{"data":{"id":"10.6084/m9.figshare.1632843","type":"dois","attributes":{"doi":"10.6084/m9.figshare.1632843","prefix":"10.6084","suffix":"m9.figshare.1632843","identifiers":[],"alternateIdentifiers":[],"creators":[{"name":"Bouché, Valentina","nameType":"Personal","givenName":"Valentina","familyName":"Bouché","affiliation":[],"nameIdentifiers":[]},{"name":"Espinosa, Alma Perez","nameType":"Personal","givenName":"Alma Perez","familyName":"Espinosa","affiliation":[],"nameIdentifiers":[]},{"name":"Leone, Luigi","nameType":"Personal","givenName":"Luigi","familyName":"Leone","affiliation":[],"nameIdentifiers":[]},{"name":"Sardiello, Marco","nameType":"Personal","givenName":"Marco","familyName":"Sardiello","affiliation":[],"nameIdentifiers":[]},{"name":"Ballabio, Andrea","nameType":"Personal","givenName":"Andrea","familyName":"Ballabio","affiliation":[],"nameIdentifiers":[]},{"name":"Botas, Juan","nameType":"Personal","givenName":"Juan","familyName":"Botas","affiliation":[],"nameIdentifiers":[]}],"titles":[{"title":"\u003ci\u003eDrosophila\u003c/i\u003e Mitf regulates the V-ATPase and the lysosomal-autophagic pathway"}],"publisher":"Taylor \u0026 Francis","container":{},"publicationYear":2016,"subjects":[{"subject":"Cell Biology"},{"subject":"Genetics"},{"subject":"FOS: Biological sciences","schemeUri":"http://www.oecd.org/science/inno/38235147.pdf","subjectScheme":"Fields of Science and Technology (FOS)"},{"subject":"FOS: Biological sciences","subjectScheme":"Fields of Science and Technology (FOS)"},{"subject":"Molecular Biology"},{"subject":"Neuroscience"},{"subject":"59999 Environmental Sciences not elsewhere classified","subjectScheme":"FOR"},{"subject":"FOS: Earth and related environmental sciences","schemeUri":"http://www.oecd.org/science/inno/38235147.pdf","subjectScheme":"Fields of Science and Technology (FOS)"},{"subject":"FOS: Earth and related environmental sciences","subjectScheme":"Fields of Science and Technology (FOS)"},{"subject":"Immunology"},{"subject":"FOS: Clinical medicine","schemeUri":"http://www.oecd.org/science/inno/38235147.pdf","subjectScheme":"Fields of Science and Technology (FOS)"},{"subject":"FOS: Clinical medicine","subjectScheme":"Fields of Science and Technology (FOS)"},{"subject":"Developmental Biology"},{"subject":"111714 Mental Health","subjectScheme":"FOR"},{"subject":"FOS: Health sciences","schemeUri":"http://www.oecd.org/science/inno/38235147.pdf","subjectScheme":"Fields of Science and Technology (FOS)"},{"subject":"FOS: Health sciences","subjectScheme":"Fields of Science and Technology (FOS)"},{"subject":"Computational Biology"}],"contributors":[],"dates":[{"date":"2016-01-13","dateType":"Created"},{"date":"2016-04-06","dateType":"Updated"},{"date":"2016","dateType":"Issued"}],"language":null,"types":{"ris":"DATA","bibtex":"misc","citeproc":"dataset","schemaOrg":"Dataset","resourceType":"Dataset","resourceTypeGeneral":"Dataset"},"relatedIdentifiers":[{"relationType":"IsSupplementTo","relatedIdentifier":"10.1080/15548627.2015.1134081","relatedIdentifierType":"DOI"}],"relatedItems":[],"sizes":["2866386 Bytes"],"formats":[],"version":null,"rightsList":[{"rights":"Creative Commons Attribution 4.0 International","rightsUri":"https://creativecommons.org/licenses/by/4.0/legalcode","schemeUri":"https://spdx.org/licenses/","rightsIdentifier":"cc-by-4.0","rightsIdentifierScheme":"SPDX"}],"descriptions":[{"description":"An evolutionarily conserved gene network regulates the expression of genes involved in lysosome biogenesis, autophagy, and lipid metabolism. In mammals, TFEB and other members of the MiTF-TFE family of transcription factors control this network. Here we report that the lysosomal-autophagy pathway is controlled by \u003ci\u003eMitf\u003c/i\u003e gene in \u003ci\u003eDrosophila melanogaster\u003c/i\u003e. Mitf is the single MiTF-TFE family member in \u003ci\u003eDrosophila\u003c/i\u003e and prior to this work was known only for its function in eye development. We show that Mitf regulates the expression of genes encoding V-ATPase subunits as well as many additional genes involved in the lysosomal-autophagy pathway. Reduction of Mitf function leads to abnormal lysosomes and impairs autophagosome fusion and lipid breakdown during the response to starvation. In contrast, elevated Mitf levels increase the number of lysosomes, autophagosomes and autolysosomes, and decrease the size of lipid droplets. Inhibition of \u003ci\u003eDrosophila\u003c/i\u003e MTORC1 induces Mitf translocation to the nucleus, underscoring conserved regulatory mechanisms between \u003ci\u003eDrosophila\u003c/i\u003e and mammalian systems. Furthermore, we show Mitf-mediated clearance of cytosolic and nuclear expanded ATXN1 (ataxin 1) in a cellular model of spinocerebellar ataxia type 1 (SCA1). This remarkable observation illustrates the potential of the lysosomal-autophagy system to prevent toxic protein aggregation in both the cytoplasmic and nuclear compartments. We anticipate that the genetics of the \u003ci\u003eDrosophila\u003c/i\u003e model and the absence of redundant MIT transcription factors will be exploited to investigate the regulation and function of the lysosomal-autophagy gene network.","descriptionType":"Abstract"}],"geoLocations":[],"fundingReferences":[],"xml":"PD94bWwgdmVyc2lvbj0iMS4wIiBlbmNvZGluZz0iVVRGLTgiPz4KPHJlc291cmNlIHhtbG5zPSJodHRwOi8vZGF0YWNpdGUub3JnL3NjaGVtYS9rZXJuZWwtNCIgeG1sbnM6eHNpPSJodHRwOi8vd3d3LnczLm9yZy8yMDAxL1hNTFNjaGVtYS1pbnN0YW5jZSIgeHNpOnNjaGVtYUxvY2F0aW9uPSJodHRwOi8vZGF0YWNpdGUub3JnL3NjaGVtYS9rZXJuZWwtNCBodHRwOi8vc2NoZW1hLmRhdGFjaXRlLm9yZy9tZXRhL2tlcm5lbC00LjEvbWV0YWRhdGEueHNkIj4KICA8aWRlbnRpZmllciBpZGVudGlmaWVyVHlwZT0iRE9JIj4xMC42MDg0L005LkZJR1NIQVJFLjE2MzI4NDM8L2lkZW50aWZpZXI+CiAgPGNyZWF0b3JzPgogICAgPGNyZWF0b3I+CiAgICAgIDxjcmVhdG9yTmFtZT5WYWxlbnRpbmEgQm91Y2jDqTwvY3JlYXRvck5hbWU+CiAgICA8L2NyZWF0b3I+CiAgICA8Y3JlYXRvcj4KICAgICAgPGNyZWF0b3JOYW1lPkFsbWEgUGVyZXogRXNwaW5vc2E8L2NyZWF0b3JOYW1lPgogICAgPC9jcmVhdG9yPgogICAgPGNyZWF0b3I+CiAgICAgIDxjcmVhdG9yTmFtZT5MdWlnaSBMZW9uZTwvY3JlYXRvck5hbWU+CiAgICA8L2NyZWF0b3I+CiAgICA8Y3JlYXRvcj4KICAgICAgPGNyZWF0b3JOYW1lPk1hcmNvIFNhcmRpZWxsbzwvY3JlYXRvck5hbWU+CiAgICA8L2NyZWF0b3I+CiAgICA8Y3JlYXRvcj4KICAgICAgPGNyZWF0b3JOYW1lPkFuZHJlYSBCYWxsYWJpbzwvY3JlYXRvck5hbWU+CiAgICA8L2NyZWF0b3I+CiAgICA8Y3JlYXRvcj4KICAgICAgPGNyZWF0b3JOYW1lPkp1YW4gQm90YXM8L2NyZWF0b3JOYW1lPgogICAgPC9jcmVhdG9yPgogIDwvY3JlYXRvcnM+CiAgPHRpdGxlcz4KICAgIDx0aXRsZT4mbHQ7aSZndDtEcm9zb3BoaWxhJmx0Oy9pJmd0OyBNaXRmIHJlZ3VsYXRlcyB0aGUgVi1BVFBhc2UgYW5kIHRoZSBseXNvc29tYWwtYXV0b3BoYWdpYyBwYXRod2F5PC90aXRsZT4KICA8L3RpdGxlcz4KICA8ZGVzY3JpcHRpb25zPgogICAgPGRlc2NyaXB0aW9uIGRlc2NyaXB0aW9uVHlwZT0iQWJzdHJhY3QiPiZsdDtwJmd0O0FuIGV2b2x1dGlvbmFyaWx5IGNvbnNlcnZlZCBnZW5lIG5ldHdvcmsgcmVndWxhdGVzIHRoZSBleHByZXNzaW9uIG9mIGdlbmVzIGludm9sdmVkIGluIGx5c29zb21lIGJpb2dlbmVzaXMsIGF1dG9waGFneSwgYW5kIGxpcGlkIG1ldGFib2xpc20uIEluIG1hbW1hbHMsIFRGRUIgYW5kIG90aGVyIG1lbWJlcnMgb2YgdGhlIE1pVEYtVEZFIGZhbWlseSBvZiB0cmFuc2NyaXB0aW9uIGZhY3RvcnMgY29udHJvbCB0aGlzIG5ldHdvcmsuIEhlcmUgd2UgcmVwb3J0IHRoYXQgdGhlIGx5c29zb21hbC1hdXRvcGhhZ3kgcGF0aHdheSBpcyBjb250cm9sbGVkIGJ5ICZsdDtpJmd0O01pdGYmbHQ7L2kmZ3Q7IGdlbmUgaW4gJmx0O2kmZ3Q7RHJvc29waGlsYSBtZWxhbm9nYXN0ZXImbHQ7L2kmZ3Q7LiBNaXRmIGlzIHRoZSBzaW5nbGUgTWlURi1URkUgZmFtaWx5IG1lbWJlciBpbiAmbHQ7aSZndDtEcm9zb3BoaWxhJmx0Oy9pJmd0OyBhbmQgcHJpb3IgdG8gdGhpcyB3b3JrIHdhcyBrbm93biBvbmx5IGZvciBpdHMgZnVuY3Rpb24gaW4gZXllIGRldmVsb3BtZW50LiBXZSBzaG93IHRoYXQgTWl0ZiByZWd1bGF0ZXMgdGhlIGV4cHJlc3Npb24gb2YgZ2VuZXMgZW5jb2RpbmcgVi1BVFBhc2Ugc3VidW5pdHMgYXMgd2VsbCBhcyBtYW55IGFkZGl0aW9uYWwgZ2VuZXMgaW52b2x2ZWQgaW4gdGhlIGx5c29zb21hbC1hdXRvcGhhZ3kgcGF0aHdheS4gUmVkdWN0aW9uIG9mIE1pdGYgZnVuY3Rpb24gbGVhZHMgdG8gYWJub3JtYWwgbHlzb3NvbWVzIGFuZCBpbXBhaXJzIGF1dG9waGFnb3NvbWUgZnVzaW9uIGFuZCBsaXBpZCBicmVha2Rvd24gZHVyaW5nIHRoZSByZXNwb25zZSB0byBzdGFydmF0aW9uLiBJbiBjb250cmFzdCwgZWxldmF0ZWQgTWl0ZiBsZXZlbHMgaW5jcmVhc2UgdGhlIG51bWJlciBvZiBseXNvc29tZXMsIGF1dG9waGFnb3NvbWVzIGFuZCBhdXRvbHlzb3NvbWVzLCBhbmQgZGVjcmVhc2UgdGhlIHNpemUgb2YgbGlwaWQgZHJvcGxldHMuIEluaGliaXRpb24gb2YgJmx0O2kmZ3Q7RHJvc29waGlsYSZsdDsvaSZndDsgTVRPUkMxIGluZHVjZXMgTWl0ZiB0cmFuc2xvY2F0aW9uIHRvIHRoZSBudWNsZXVzLCB1bmRlcnNjb3JpbmcgY29uc2VydmVkIHJlZ3VsYXRvcnkgbWVjaGFuaXNtcyBiZXR3ZWVuICZsdDtpJmd0O0Ryb3NvcGhpbGEmbHQ7L2kmZ3Q7IGFuZCBtYW1tYWxpYW4gc3lzdGVtcy4gRnVydGhlcm1vcmUsIHdlIHNob3cgTWl0Zi1tZWRpYXRlZCBjbGVhcmFuY2Ugb2YgY3l0b3NvbGljIGFuZCBudWNsZWFyIGV4cGFuZGVkIEFUWE4xIChhdGF4aW4gMSkgaW4gYSBjZWxsdWxhciBtb2RlbCBvZiBzcGlub2NlcmViZWxsYXIgYXRheGlhIHR5cGUgMSAoU0NBMSkuIFRoaXMgcmVtYXJrYWJsZSBvYnNlcnZhdGlvbiBpbGx1c3RyYXRlcyB0aGUgcG90ZW50aWFsIG9mIHRoZSBseXNvc29tYWwtYXV0b3BoYWd5IHN5c3RlbSB0byBwcmV2ZW50IHRveGljIHByb3RlaW4gYWdncmVnYXRpb24gaW4gYm90aCB0aGUgY3l0b3BsYXNtaWMgYW5kIG51Y2xlYXIgY29tcGFydG1lbnRzLiBXZSBhbnRpY2lwYXRlIHRoYXQgdGhlIGdlbmV0aWNzIG9mIHRoZSAmbHQ7aSZndDtEcm9zb3BoaWxhJmx0Oy9pJmd0OyBtb2RlbCBhbmQgdGhlIGFic2VuY2Ugb2YgcmVkdW5kYW50IE1JVCB0cmFuc2NyaXB0aW9uIGZhY3RvcnMgd2lsbCBiZSBleHBsb2l0ZWQgdG8gaW52ZXN0aWdhdGUgdGhlIHJlZ3VsYXRpb24gYW5kIGZ1bmN0aW9uIG9mIHRoZSBseXNvc29tYWwtYXV0b3BoYWd5IGdlbmUgbmV0d29yay4mbHQ7L3AmZ3Q7PC9kZXNjcmlwdGlvbj4KICA8L2Rlc2NyaXB0aW9ucz4KICA8c3ViamVjdHM+CiAgICA8c3ViamVjdD5DZWxsIEJpb2xvZ3k8L3N1YmplY3Q+CiAgICA8c3ViamVjdD5HZW5ldGljczwvc3ViamVjdD4KICAgIDxzdWJqZWN0Pk1vbGVjdWxhciBCaW9sb2d5PC9zdWJqZWN0PgogICAgPHN1YmplY3Q+TmV1cm9zY2llbmNlPC9zdWJqZWN0PgogICAgPHN1YmplY3Qgc2NoZW1lVVJJPSJodHRwOi8vd3d3LmFicy5nb3YuYXUvYXVzc3RhdHMvYWJzQC5uc2YvMC82QkI0MjdBQjk2OTZDMjI1Q0EyNTc0MTgwMDA0NDYzRSIgc3ViamVjdFNjaGVtZT0iRk9SIj41OTk5OSBFbnZpcm9ubWVudGFsIFNjaWVuY2VzIG5vdCBlbHNld2hlcmUgY2xhc3NpZmllZDwvc3ViamVjdD4KICAgIDxzdWJqZWN0PkltbXVub2xvZ3k8L3N1YmplY3Q+CiAgICA8c3ViamVjdD5EZXZlbG9wbWVudGFsIEJpb2xvZ3k8L3N1YmplY3Q+CiAgICA8c3ViamVjdCBzY2hlbWVVUkk9Imh0dHA6Ly93d3cuYWJzLmdvdi5hdS9hdXNzdGF0cy9hYnNALm5zZi8wLzZCQjQyN0FCOTY5NkMyMjVDQTI1NzQxODAwMDQ0NjNFIiBzdWJqZWN0U2NoZW1lPSJGT1IiPjExMTcxNCBNZW50YWwgSGVhbHRoPC9zdWJqZWN0PgogICAgPHN1YmplY3Q+Q29tcHV0YXRpb25hbCAgQmlvbG9neTwvc3ViamVjdD4KICA8L3N1YmplY3RzPgogIDxwdWJsaXNoZXI+VGF5bG9yICZhbXA7IEZyYW5jaXM8L3B1Ymxpc2hlcj4KICA8cHVibGljYXRpb25ZZWFyPjIwMTY8L3B1YmxpY2F0aW9uWWVhcj4KICA8ZGF0ZXM+CiAgICA8ZGF0ZSBkYXRlVHlwZT0iQ3JlYXRlZCI+MjAxNi0wMS0xMzwvZGF0ZT4KICAgIDxkYXRlIGRhdGVUeXBlPSJVcGRhdGVkIj4yMDE2LTA0LTA2PC9kYXRlPgogIDwvZGF0ZXM+CiAgPHJlc291cmNlVHlwZSByZXNvdXJ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