{"database":"biostudies-literature","file_versions":[],"scores":null,"additional":{"submitter":["Neuman SD"],"funding":["NINDS NIH HHS","NCI NIH HHS","NIH HHS","NIGMS NIH HHS","Brown University Carney Institute for Brain Science"],"pagination":["1356-1368"],"full_dataset_link":["https://www.ebi.ac.uk/biostudies/studies/S-EPMC12273619"],"repository":["biostudies-literature"],"omics_type":["Unknown"],"volume":["40(7)"],"pubmed_abstract":["<h4>Background</h4>Bridge-like lipid transfer proteins (BLTPs) mediate bulk lipid transport at membrane contact sites. Mutations in BLTPs are linked to both early-onset neurodevelopmental and later-onset neurodegenerative diseases, including movement disorders. The tissue specificity and temporal requirements of BLTPs in disease pathogenesis remain poorly understood.<h4>Objective</h4>The objective of this study was to determine tissue-specific and aging-dependent roles for VPS13A and BLTP2 using Drosophila models.<h4>Methods</h4>We generated tissue-specific knockdowns of the VPS13A ortholog (Vps13) and the BLTP2 ortholog (hobbit) in neurons and muscles of Drosophila. We analyzed age-dependent locomotor behavior, neurodegeneration, and synapse development and function.<h4>Results</h4>Neuron"],"journal":["Movement disorders : official journal of the Movement Disorder Society"],"pubmed_title":["Neurodegenerative and Neurodevelopmental Roles for Bulk Lipid Transporters VPS13A and BLTP2."],"pmcid":["PMC12273619"],"funding_grant_id":["R01NS078179","1R01GM155154","R01 NS078179","R01 GM155154","P30 CA014520","S10 OD023526","P40 OD018537"],"pubmed_authors":["Thakur RS","Gratz SJ","O'Connor-Giles KM","Bashirullah A","Neuman SD"],"additional_accession":[]},"is_claimable":false,"name":"Neurodegenerative and Neurodevelopmental Roles for Bulk Lipid Transporters VPS13A and BLTP2.","description":"<h4>Background</h4>Bridge-like lipid transfer proteins (BLTPs) mediate bulk lipid transport at membrane contact sites. Mutations in BLTPs are linked to both early-onset neurodevelopmental and later-onset neurodegenerative diseases, including movement disorders. The tissue specificity and temporal requirements of BLTPs in disease pathogenesis remain poorly understood.<h4>Objective</h4>The objective of this study was to determine tissue-specific and aging-dependent roles for VPS13A and BLTP2 using Drosophila models.<h4>Methods</h4>We generated tissue-specific knockdowns of the VPS13A ortholog (Vps13) and the BLTP2 ortholog (hobbit) in neurons and muscles of Drosophila. We analyzed age-dependent locomotor behavior, neurodegeneration, and synapse development and function.<h4>Results</h4>Neuron","dates":{"release":"2025-01-01T00:00:00Z","publication":"2025 Jul","modification":"2026-03-27T15:21:23.041Z","creation":"2025-08-23T03:09:07.334Z"},"accession":"S-EPMC12273619","cross_references":{"pubmed":["40152532"],"doi":["10.1002/mds.30178"]}}