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Publication : The neurological mutant flailer is closely linked to dilute on mouse Chromosome 9

First Author  Jones JM Year  1996
Journal  10th International Mouse Genome Conference 10/7/96 - 10/10/96, Paris, France Pages  2 (Abstr.)
Mgi Jnum  J:42513 Mgi Id  MGI:1095871
Citation  Jones JM, et al. (1996) The neurological mutant flailer is closely linked to dilute on mouse Chromosome 9. 10th International Mouse Genome Conference 10/7/96 - 10/10/96, Paris, France :2 (Abstr.)
abstractText  Full text of Abstract: 2. The neurological mutant flailer is closely linked to dilute on mouse chromosome 9 Julie M. Jones1, Nancy A. Jenkins2, Neal G. Copeland2, Bruce A. Hamilton3, Miriam H. Meisler1. 1Dept. of Human Genetics, University of Michigan, Ann Arbor, MI 48109-0614, U.S.A. 2ABL-Basic Research Program, NCI-Frederick Cancer Research and Development Center Frederick, MD 21702, U.S.A. 3Whitehead Institute, MIT, Cambridge, MA 02139, U.S.A. Flailer is an autosomal recessive neurological mutant with a variable phenotype. Approximately 50% of the homozygotes are severely ataxic and survive for only 3 weeks; they fall frequently and have difficulty in righting themselves, leading to the characteristic rapid flailing of their legs. The other 50% of homozygotes have a transient ataxia at 3 weeks of age. These animals recover and are fertile with a normal lifespan. We previously mapped flailer to the 19 cM interval between D9Mit4 and D9Mit50. We now report the analysis of 218 affected F2 animals from a (flail/flail x CAST/Ei) F1 intercross. Typing of the F2 animals for microsatellite markers in the above interval localized flailer to the 0.5 cM interval between D9Mit123 and D9Mit261. F2 animals with crossovers in the flailer region were typed by RFLP for Myo5a, the gene that is mutated in the dilute mouse. No recombinants between flailer and Myo5a were observed. In addition to diluted pigmentation, several alleles of dilute result in neurological symptoms that resemble the flailer phenotype. To test for allelism between dilute and flailer, we have crossed dilute compound heterozygotes of genotype dv/d120J with flailer homozygotes. The results of the allelism test and evaluation of Myo5a as a candidate gene for the flailer mutant will be discussed.
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