A motor function for the DEAD-box RNA helicase, Gemin3, in Drosophila
The survival motor neuron (SMN) protein, the determining factor for spinal muscular atrophy (SMA), is complexed with a group of proteins in human cells. Gemin3 is the only RNA helicase in the SMN complex. Here, we report the identification of Drosophila melanogaster Gemin3 and investigate its functi...
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| Veröffentlicht in: | PLoS genetics 2008-11, Vol.4 (11), p.e1000265-e1000265 |
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| creator | Cauchi, Ruben J Davies, Kay E Liu, Ji-Long |
| description | The survival motor neuron (SMN) protein, the determining factor for spinal muscular atrophy (SMA), is complexed with a group of proteins in human cells. Gemin3 is the only RNA helicase in the SMN complex. Here, we report the identification of Drosophila melanogaster Gemin3 and investigate its function in vivo. Like in vertebrates, Gemin3 physically interacts with SMN in Drosophila. Loss of function of gemin3 results in lethality at larval and/or prepupal stages. Before they die, gemin3 mutant larvae exhibit declined mobility and expanded neuromuscular junctions. Expression of a dominant-negative transgene and knockdown of Gemin3 in mesoderm cause lethality. A less severe Gemin3 disruption in developing muscles leads to flightless adults and flight muscle degeneration. Our findings suggest that Drosophila Gemin3 is required for larval development and motor function. |
| doi_str_mv | 10.1371/journal.pgen.1000265 |
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Gemin3 is the only RNA helicase in the SMN complex. Here, we report the identification of Drosophila melanogaster Gemin3 and investigate its function in vivo. Like in vertebrates, Gemin3 physically interacts with SMN in Drosophila. Loss of function of gemin3 results in lethality at larval and/or prepupal stages. Before they die, gemin3 mutant larvae exhibit declined mobility and expanded neuromuscular junctions. Expression of a dominant-negative transgene and knockdown of Gemin3 in mesoderm cause lethality. A less severe Gemin3 disruption in developing muscles leads to flightless adults and flight muscle degeneration. 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This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited: Cauchi RJ, Davies KE, Liu J-L (2008) A Motor Function for the DEAD-Box RNA Helicase, Gemin3, in Drosophila. 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Gemin3 is the only RNA helicase in the SMN complex. Here, we report the identification of Drosophila melanogaster Gemin3 and investigate its function in vivo. Like in vertebrates, Gemin3 physically interacts with SMN in Drosophila. Loss of function of gemin3 results in lethality at larval and/or prepupal stages. Before they die, gemin3 mutant larvae exhibit declined mobility and expanded neuromuscular junctions. Expression of a dominant-negative transgene and knockdown of Gemin3 in mesoderm cause lethality. A less severe Gemin3 disruption in developing muscles leads to flightless adults and flight muscle degeneration. Our findings suggest that Drosophila Gemin3 is required for larval development and motor function.</description><subject>Animals</subject><subject>Cell Biology</subject><subject>Cell Biology/Nuclear Structure and Function</subject><subject>Chromosomal proteins</subject><subject>DEAD Box Protein 20 - genetics</subject><subject>DEAD Box Protein 20 - physiology</subject><subject>DEAD-box RNA Helicases - genetics</subject><subject>DEAD-box RNA Helicases - physiology</subject><subject>Developmental Biology</subject><subject>Drosophila</subject><subject>Drosophila melanogaster</subject><subject>Drosophila melanogaster - embryology</subject><subject>Drosophila melanogaster - enzymology</subject><subject>Drosophila melanogaster - genetics</subject><subject>Drosophila Proteins</subject><subject>Genes</subject><subject>Genetic aspects</subject><subject>Genetics</subject><subject>Genetics and Genomics</subject><subject>Helicases</subject><subject>Humans</subject><subject>Larva - metabolism</subject><subject>Mice</subject><subject>Motor Neurons - enzymology</subject><subject>Mutation</subject><subject>Neuroscience/Neuronal and Glial Cell Biology</subject><subject>Physiological aspects</subject><subject>Physiology/Motor Systems</subject><subject>Physiology/Muscle and Connective Tissue</subject><subject>Proteins</subject><subject>Risk factors</subject><subject>Spinal muscular atrophy</subject><subject>Studies</subject><subject>Survival of Motor Neuron 1 Protein - metabolism</subject><subject>Transgenes</subject><issn>1553-7404</issn><issn>1553-7390</issn><issn>1553-7404</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2008</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><sourceid>DOA</sourceid><recordid>eNqVk22L1DAQx4so3nn6DUQLwoFwuyaTpEnfCOV2PReOOzgf3oY0TbdZss3atHJ-e7Nu1S0IavIiyeQ3_yGZmSR5jtEcE47fbPzQtcrNd2vTzjFCCDL2IDnFjJEZp4g-PNqfJE9C2CBEmMj54-QE5wgIRew0WRbp1ve-S-uh1b31bVrHQ9-YdLEsFrPS36d3N0XaGGe1CuYivTJb25KL1LbpovPB7xrr1NPkUa1cMM_G9Sz59G758fL97Pr2anVZXM80B97PhIIsg5IQEBqXUENWloKWpQGS5TQORXOoKiQUB8wUx1pwrfKKM0Qqpktylrw86O6cD3L8gSAxwYRRAIEjsToQlVcbuevsVnXfpFdW_jD4bi1V11vtjDQaU1aB1goDNRrlOtdYCJVVXFFe6qj1dow2lFtTadP2nXIT0elNaxu59l8lMM5zYFHgfBTo_JfBhF5ubdDGOdUaPwSZ5QJgn6S_gYAIEpSgCL46gGsVX2Db2sfAeg_LAhBQIWjOIzX_AxVnFZOnfWtqG-0Th9cTh8j05r5fqyEEufpw9x_szb-zt5-n7PkR2xjl-iZ4N-yLMkxBegB1rL_QmfpXRjCS-9b4WRhy3xpybI3o9uI4m7-dxl4g3wEpTQWC</recordid><startdate>20081101</startdate><enddate>20081101</enddate><creator>Cauchi, Ruben J</creator><creator>Davies, Kay E</creator><creator>Liu, Ji-Long</creator><general>Public Library of Science</general><general>Public Library of Science (PLoS)</general><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>IOV</scope><scope>ISN</scope><scope>ISR</scope><scope>7SS</scope><scope>7TK</scope><scope>7TM</scope><scope>8FD</scope><scope>FR3</scope><scope>P64</scope><scope>RC3</scope><scope>7X8</scope><scope>5PM</scope><scope>DOA</scope></search><sort><creationdate>20081101</creationdate><title>A motor function for the DEAD-box RNA helicase, Gemin3, in Drosophila</title><author>Cauchi, Ruben J ; Davies, Kay E ; Liu, Ji-Long</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c727t-8a2662b3328c1b2f26bb84bbe23694444a492dd08a7215a71c87ca9d7503d5cb3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2008</creationdate><topic>Animals</topic><topic>Cell Biology</topic><topic>Cell Biology/Nuclear Structure and Function</topic><topic>Chromosomal proteins</topic><topic>DEAD Box Protein 20 - genetics</topic><topic>DEAD Box Protein 20 - physiology</topic><topic>DEAD-box RNA Helicases - genetics</topic><topic>DEAD-box RNA Helicases - physiology</topic><topic>Developmental Biology</topic><topic>Drosophila</topic><topic>Drosophila melanogaster</topic><topic>Drosophila melanogaster - embryology</topic><topic>Drosophila melanogaster - enzymology</topic><topic>Drosophila melanogaster - genetics</topic><topic>Drosophila Proteins</topic><topic>Genes</topic><topic>Genetic aspects</topic><topic>Genetics</topic><topic>Genetics and Genomics</topic><topic>Helicases</topic><topic>Humans</topic><topic>Larva - metabolism</topic><topic>Mice</topic><topic>Motor Neurons - enzymology</topic><topic>Mutation</topic><topic>Neuroscience/Neuronal and Glial Cell Biology</topic><topic>Physiological aspects</topic><topic>Physiology/Motor Systems</topic><topic>Physiology/Muscle and Connective Tissue</topic><topic>Proteins</topic><topic>Risk factors</topic><topic>Spinal muscular atrophy</topic><topic>Studies</topic><topic>Survival of Motor Neuron 1 Protein - metabolism</topic><topic>Transgenes</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Cauchi, Ruben J</creatorcontrib><creatorcontrib>Davies, Kay E</creatorcontrib><creatorcontrib>Liu, Ji-Long</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>Gale In Context: Opposing Viewpoints</collection><collection>Gale In Context: Canada</collection><collection>Gale In Context: Science</collection><collection>Entomology Abstracts (Full archive)</collection><collection>Neurosciences Abstracts</collection><collection>Nucleic Acids Abstracts</collection><collection>Technology Research Database</collection><collection>Engineering Research Database</collection><collection>Biotechnology and BioEngineering Abstracts</collection><collection>Genetics Abstracts</collection><collection>MEDLINE - Academic</collection><collection>PubMed Central (Full Participant titles)</collection><collection>DOAJ Directory of Open Access Journals</collection><jtitle>PLoS genetics</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Cauchi, Ruben J</au><au>Davies, Kay E</au><au>Liu, Ji-Long</au><au>Cox, Gregory A.</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>A motor function for the DEAD-box RNA helicase, Gemin3, in Drosophila</atitle><jtitle>PLoS genetics</jtitle><addtitle>PLoS Genet</addtitle><date>2008-11-01</date><risdate>2008</risdate><volume>4</volume><issue>11</issue><spage>e1000265</spage><epage>e1000265</epage><pages>e1000265-e1000265</pages><issn>1553-7404</issn><issn>1553-7390</issn><eissn>1553-7404</eissn><abstract>The survival motor neuron (SMN) protein, the determining factor for spinal muscular atrophy (SMA), is complexed with a group of proteins in human cells. Gemin3 is the only RNA helicase in the SMN complex. Here, we report the identification of Drosophila melanogaster Gemin3 and investigate its function in vivo. Like in vertebrates, Gemin3 physically interacts with SMN in Drosophila. Loss of function of gemin3 results in lethality at larval and/or prepupal stages. Before they die, gemin3 mutant larvae exhibit declined mobility and expanded neuromuscular junctions. Expression of a dominant-negative transgene and knockdown of Gemin3 in mesoderm cause lethality. A less severe Gemin3 disruption in developing muscles leads to flightless adults and flight muscle degeneration. Our findings suggest that Drosophila Gemin3 is required for larval development and motor function.</abstract><cop>United States</cop><pub>Public Library of Science</pub><pmid>19023405</pmid><doi>10.1371/journal.pgen.1000265</doi><oa>free_for_read</oa></addata></record> |
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| subjects | Animals Cell Biology Cell Biology/Nuclear Structure and Function Chromosomal proteins DEAD Box Protein 20 - genetics DEAD Box Protein 20 - physiology DEAD-box RNA Helicases - genetics DEAD-box RNA Helicases - physiology Developmental Biology Drosophila Drosophila melanogaster Drosophila melanogaster - embryology Drosophila melanogaster - enzymology Drosophila melanogaster - genetics Drosophila Proteins Genes Genetic aspects Genetics Genetics and Genomics Helicases Humans Larva - metabolism Mice Motor Neurons - enzymology Mutation Neuroscience/Neuronal and Glial Cell Biology Physiological aspects Physiology/Motor Systems Physiology/Muscle and Connective Tissue Proteins Risk factors Spinal muscular atrophy Studies Survival of Motor Neuron 1 Protein - metabolism Transgenes |
| title | A motor function for the DEAD-box RNA helicase, Gemin3, in Drosophila |
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