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More than 100 distinct forms of CDGs have been identified and most of them cause severe neurological impairment. The Conserved Oligomeric Golgi (COG) complex mediates tethering of vesicles carrying glycosylation enzymes across the Golgi cisternae. Mutations affecting human COG1, COG2 and COG4\u2013COG8 cause monogenic forms of inherited, autosomal recessive CDGs. We have generated a Drosophila COG7-CDG model that closely parallels the pathological characteristics of COG7-CDG patients, including pronounced neuromotor defects associated with altered N-glycome profiles. Consistent with these alterations, larval neuromuscular junctions of Cog7 mutants exhibit a significant reduction in bouton numbers. We demonstrate that the COG complex cooperates with Rab1 and Golgi phosphoprotein 3 to regulate Golgi trafficking and that overexpression of Rab1 can rescue the cytokinesis and locomotor defects associated with loss of Cog7. Our results suggest that the Drosophila COG7-CDG model can be used to test novel potential therapeutic strategies by modulating trafficking pathways.<\/jats:p>","DOI":"10.1242\/jcs.209049","type":"journal-article","created":{"date-parts":[[2017,9,7]],"date-time":"2017-09-07T20:25:20Z","timestamp":1504815920000},"page":"3637-3649","source":"Crossref","is-referenced-by-count":27,"title":["COG7 deficiency in\n                    <i>Drosophila<\/i>\n                    generates multifaceted developmental, behavioral and protein glycosylation phenotypes"],"prefix":"10.1242","volume":"130","author":[{"given":"Anna","family":"Frappaolo","sequence":"first","affiliation":[{"name":"Istituto di Biologia e Patologia Molecolari del CNR 1 , Dipartimento di Biologia e Biotecnologie , , Roma, Piazzale A. Moro 5, 00185 Roma , Italy"},{"name":"Universit\u00e0 Sapienza di Roma 1 , Dipartimento di Biologia e Biotecnologie , , Roma, Piazzale A. Moro 5, 00185 Roma , Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Stefano","family":"Sechi","sequence":"additional","affiliation":[{"name":"Istituto di Biologia e Patologia Molecolari del CNR 1 , Dipartimento di Biologia e Biotecnologie , , Roma, Piazzale A. Moro 5, 00185 Roma , Italy"},{"name":"Universit\u00e0 Sapienza di Roma 1 , Dipartimento di Biologia e Biotecnologie , , Roma, Piazzale A. Moro 5, 00185 Roma , Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tadahiro","family":"Kumagai","sequence":"additional","affiliation":[{"name":"Complex Carbohydrate Research Center, The University of Georgia 2 , 315 Riverbend Rd., Athens, GA 30602 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sarah","family":"Robinson","sequence":"additional","affiliation":[{"name":"Complex Carbohydrate Research Center, The University of Georgia 2 , 315 Riverbend Rd., Athens, GA 30602 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Roberta","family":"Fraschini","sequence":"additional","affiliation":[{"name":"Universit\u00e0 degli studi di Milano Bicocca 3 Dipartimento di Biotecnologie e Bioscienze , , 20126 Milano , Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Angela","family":"Karimpour-Ghahnavieh","sequence":"additional","affiliation":[{"name":"Istituto di Biologia e Patologia Molecolari del CNR 1 , Dipartimento di Biologia e Biotecnologie , , Roma, Piazzale A. Moro 5, 00185 Roma , Italy"},{"name":"Universit\u00e0 Sapienza di Roma 1 , Dipartimento di Biologia e Biotecnologie , , Roma, Piazzale A. Moro 5, 00185 Roma , Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Giorgio","family":"Belloni","sequence":"additional","affiliation":[{"name":"Istituto di Biologia e Patologia Molecolari del CNR 1 , Dipartimento di Biologia e Biotecnologie , , Roma, Piazzale A. Moro 5, 00185 Roma , Italy"},{"name":"Universit\u00e0 Sapienza di Roma 1 , Dipartimento di Biologia e Biotecnologie , , Roma, Piazzale A. Moro 5, 00185 Roma , Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Roberto","family":"Piergentili","sequence":"additional","affiliation":[{"name":"Istituto di Biologia e Patologia Molecolari del CNR 1 , Dipartimento di Biologia e Biotecnologie , , Roma, Piazzale A. Moro 5, 00185 Roma , Italy"},{"name":"Universit\u00e0 Sapienza di Roma 1 , Dipartimento di Biologia e Biotecnologie , , Roma, Piazzale A. 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