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L. Liang, J. Toonen, L. Jackson, J. Besharse, D. J. Sidjanin; Mapping of the Wavy With Open Eye Lids 2 (woe2) Locus in Mice. Invest. Ophthalmol. Vis. Sci. 2007;48(13):5999.
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Waved with open eyes 2 (woe2) is an autosomal recessive mouse mutant that arose spontaneously on 129 X C57BL/6 background. The purpose of this study is to identify the mutation responsible for the woe2 phenotype.
The woe2 mice were evaluated with a slit lamp biomicroscope and an indirect ophthalmoscope following mydriasis with 1% atropine. For linkage analysis woe2 mice were outcrossed to wild type C3A.BLiA-Pde6b+/J. The F1 mice were backcrossed to woe2 and 80 F2 progeny were generated. At weaning the progeny were evaluated for the coat and eye phenotype and were euthanized. Genomic DNA was isolated from collected spleens. Microsatellite markers were selected to evenly cover the mouse genome. Genotypes were determined by PCR amplification of genomic DNA and alleles were scored.
Phenotypically, the woe2 mice show wavy coat, eyelids open at birth, microphthalmia/anophthalmia, corneal opacity, corneal neovascularization and white retinal spots. The woe2 locus was mapped to 3.4 cM region on mouse chromosome 7. The linkage map established the woe2 critical region between D7Mit340 and D7Mit224. Evaluation of the mouse genome map (http://genome.ucsc.edu/cgi-bin/hgGateway) identified Protein Phosphatase 1, Regulatory (inhibitor) Subunit 13 Like (Ppp1r13l) as a candidate gene.
The woe2 mouse is a mutation in a gene that plays an essential role in the normal eye development. Our mapping results established linkage of the woe2 locus to centromeric end of mouse chromosome 7. Evaluation of the genes in the critical region identified Ppp1r13l as a candidate gene. Sequencing of the Ppp1r13l gene in the woe2 mouse is currently under way
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