@article{be6e5c3e047c4fc4a8f8149cb0e5ee89,
title = "Fragile X protein functions with Lgl and the PAR complex in flies and mice",
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.",
author = "Zarnescu, {Daniela C.} and Peng Jin and Joerg Betschinger and Mika Nakamoto and Yan Wang and Dockendorff, {Thomas C.} and Yue Feng and Jongens, {Thomas A.} and Sisson, {John C.} and Knoblich, {Juergen A.} and Warren, {Stephen T.} and Kevin Moses",
note = "Funding Information: We thank the Bloomington Drosophila stock center; V. Budnik, C. Doe, J. Fawcet, F. Matsuzaki, T. Pawson, K. Zinsmaier, A. Wodarz, and especially B. Mechler for reagents; R. Ordway, S. Ceman, and especially V. Faundez for technical advice; and S. Cook, R. Airinei, D. Saiman, M. Dawson, W. Pierre, and the Emory IM&MF for technical assistance. We thank K. Moberg and members of the Moses lab for comments on the manuscript. This paper is dedicated to the memory of M. Diaconu. D.C.Z. and T.C.D. were supported by fellowships from FRAXA. T.C.D. and T.A.J. were supported by NIH HD33834. P.J. is supported by Rett Syndrome Research Foundation, Y.F. by NIH 5 PO1 HD355765, and S.T.W. by NIH R37HD20521, P01HD35576, and P30HD024064. J.C.S. supported by a March of Dimes Basil O'Connor Award and RO1 GM067013 from the NIH. J.A.K.'s lab supported by Boehringer Ingelheim, the Austrian Academy of Sciences, and the Wiener Wirtschaftsfoerderungsfonds. D.C.Z. is supported by NIH F32 NS046880. This work was chiefly supported by NIH R21 NS43536. ",
year = "2005",
month = jan,
doi = "10.1016/j.devcel.2004.10.020",
language = "English (US)",
volume = "8",
pages = "43--52",
journal = "Developmental Cell",
issn = "1534-5807",
publisher = "Cell Press",
number = "1",
}