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Pax2 expression and retinal morphogenesis in the normal and Krd mouse.
- Source :
-
Developmental biology [Dev Biol] 1998 Jan 15; Vol. 193 (2), pp. 209-24. - Publication Year :
- 1998
-
Abstract
- The Kidney and retinal defects (Krd) mouse carries a 7-cM transgene-induced deletion on chromosome 19 that includes the Pax2 locus. Adult mice heterozygous for the Krd deletion (Krd/+) are haploid for Pax2 and have a variable, semidominant phenotype characterized by structural defects of the kidney, retina, and optic disc. Renal and ocular anomalies present in heterozygous Pax2 mutants in both mice and humans support the hypothesis that haploinsufficiency of Pax2 underlies the Krd phenotype. To understand the embryonic basis of ocular defects observed in adult Krd/+ mice, we used immunohistochemistry, digital three-dimensional reconstructions, and quantitative morphometry to examine Pax2 protein distribution and ocular development in normal and Krd/+ mice from E10.5 to P2. In +/+ embryos, Pax2 immunopositive (Pax2+) cells demarcate the embryonic fissure as it forms in the ventral optic cup and optic stalk. After closure of the embryonic fissure, Pax2 immunostaining disappears from the ventral retina, but persists in a cuff of cells encircling the developing optic disc, the site where ganglion cell axons exit the retina. In Krd/+ embryos, Pax2+ cells in the posterior optic cup and the optic stalk undergo abnormal morphogenetic movements and the embryonic fissure fails to form normally. This results in an abnormal organization of the Pax2+ cells and ganglion cell axons at the nascent optic disc. The abnormal morphogenetic movements of the Pax2+ cells in the embryonic retina and optic stalk and the initial misrouting of the ganglion cell axons give rise to retinal and optic disc defects observed in the adult Krd/+ mice.<br /> (Copyright 1998 Academic Press.)
- Subjects :
- Animals
DNA-Binding Proteins analysis
DNA-Binding Proteins genetics
Immunohistochemistry
Kidney abnormalities
Kidney embryology
Mice
Mice, Inbred C57BL
Mice, Transgenic
Optic Disk abnormalities
Optic Disk chemistry
Optic Disk embryology
Optic Nerve abnormalities
Optic Nerve chemistry
Optic Nerve embryology
PAX2 Transcription Factor
Retina abnormalities
Staining and Labeling
Transcription Factors analysis
Transcription Factors genetics
DNA-Binding Proteins biosynthesis
Morphogenesis genetics
Retina embryology
Transcription Factors biosynthesis
Subjects
Details
- Language :
- English
- ISSN :
- 0012-1606
- Volume :
- 193
- Issue :
- 2
- Database :
- MEDLINE
- Journal :
- Developmental biology
- Publication Type :
- Academic Journal
- Accession number :
- 9473325
- Full Text :
- https://doi.org/10.1006/dbio.1997.8794