Fragile X protein functions with Lgl and the PAR complex in flies and mice

Daniela C Zarnescu, 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

Research output: Contribution to journalArticle

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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.

Original languageEnglish (US)
Pages (from-to)43-52
Number of pages10
JournalDevelopmental Cell
Volume8
Issue number1
DOIs
StatePublished - Jan 2005
Externally publishedYes

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Diptera
Intellectual Disability
Fragile X Mental Retardation Protein
Genes
RNA Transport
Fragile X Syndrome
Messenger RNA
Mutation
Cytoskeletal Proteins
Drosophila
Larva
Phosphorylation
Proteins
Genome
Sorting
Tumors
Neoplasms

ASJC Scopus subject areas

  • Developmental Biology

Cite this

Zarnescu, D. C., Jin, P., Betschinger, J., Nakamoto, M., Wang, Y., Dockendorff, T. C., ... Moses, K. (2005). Fragile X protein functions with Lgl and the PAR complex in flies and mice. Developmental Cell, 8(1), 43-52. https://doi.org/10.1016/j.devcel.2004.10.020

Fragile X protein functions with Lgl and the PAR complex in flies and mice. / Zarnescu, Daniela C; Jin, Peng; Betschinger, Joerg; Nakamoto, Mika; Wang, Yan; Dockendorff, Thomas C.; Feng, Yue; Jongens, Thomas A.; Sisson, John C.; Knoblich, Juergen A.; Warren, Stephen T.; Moses, Kevin.

In: Developmental Cell, Vol. 8, No. 1, 01.2005, p. 43-52.

Research output: Contribution to journalArticle

Zarnescu, DC, Jin, P, Betschinger, J, Nakamoto, M, Wang, Y, Dockendorff, TC, Feng, Y, Jongens, TA, Sisson, JC, Knoblich, JA, Warren, ST & Moses, K 2005, 'Fragile X protein functions with Lgl and the PAR complex in flies and mice', Developmental Cell, vol. 8, no. 1, pp. 43-52. https://doi.org/10.1016/j.devcel.2004.10.020
Zarnescu, Daniela C ; Jin, Peng ; Betschinger, Joerg ; Nakamoto, Mika ; Wang, Yan ; Dockendorff, Thomas C. ; Feng, Yue ; Jongens, Thomas A. ; Sisson, John C. ; Knoblich, Juergen A. ; Warren, Stephen T. ; Moses, Kevin. / Fragile X protein functions with Lgl and the PAR complex in flies and mice. In: Developmental Cell. 2005 ; Vol. 8, No. 1. pp. 43-52.
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