Fragile X protein functions with lgl and the par complex in flies and mice - PubMed (original) (raw)
Comparative Study
doi: 10.1016/j.devcel.2004.10.020.
Peng Jin, Joerg Betschinger, Mika Nakamoto, Yan Wang, Thomas C Dockendorff, Yue Feng, Thomas A Jongens, John C Sisson, Juergen A Knoblich, Stephen T Warren, Kevin Moses
Affiliations
- PMID: 15621528
- DOI: 10.1016/j.devcel.2004.10.020
Free article
Comparative Study
Fragile X protein functions with lgl and the par complex in flies and mice
Daniela C Zarnescu et al. Dev Cell. 2005 Jan.
Free article
Abstract
Fragile X syndrome, the most common form of inherited mental retardation, is caused by loss of function for the Fragile X Mental Retardation 1 gene (FMR1). FMR1 protein (FMRP) has specific mRNA targets and is thought to be involved in their transport to subsynaptic sites as well as translation regulation. We report a saturating genetic screen of the Drosophila autosomal genome to identify functional partners of dFmr1. We recovered 19 mutations in the tumor suppressor lethal (2) giant larvae (dlgl) gene and 90 mutations at other loci. dlgl encodes a cytoskeletal protein involved in cellular polarity and cytoplasmic transport and is regulated by the PAR complex through phosphorylation. We provide direct evidence for a Fmrp/Lgl/mRNA complex, which functions in neural development in flies and is developmentally regulated in mice. Our data suggest that Lgl may regulate Fmrp/mRNA sorting, transport, and anchoring via the PAR complex.
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