Testing of SHIRPA, a mouse phenotypic assessment protocol, on Dmd
mdx
and Dmd
mdx3cv
dystrophin-deficient mice |
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Authors: | Jill A Rafael Yumiko Nitta Jo Peters Kay E Davies |
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Institution: | (1) Department of Human Anatomy and Genetics, University of Oxford, South Parks Road, Oxford OX1 3QX, UK, GB;(2) Mammalian Genetics Unit, Medical Research Council Harwell, Didcot, Oxfordshire OX11 ORD, UK, GB |
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Abstract: | The SHIRPA protocol was proposed as a rapid, comprehensive screening method for qualitatively abnormal phenotypes in the
mouse (Rogers et al., Mamm Genome 8, 711, 1997). This screening technique is currently being used to identify mutants induced
by N-ethylnitrosourea (ENU) mutagenesis (Brown and Nolan, Hum Mol Genet 7, 1627, 1998). SHIRPA can be used to identify mutants
with neuromuscular abnormalities, but the sensitivity of the protocol is unknown. We tested two dystrophin-deficient mutants
Dmd
mdx
and Dmd
mdx3cv
, both of which are indistinguishable from wild-type by a simple visual assessment, at different ages, using the primary screen
of the SHIRPA protocol. The most dramatic observation was that both Dmd
mdx
and Dmd
mdx3cv
mice showed extreme fatigue after testing, while mice from the same C57BL strains appeared unaffected. Each strain of dystrophin-deficient
mice showed a different profile in locomotor activity and deficiencies in the wire maneuver, righting reflex, and negative
geotaxis tests. Furthermore, the wire maneuver test indicated an earlier onset of muscular impairment in Dmd
mdx
than Dmd
mdx3cv
mice. These data suggest that the SHIRPA primary screen is effective not only in identifying subtle neuromuscular mutants,
but also in distinguishing qualitative differences between mutants with neuromuscular abnormalities.
Received: 5 August 1999 / Accepted: 14 April 2000 |
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Keywords: | |
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