and demyelinating neuropathy in two cats. The cats developed progressive muscle weakness, and atrophy. Either hypotonia or contractures resulted in recumbency, necessitating euthanasia. Muscle biopsies showed dystrophic changes including marked endomysial fibrosis, myofiber necrosis, variability of fiber size, and perimysial lipid accumulation. Immunohistochemistry showed that laminin α2 chain was absent or reduced, while dystrophin and all the components of the dystrophin-associated glycoprotein complex were present and normal. One cat was examined in detail. Motor nerve conduction velocity (MNCV) was decreased, and ultrastructurally the peripheral nerves showed Schwann cell degeneration and demyelination. Brain imaging was not performed, but white matter changes were not apparent in the brain at necropsy. The disease in these cats is similar to primary or secondary merosin (laminin α2)-deficient congenital muscular dystrophy (CMD) in humans and to
dystrophia muscularis in mice. |
| Author | O'Brien, Dennis P Liu, Ling A Engvall, Eva Shelton, G.Diane Powell, Henry C Johnson, Gayle C Guo, Ling T |
| Author_xml | – sequence: 1 givenname: Dennis P surname: O'Brien fullname: O'Brien, Dennis P email: OBrienD@missouri.edu organization: Department of Veterinary Medicine and Surgery, University of Missouri, 379 E. Campus Drive, Columbia, MO 65211, USA – sequence: 2 givenname: Gayle C surname: Johnson fullname: Johnson, Gayle C organization: Veterinary Diagnostic Laboratory University of Missouri, Columbia, MO, USA – sequence: 3 givenname: Ling A surname: Liu fullname: Liu, Ling A organization: The Burnham Institute, La Jolla, CA, USA – sequence: 4 givenname: Ling T surname: Guo fullname: Guo, Ling T organization: The Burnham Institute, La Jolla, CA, USA – sequence: 5 givenname: Eva surname: Engvall fullname: Engvall, Eva organization: The Burnham Institute, La Jolla, CA, USA – sequence: 6 givenname: Henry C surname: Powell fullname: Powell, Henry C organization: University of California, San Diego, La Jolla, CA, USA – sequence: 7 givenname: G.Diane surname: Shelton fullname: Shelton, G.Diane organization: University of California, San Diego, La Jolla, CA, USA |
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| Keywords | Merosin Animal model Laminin Congenital muscular dystrophy Demyelination Feline Peripheral neuropathy |
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| Snippet | We report laminin α2 (merosin) deficiency associated with muscular dystrophy and demyelinating neuropathy in two cats. The cats developed progressive muscle... |
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| SubjectTerms | Animal model Congenital muscular dystrophy Demyelination Feline Laminin Merosin Peripheral neuropathy |
| Title | Laminin α2 (merosin)-deficient muscular dystrophy and demyelinating neuropathy in two cats |
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