The circling behavior of the deafblind LEW-ci2 rat is linked to a segment of RNO10 containing Myo15 and Kcnj12

The LEW/Ztm-ci2 rat is an autosomal recessive mutant that displays circling behavior, deafness, progressive retinopathy, locomotor hyperactivity, ataxia, and opisthotonus. We performed a genome-wide scan of a (LEW/Ztm-ci2 x BN/Ztm) F1 x LEW/Ztm-ci2 backcross population with anonymous microsatellite...

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Bibliographic Details
Published in:Mammalian genome 2003-09, Vol.14 (9), p.620-627
Main Authors: Chwalisz, Wojciech T, Koelsch, Bernd U, Kindler-Röhrborn, Andrea, Hedrich, Hans J, Wedekind, Dirk
Format: Article
Language:English
Subjects:
Animals
Base Sequence
Behavior, Animal
Blindness - genetics
Chromosome Mapping
Deafness - genetics
DNA, Complementary - genetics
Genetic Markers
Humans
Mice
Myosins - genetics
Phenotype
Potassium Channels - genetics
Rats
Rats, Inbred BN
Rats, Inbred Lew
Rats, Mutant Strains
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Summary:The LEW/Ztm-ci2 rat is an autosomal recessive mutant that displays circling behavior, deafness, progressive retinopathy, locomotor hyperactivity, ataxia, and opisthotonus. We performed a genome-wide scan of a (LEW/Ztm-ci2 x BN/Ztm) F1 x LEW/Ztm-ci2 backcross population with anonymous microsatellite markers to analyze the genetics of this mutant rat. This linkage analysis demonstrated a very strong association of RNO10 SSLP markers to the phenotype with a core region in the central part of the chromosome. The knowledge of genes mapping to this part of the rat genome and their linkage to SSLP markers is still poor. We developed SSLP markers closely linked to genes, which might be responsible for the mutant phenotype by using the growing amount of rat-specific DNA sequences available at World Wide Web databases. Application of this method facilitated the search for candidate genes for the phenotype of the LEW-ci2 rat. We were able to map Myo15 and its neighboring genes, Znf179 and Aldh3a1, to the region of interest and Myo1c to a more distal location on RNO10. Further rat BAC clones were used to create a physical map of the region of interest. This map revealed the position of further genes. Among those is Kcnj12. Owing to their localization on RNO10 and their involvement in a similar pathology in human and mouse, Myo15 and Kcnj12 can be regarded as candidate genes for the deafblind phenotype of the LEW-ci2 rat.
ISSN:0938-8990
1432-1777
DOI:10.1007/s00335-003-3009-x