Dominant Drop mutants are gain-of-function alleles of the muscle segment homeobox gene (msh) whose overexpression leads to the arrest of eye development

Developmental Biology
B A Mozer

Abstract

Dominant Drop (Dr) mutations are nearly eyeless and have additional recessive phenotypes including lethality and patterning defects in eye and sensory bristles due to cis-regulatory lesions in the cell cycle regulator string (stg). Genetic analysis demonstrates that the dominant small eye phenotype is the result of separate gain-of-function mutations in the closely linked muscle segment homeobox (msh) gene, encoding a homeodomain transcription factor required for patterning of muscle and nervous system. Reversion of the Dr(Mio) allele was coincident with the generation of lethal loss-of-function mutations in msh in cis, suggesting that the dominant eye phenotype is the result of ectopic expression. Molecular genetic analysis revealed that two dominant Dr alleles contain lesions upstream of the msh transcription start site. In the Dr(Mio) mutant, a 3S18 retrotransposon insertion is the target of second-site mutations (P-element insertions or deletions) which suppress the dominant eye phenotype following reversion. The pattern of 3S18 expression and the absence of msh in eye imaginal discs suggest that transcriptional activation of the msh promoter accounts for ectopic expression. Dr dominant mutations arrest eye development by b...Continue Reading

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Citations

Jul 24, 2013·The Journal of Cell Biology·Todd SchoborgMariano Labrador
Mar 29, 2014·Ophthalmic Genetics·Julie PlaisanciéHélène Dollfus
Oct 2, 2009·Developmental Dynamics : an Official Publication of the American Association of Anatomists·Tonia Von OhlenWill Poulson
Oct 23, 2004·BioEssays : News and Reviews in Molecular, Cellular and Developmental Biology·Gregg Roman
Sep 20, 2005·Trends in Genetics : TIG·Casto Ramos, Benoît Robert

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