Ocular retardation mouse caused by Chx10 homeobox null allele: Impaired retinal progenitor proliferation and bipolar cell differentiation

Margit Burmeister, Jakub Novak, Mei Ying Liang, Sharmila Basu, Lynda Ploder, Norman L. Hawes, Danka Vidgen, Frank Hoover, Daniel Goldman, Vitauts I. Kalnins, Thomas H. Roderick, Benjamin A. Taylor, Mark Hankin, Roderick R. McInnes

Research output: Contribution to journalArticle

406 Citations (Scopus)

Abstract

Ocular retardation (or) is a murine eye mutation causing microphthalmia, a thin hypocellular retina and optic nerve aplasia. Here we show that mice carrying the or(J) allele have a premature stop codon in the homeobox of the Chx10 gene, a gene expressed at high levels in uncommitted retinal progenitor cells and mature bipolar cells. No CHX10 protein was detectable in the retinal neuroepithelium of or(J) homozygotes. The loss of CHX10 leads both to reduced proliferation of retinal progenitors and to a specific absence of differentiated bipolar cells. Other major retinal cell types were present and correctly positioned in the mutant retina, although rod outer segments were short and retinal lamination was incomplete. These results indicate that Chx10 is an essential component in the network of genes required for the development of the mammalian eye, with profound effects on retinal progenitor proliferation and bipolar cell specification or differentiation.

Original languageEnglish (US)
Pages (from-to)376-384
Number of pages9
JournalNature Genetics
Volume12
Issue number4
DOIs
StatePublished - Apr 1 1996
Externally publishedYes

Fingerprint

Homeobox Genes
Cell Differentiation
Alleles
Retina
Microphthalmos
Rod Cell Outer Segment
Nonsense Codon
Gene Regulatory Networks
Homozygote
Optic Nerve
Stem Cells
Cell Proliferation
Mutation
Genes
Proteins

ASJC Scopus subject areas

  • Genetics

Cite this

Burmeister, M., Novak, J., Liang, M. Y., Basu, S., Ploder, L., Hawes, N. L., ... McInnes, R. R. (1996). Ocular retardation mouse caused by Chx10 homeobox null allele: Impaired retinal progenitor proliferation and bipolar cell differentiation. Nature Genetics, 12(4), 376-384. https://doi.org/10.1038/ng0496-376

Ocular retardation mouse caused by Chx10 homeobox null allele : Impaired retinal progenitor proliferation and bipolar cell differentiation. / Burmeister, Margit; Novak, Jakub; Liang, Mei Ying; Basu, Sharmila; Ploder, Lynda; Hawes, Norman L.; Vidgen, Danka; Hoover, Frank; Goldman, Daniel; Kalnins, Vitauts I.; Roderick, Thomas H.; Taylor, Benjamin A.; Hankin, Mark; McInnes, Roderick R.

In: Nature Genetics, Vol. 12, No. 4, 01.04.1996, p. 376-384.

Research output: Contribution to journalArticle

Burmeister, M, Novak, J, Liang, MY, Basu, S, Ploder, L, Hawes, NL, Vidgen, D, Hoover, F, Goldman, D, Kalnins, VI, Roderick, TH, Taylor, BA, Hankin, M & McInnes, RR 1996, 'Ocular retardation mouse caused by Chx10 homeobox null allele: Impaired retinal progenitor proliferation and bipolar cell differentiation', Nature Genetics, vol. 12, no. 4, pp. 376-384. https://doi.org/10.1038/ng0496-376
Burmeister, Margit ; Novak, Jakub ; Liang, Mei Ying ; Basu, Sharmila ; Ploder, Lynda ; Hawes, Norman L. ; Vidgen, Danka ; Hoover, Frank ; Goldman, Daniel ; Kalnins, Vitauts I. ; Roderick, Thomas H. ; Taylor, Benjamin A. ; Hankin, Mark ; McInnes, Roderick R. / Ocular retardation mouse caused by Chx10 homeobox null allele : Impaired retinal progenitor proliferation and bipolar cell differentiation. In: Nature Genetics. 1996 ; Vol. 12, No. 4. pp. 376-384.
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