{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,11,3]],"date-time":"2026-11-03T14:54:34Z","timestamp":1793717674109,"version":"4.6.1"},"reference-count":101,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2023,4,13]],"date-time":"2023-04-13T00:00:00Z","timestamp":1681344000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"funder":[{"DOI":"10.13039\/100001116","name":"Foundation Fighting Blindness","doi-asserted-by":"publisher","award":["PPA\u20100717\u20100719\u2010RAD"],"award-info":[{"award-number":["PPA\u20100717\u20100719\u2010RAD"]}],"id":[{"id":"10.13039\/100001116","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Basic Clin Pharma Tox"],"published-print":{"date-parts":[[2023,10]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>VLGR1\/ADGRV1 (very large G protein\u2010coupled receptor\u20101) is the largest known adhesion G protein\u2010coupled receptor. Mutations in <jats:italic>VLGR1<\/jats:italic>\/<jats:italic>ADGRV1<\/jats:italic> cause Usher syndrome (USH), the most common form of hereditary deaf\u2010blindness, and have been additionally linked to epilepsy. Although <jats:italic>VLGR1\/ADGRV1<\/jats:italic> is almost ubiquitously expressed, little is known about the subcellular function and signalling of the VLGR1 protein and thus about mechanisms underlying the development of diseases. Using affinity proteomics, we identified key components of autophagosomes as putative interacting proteins of VLGR1. In addition, whole transcriptome sequencing of the retinae of the Vlgr1\/del7TM mouse model revealed altered expression profiles of gene\u2010related autophagy. Monitoring autophagy by immunoblotting and immunocytochemistry of the LC3 and p62 as autophagy marker proteins revealed evoked autophagy in VLGR1\u2010deficient hTERT\u2010RPE1 cells and USH2C patient\u2010derived fibroblasts. Our data demonstrate the molecular and functional interaction of VLGR1 with key components of the autophagy process and point to an essential role of VLGR1 in the regulation of autophagy at internal membranes. The close association of VLGR1 with autophagy helps to explain the pathomechanisms underlying human USH and epilepsy related to VLGR1 defects.<\/jats:p>","DOI":"10.1111\/bcpt.13869","type":"journal-article","created":{"date-parts":[[2023,4,1]],"date-time":"2023-04-01T09:43:04Z","timestamp":1680342184000},"page":"313-330","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":18,"title":["The adhesion G protein\u2010coupled receptor VLGR1\/ADGRV1 controls autophagy"],"prefix":"10.1111","volume":"133","author":[{"ORCID":"https:\/\/orcid.org\/0009-0002-9253-9144","authenticated-orcid":false,"given":"Joshua","family":"Linnert","sequence":"first","affiliation":[{"name":"Institute of Molecular Physiology, Molecular Cell Biology Johannes Gutenberg University Mainz  Mainz Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7967-9041","authenticated-orcid":false,"given":"Baran E.","family":"G\u00fcler","sequence":"additional","affiliation":[{"name":"Institute of Molecular Physiology, Molecular Cell Biology Johannes Gutenberg University Mainz  Mainz Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jacek","family":"Krzysko","sequence":"additional","affiliation":[{"name":"Institute of Molecular Physiology, Molecular Cell Biology Johannes Gutenberg University Mainz  Mainz Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4756-5872","authenticated-orcid":false,"given":"Uwe","family":"Wolfrum","sequence":"additional","affiliation":[{"name":"Institute of Molecular Physiology, Molecular Cell Biology Johannes Gutenberg University Mainz  Mainz Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2023,4,13]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.0693\u201006.2006"},{"key":"e_1_2_8_3_1","doi-asserted-by":"publisher","DOI":"10.1007\/978\u20101\u20104419\u20107913\u20101_6"},{"key":"e_1_2_8_4_1","doi-asserted-by":"publisher","DOI":"10.1007\/978\u20103\u2010319\u201041523\u20109_6"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41392\u2010022\u201001083\u2010y"},{"key":"e_1_2_8_6_1","doi-asserted-by":"publisher","DOI":"10.3390\/molecules27103108"},{"key":"e_1_2_8_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.exer.2005.11.010"},{"key":"e_1_2_8_8_1","doi-asserted-by":"publisher","DOI":"10.3390\/ijms22136723"},{"key":"e_1_2_8_9_1","doi-asserted-by":"publisher","DOI":"10.1111\/epi.13980"},{"key":"e_1_2_8_10_1","doi-asserted-by":"publisher","DOI":"10.3389\/fneur.2021.738272"},{"key":"e_1_2_8_11_1","doi-asserted-by":"publisher","DOI":"10.3389\/fnmol.2022.864074"},{"key":"e_1_2_8_12_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ydbio.2005.01.014"},{"key":"e_1_2_8_13_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/ddm285"},{"key":"e_1_2_8_14_1","doi-asserted-by":"publisher","DOI":"10.1111\/nyas.14220"},{"key":"e_1_2_8_15_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.isci.2021.102283"},{"key":"e_1_2_8_16_1","doi-asserted-by":"publisher","DOI":"10.1111\/bcpt.13860"},{"key":"e_1_2_8_17_1","doi-asserted-by":"publisher","DOI":"10.3390\/cells11182790"},{"key":"e_1_2_8_18_1","doi-asserted-by":"publisher","DOI":"10.1038\/nature11910"},{"key":"e_1_2_8_19_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cell.2011.10.026"},{"key":"e_1_2_8_20_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1348\u20100421.2010.00271.x"},{"key":"e_1_2_8_21_1","doi-asserted-by":"publisher","DOI":"10.1038\/nri3532"},{"key":"e_1_2_8_22_1","doi-asserted-by":"publisher","DOI":"10.1007\/BF03402040"},{"key":"e_1_2_8_23_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41419\u2010021\u201004121\u20109"},{"key":"e_1_2_8_24_1","doi-asserted-by":"publisher","DOI":"10.1515\/hsz\u20102012\u20100119"},{"key":"e_1_2_8_25_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cell.2020.02.017"},{"key":"e_1_2_8_26_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41580\u2010022\u201000542\u20102"},{"key":"e_1_2_8_27_1","doi-asserted-by":"publisher","DOI":"10.1080\/15548627.2018.1463121"},{"key":"e_1_2_8_28_1","doi-asserted-by":"publisher","DOI":"10.3389\/fonc.2021.632956"},{"key":"e_1_2_8_29_1","doi-asserted-by":"publisher","DOI":"10.1002\/path.3025"},{"key":"e_1_2_8_30_1","unstructured":"Usher Syndrome Database. Accessed November 9 2022.https:\/\/databases.lovd.nl\/shared\/variants\/GPR98\/unique"},{"key":"e_1_2_8_31_1","doi-asserted-by":"publisher","DOI":"10.1038\/ncomms11491"},{"key":"e_1_2_8_32_1","unstructured":"RStudio Team.RStudio: Integrated Development Environment for R.2020."},{"key":"e_1_2_8_33_1","doi-asserted-by":"publisher","DOI":"10.1111\/bcpt.13492"},{"key":"e_1_2_8_34_1","doi-asserted-by":"publisher","DOI":"10.1038\/cdd.2013.187"},{"key":"e_1_2_8_35_1","doi-asserted-by":"publisher","DOI":"10.1080\/15548627.2016.1241924"},{"key":"e_1_2_8_36_1","doi-asserted-by":"publisher","DOI":"10.1038\/ncb2589"},{"key":"e_1_2_8_37_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.molcel.2013.12.014"},{"key":"e_1_2_8_38_1","doi-asserted-by":"publisher","DOI":"10.1038\/embor.2013.22"},{"key":"e_1_2_8_39_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.molcel.2013.08.041"},{"key":"e_1_2_8_40_1","doi-asserted-by":"publisher","DOI":"10.3390\/cells8080849"},{"key":"e_1_2_8_41_1","doi-asserted-by":"publisher","DOI":"10.1080\/15548627.2019.1596489"},{"key":"e_1_2_8_42_1","doi-asserted-by":"publisher","DOI":"10.1080\/27694127.2022.2131212"},{"key":"e_1_2_8_43_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.molcel.2019.03.033"},{"key":"e_1_2_8_44_1","doi-asserted-by":"publisher","DOI":"10.1242\/jcs.03172"},{"key":"e_1_2_8_45_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M109.048140"},{"key":"e_1_2_8_46_1","doi-asserted-by":"publisher","DOI":"10.4161\/auto.25399"},{"key":"e_1_2_8_47_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.febslet.2013.06.011"},{"key":"e_1_2_8_48_1","doi-asserted-by":"publisher","DOI":"10.1038\/srep01055"},{"key":"e_1_2_8_49_1","doi-asserted-by":"publisher","DOI":"10.33594\/000000208"},{"key":"e_1_2_8_50_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41419\u2010021\u201003694\u20109"},{"key":"e_1_2_8_51_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.mam.2006.08.005"},{"key":"e_1_2_8_52_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41421\u2010020\u20100141\u20107"},{"key":"e_1_2_8_53_1","doi-asserted-by":"publisher","DOI":"10.1002\/jnr.24034"},{"key":"e_1_2_8_54_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cell.2012.11.001"},{"key":"e_1_2_8_55_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.bbamcr.2009.09.011"},{"key":"e_1_2_8_56_1","doi-asserted-by":"publisher","DOI":"10.4161\/auto.25413"},{"key":"e_1_2_8_57_1","doi-asserted-by":"publisher","DOI":"10.4161\/auto.22739"},{"key":"e_1_2_8_58_1","doi-asserted-by":"publisher","DOI":"10.15252\/embr.201643147"},{"key":"e_1_2_8_59_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cell.2016.11.042"},{"key":"e_1_2_8_60_1","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.201007013"},{"key":"e_1_2_8_61_1","doi-asserted-by":"publisher","DOI":"10.1038\/ncb2012"},{"key":"e_1_2_8_62_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41467\u2010018\u201005722\u20103"},{"key":"e_1_2_8_63_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0094634"},{"key":"e_1_2_8_64_1","doi-asserted-by":"publisher","DOI":"10.1080\/15548627.2015.1042195"},{"key":"e_1_2_8_65_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41467\u2010021\u201026824\u20105"},{"key":"e_1_2_8_66_1","doi-asserted-by":"publisher","DOI":"10.1038\/srep14827"},{"key":"e_1_2_8_67_1","doi-asserted-by":"publisher","DOI":"10.1126\/science.aag1417"},{"key":"e_1_2_8_68_1","doi-asserted-by":"publisher","DOI":"10.1038\/nature05925"},{"key":"e_1_2_8_69_1","doi-asserted-by":"publisher","DOI":"10.4161\/auto.7.7.15844"},{"key":"e_1_2_8_70_1","doi-asserted-by":"publisher","DOI":"10.3109\/10715762.2014.936431"},{"key":"e_1_2_8_71_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41388\u2010021\u201001820\u20107"},{"key":"e_1_2_8_72_1","doi-asserted-by":"publisher","DOI":"10.4161\/auto.22742"},{"key":"e_1_2_8_73_1","doi-asserted-by":"publisher","DOI":"10.1038\/ncb2021"},{"key":"e_1_2_8_74_1","doi-asserted-by":"publisher","DOI":"10.4161\/auto.26168"},{"key":"e_1_2_8_75_1","doi-asserted-by":"publisher","DOI":"10.1007\/s13277\u2010013\u20100794\u2010z"},{"key":"e_1_2_8_76_1","doi-asserted-by":"publisher","DOI":"10.1038\/cr.2009.129"},{"key":"e_1_2_8_77_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.molcel.2019.10.035"},{"key":"e_1_2_8_78_1","doi-asserted-by":"publisher","DOI":"10.15252\/embr.201845889"},{"key":"e_1_2_8_79_1","doi-asserted-by":"publisher","DOI":"10.1038\/emboj.2013.8"},{"key":"e_1_2_8_80_1","doi-asserted-by":"publisher","DOI":"10.1242\/jcs.093773"},{"key":"e_1_2_8_81_1","doi-asserted-by":"publisher","DOI":"10.1080\/15548627.2020.1797280"},{"key":"e_1_2_8_82_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41467\u2010020\u201020185\u20101"},{"key":"e_1_2_8_83_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41467\u2010019\u201011636\u20105"},{"key":"e_1_2_8_84_1","doi-asserted-by":"publisher","DOI":"10.3390\/ijms23063025"},{"key":"e_1_2_8_85_1","doi-asserted-by":"publisher","DOI":"10.1053\/j.gastro.2009.04.003"},{"key":"e_1_2_8_86_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/ddm100"},{"key":"e_1_2_8_87_1","doi-asserted-by":"publisher","DOI":"10.4161\/auto.25684"},{"key":"e_1_2_8_88_1","doi-asserted-by":"publisher","DOI":"10.1038\/ncomms6637"},{"key":"e_1_2_8_89_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13041\u2010016\u20100212\u20108"},{"key":"e_1_2_8_90_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.tem.2014.03.006"},{"key":"e_1_2_8_91_1","doi-asserted-by":"publisher","DOI":"10.1152\/physrev.00027.2021"},{"key":"e_1_2_8_92_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.1318501110"},{"key":"e_1_2_8_93_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M114.549816"},{"key":"e_1_2_8_94_1","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.202004179"},{"key":"e_1_2_8_95_1","doi-asserted-by":"publisher","DOI":"10.1002\/hep.22146"},{"key":"e_1_2_8_96_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.celrep.2016.04.065"},{"key":"e_1_2_8_97_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41467\u2010021\u201023599\u20107"},{"key":"e_1_2_8_98_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.heares.2019.02.007"},{"key":"e_1_2_8_99_1","volume-title":"Usher Syndrome: Pathogenesis, Diagnosis and Therapy","author":"Wolfrum U","year":"2011"},{"key":"e_1_2_8_100_1","doi-asserted-by":"publisher","DOI":"10.1093\/HMG\/DDAA004"},{"key":"e_1_2_8_101_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00702\u2010014\u20101312\u20101"},{"key":"e_1_2_8_102_1","doi-asserted-by":"publisher","DOI":"10.3390\/ijms21051642"}],"container-title":["Basic &amp; Clinical Pharmacology &amp; Toxicology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/bcpt.13869","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,11,14]],"date-time":"2023-11-14T10:20:54Z","timestamp":1699957254000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/bcpt.13869"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,4,13]]},"references-count":101,"journal-issue":{"issue":"4","published-print":{"date-parts":[[2023,10]]}},"alternative-id":["10.1111\/bcpt.13869"],"URL":"https:\/\/doi.org\/10.1111\/bcpt.13869","archive":["Portico"],"relation":{},"ISSN":["1742-7835","1742-7843"],"issn-type":[{"value":"1742-7835","type":"print"},{"value":"1742-7843","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,4,13]]},"assertion":[{"value":"2023-01-03","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2023-03-28","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2023-04-13","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}