{"database":"Pride","file_versions":[{"headers":{"Content-Type":["application/json"]},"body":{"files":{"Txt":["ftp://ftp.pride.ebi.ac.uk/pride/data/archive/2023/03/PXD021514/Q53R_1_DTASelect-filter.txt","ftp://ftp.pride.ebi.ac.uk/pride/data/archive/2023/03/PXD021514/WT_2_DTASelect-filter.txt","ftp://ftp.pride.ebi.ac.uk/pride/data/archive/2023/03/PXD021514/Q53R_2_DTASelect-filter.txt","ftp://ftp.pride.ebi.ac.uk/pride/data/archive/2023/03/PXD021514/WT_1_DTASelect-filter.txt"],"Raw":["ftp://ftp.pride.ebi.ac.uk/pride/data/archive/2023/03/PXD021514/20190319_50cmColumn_YSU_KYI_OSBPL2_Q53R_2.raw","ftp://ftp.pride.ebi.ac.uk/pride/data/archive/2023/03/PXD021514/20190319_50cmColumn_YSU_KYI_OSBPL2_WT_1.raw","ftp://ftp.pride.ebi.ac.uk/pride/data/archive/2023/03/PXD021514/20190319_50cmColumn_YSU_KYI_OSBPL2_WT_2.raw","ftp://ftp.pride.ebi.ac.uk/pride/data/archive/2023/03/PXD021514/20190319_50cmColumn_YSU_KYI_OSBPL2_Q53R_1.raw"],"Mzxml":["ftp://ftp.pride.ebi.ac.uk/pride/data/archive/2023/03/PXD021514/20190319_50cmColumn_YSU_KYI_OSBPL2_WT_1.mzXML","ftp://ftp.pride.ebi.ac.uk/pride/data/archive/2023/03/PXD021514/20190319_50cmColumn_YSU_KYI_OSBPL2_WT_2.mzXML","ftp://ftp.pride.ebi.ac.uk/pride/data/archive/2023/03/PXD021514/20190319_50cmColumn_YSU_KYI_OSBPL2_Q53R_2.mzXML","ftp://ftp.pride.ebi.ac.uk/pride/data/archive/2023/03/PXD021514/20190319_50cmColumn_YSU_KYI_OSBPL2_Q53R_1.mzXML"],"Mzid":["ftp://ftp.pride.ebi.ac.uk/pride/data/archive/2023/03/PXD021514/WT_2_DTASelect-filter.mzid.gz","ftp://ftp.pride.ebi.ac.uk/pride/data/archive/2023/03/PXD021514/Q53R_1_DTASelect-filter.mzid.gz","ftp://ftp.pride.ebi.ac.uk/pride/data/archive/2023/03/PXD021514/Q53R_2_DTASelect-filter.mzid.gz","ftp://ftp.pride.ebi.ac.uk/pride/data/archive/2023/03/PXD021514/WT_1_DTASelect-filter.mzid.gz"],"Fasta":["ftp://ftp.pride.ebi.ac.uk/pride/data/archive/2023/03/PXD021514/20190402_Uniprot_Human_reviewed_test_Human_contaminant_04-01-2019_reversed.fasta"]},"type":"primary"},"statusCode":"OK","statusCodeValue":200}],"scores":null,"additional":{"labhead_mail":["rhduddlr91@naver.com"],"submitter":["young ik koh"],"technology_type":["Mass Spectrometry","Shotgun proteomics"],"software":["Not available"],"submitter_keywords":["Proteomics","Mutant protein","Interactors"],"full_dataset_link":["https://www.ebi.ac.uk/pride/archive/projects/PXD021514"],"tissue":["Kidney"],"sample_protocol":["Proteins (75 mg) extracted from HEK 293 cells stably expressing p3XFLAG-OSBPL2-WT and p3XFLAG-OSBPL2-mutant or -bacterial alkaline phosphatase (BAP) were incubated with 80 µL of FLAG M2 agarose beads (Sigma-Aldrich) for 48 h at 4°C in an orbital shaker. The agarose beads were washed four times with lysis buffer to restrict non-specific binding. Subsequently, 200 µL of elution buffer containing 150 ng/µL 3xFLAG peptide was added, and the samples were incubated overnight. The eluates were analyzed using immunoblotting, Coomassie blue staining, and silver staining. The eluates were digested and subjected to NanoLC-MS/MS analysis. Peptides were separated on a C18 precolumn (75 μm × 2 cm, nanoViper, Acclaim PepMap100, Thermo Fisher Scientific) and analytical C18 column (75 μm × 50 cm, PepMap RSLC, Thermo Fisher Scientific). Peptides were analyzed using an LC-MS/MS system consisting of an Easy nLC 1000 (Thermo Fisher Scientific) and an Orbitrap Fusion Lumos mass spectrometer (Thermo Fisher Scientific) equipped with a nano-electrospray source. MS/MS spectra were analyzed against the Uniprot human database using the following software analysis protocol. The reversed sequences of all proteins were appended into the database for the FDR calculation. ProLucid was used to identify the peptides, with a precursor mass error of 5 ppm and a fragment ion mass error of 200 ppm."],"repository":["Pride"],"quantification_method":["Not available"],"modification":[""],"data_protocol":["The interactors of WT and mutant OSBPL2 were analyzed using the Database for Annotation, Visualization and Integrated Discovery (DAVID) for functional annotation. The Functional Annotation Tool in the online version of DAVID (version 6.8) was run (http://david.abcc.ncifcrf.gov/) using the default parameters and GO categories representing molecular function, cellular component, and biological process were separately analyzed for enrichment. p-value < 0.05 was considered significant."],"omics_type":["Proteomics"],"labhead":["young ik koh"],"instrument_platform":[""],"labhead_affiliation":["Department of Pharmacology Yonsei University College of Medicine"],"submission_type":["COMPLETE"],"species":["Homo Sapiens (human)"],"submitter_mail":["rhduddlr91@yuhs.ac"],"publication":["35253614 Koh YI, Oh KS, Kim JA, Noh B, Choi HJ, Joo SY, Rim JH, Kim HY, Kim DY, Yu S, Kim DH, Lee SG, Jung J, Choi JY, Gee HY. <i>OSBPL2</i> mutations impair autophagy and lead to hearing loss, potentially remedied by rapamycin. Autophagy. 2022 18(11):2593-2614 10.1080/15548627.2022.2040891"],"submitter_affiliation":["Department of Pharmacology Yonsei University College of Medicine"],"submitter_country":["South Korea"],"doi":["10.6019/PXD021514"],"pubmed_abstract":["Intracellular accumulation of mutant proteins causes proteinopathies, which lack targeted therapies. Autosomal dominant hearing loss (DFNA67) is caused by frameshift mutations in <i>OSBPL2</i>. Here, we show that DFNA67 is a toxic proteinopathy. Mutant OSBPL2 accumulated intracellularly and bound to macroautophagy/autophagy proteins. Consequently, its accumulation led to defective endolysosomal homeostasis and impaired autophagy. Transgenic mice expressing mutant OSBPL2 exhibited hearing loss, but <i>osbpl2</i> knockout mice or transgenic mice expressing wild-type OSBPL2 did not. Rapamycin decreased the accumulation of mutant OSBPL2 and partially rescued hearing loss in mice. Rapamycin also partially improved hearing loss and tinnitus in individuals with DFNA67. Our findings indicate that dysfunctional autophagy is caused by mutant proteins in DFNA67; hence, we recommend rapamycin for DFNA67 treatment.<b>Abbreviations:</b> ABR: auditory brainstem response; ACTB: actin beta; CTSD: cathepsin D; dB: decibel; DFNA67: deafness non-syndromic autosomal dominant 67; DPOAE: distortion product otoacoustic emission; fs: frameshift; GFP: green fluorescent protein; HsQ53R-TG: human p.Q53Rfs*100-transgenic: HEK 293: human embryonic kidney 293; HFD: high-fat diet; KO: knockout; LAMP1: lysosomal associated membrane protein 1; MAP1LC3/LC3: microtubule-associated protein 1 light chain 3; MTOR: mechanistic target of rapamycin kinase; NSHL: non-syndromic hearing loss; OHC: outer hair cells; OSBPL2: oxysterol binding protein-like 2; SEM: scanning electron microscopy; SGN: spiral ganglion neuron; SQSTM1/p62: sequestosome 1; TEM: transmission electron microscopy; TG: transgenic; WES: whole-exome sequencing; YUHL: Yonsei University Hearing Loss; WT: wild-type."],"pubmed_title":["<i>OSBPL2</i> mutations impair autophagy and lead to hearing loss, potentially remedied by rapamycin."],"pubmed_authors":["Koh Young Ik YI, Oh Kyung Seok KS, Kim Jung Ah JA, Noh Byunghwa B, Choi Hye Ji HJ, Joo Sun Young SY, Rim John Hoon JH, Kim Hye-Youn HY, Kim Dong Yun DY, Yu Seyoung S, Kim Da Hye DH, Lee Sang-Guk SG, Jung Jinsei J, Choi Jae Young JY, Gee Heon Yung HY"],"pubmed_title_synonyms":["Transitory Deafnesses, 26E, Antibiotic AY 22989, plomo, ER-Phagy, 18R, 35-hexamethyl-11, AY 22 989, Blei, Pb, Lipophagy, plomb, Transitory Deafness, 82Pb, 30S, selective autophagy, Ribophagy, Transitory Hearing Losses, deafness, 10, ORP2, Orp2, Mutations, Deafness, (-)-Rapamycin, 14, 17, Transitory Hearing, Transitory Hearing Loss, 19R, Rapamycin, Reticulophagy, I2190A, 15R, Hearing loss, I-2190A, sirolimusum, Autophagocytosis, 18-dihydroxy-12-{(2S)-1-[(1S, Cellular Autophagy, Cellular, Hearing, 23S, 3, C130070J12Rik, 9S, 30-dimethoxy-15, Hearing Loss, 28E, 21, 16E, 23, Deafnesses, 24, 24E, 26, 29, rapamycin., 35R)-1, 4R)-4-hydroxy-3-methoxycyclohexyl]propan-2-yl}-19, ORP-2, Autophagy, I 2190A, AY 22-989, 9)]hexatriaconta-16, plumbum, 12S, 28-tetraene-2, 32S, sirolimus, Hypoacuses, mKIAA0772, Rapamune, Hypoacusis, Hearing Impairment, 20-pentone, Loss, (1R, ER Phagy, 36-dioxa-4-azatricyclo[30.3.1.0(4, Nucleophagy, lead, Transitory, 21R, AY 22989, 3R"],"description_synonyms":["ORP2, Orp2, Protein Gene Products, Mutations, Gene Proteins, native protein, ORP-2., ORP-2, Protein, proteomic analysis, Gene Products, Proteins, Gene, C130070J12Rik, proteins, protein, function, mKIAA0772"],"sample_synonyms":["293 cell, liquid chromatography tandem mass spectroscopy, Reverse, Immunoelectroblotting, determination, benzyladenine, Sepharose C1 4B, bap, protein, Orthophosphoric-monoester phosphohydrolase (alkaline optimum), peptide, Polypeptides, Electroimmunoblotting, peptido, B1, Beta-adaptin, HEK 293, Software Engineering, Human Embryonic Kidney 293, cg12532, N6-Benzyladenine, Computer Program, WMS, Sepharose, Divorced, LCMSMS, Man (Taxonomy), peptides, Immunoblottings, Open, Computer Programs and Programming, proteins, Divorces, N(6)-(benzylamino)purine, APase, C1 4B, column, ppm, D1, REA, autolysin activity, necrosis, GPHYSD2, Histological Labeling, SGS, 293 HEK, LC-MS2, 6-Benzylaminopurine, 4B, Modern, LC-MS/MS, Labeling and Staining, Source Softwares, Software Tools, Programs, ACMICD, Program, Computer Applications, DmelCG12532, [4)-3, Computer Applications Software, Sepharose C1, Computer Applications Softwares, p22, Silver, bacteriocin activity, Softwares, Reverse Immunoblottings, parent ion, Software Applications, 293, MS/MS, Source Software, AP1beta1, 6-BAP, Bcap27, MASS, 6-(benzylamino)purine, study protocol, human, organ system, Silver Nitrate, Applications, lysin activity, Labelings, precursor ion, Reverse Immunoblotting, liquid chromatography-tandem mass spectroscopy, beta1/2, Sepharose 4B, bacteriolytic toxin activity, BAD1, Bcap37, Polypeptide, BCAP37, Computer Software Applications, human being, Immunoblotting, Histological Labelings, Dot Immunoblotting, Nitrate Staining, FBN, Gene, Silver Nitrate Staining, Computer, precursor, body system, LC-MS-MS, ORP2, Orp2, Buffer, Human, method, Dot, Homo sapiens, ECTOL1, method used in an experiment, LC-MSMS, Gene Products, Histological, BiP-associated protein, system, AP2beta2, Separated, Man, Application, Open Source Softwares, anatomical systems, Staining, ORP-2, MS2, Software Application, ligand, Open Source Software, ppm., colony forming unit basophil, man, AP-beta1, Computer Software Application, OCTD, AP-1beta1, Tools, 10^[-6], MSS, Agarose, peptidos, Immunoelectroblottings, N-BENZYL-9H-PURIN-6-AMINE, 6-An-alpha-L-Galp-(1->3)-beta-D-Galp-(1->]n, PTHB1, Applications Software, CFU-Bas, ULG5, Open Source, 6-[(phenylmethyl)amino]-9H-purine, Computer Software, Proteins, Stainings, backward, buffer, CG12532, Labeling, Cell, Peptide, phoA, Tool, polypeptide, LC/MS/MS, Software Tool, basophilic stem cell, native protein, Dot Immunoblottings, C18, AU044498, Protein, chemical analysis, MFS1, C130070J12Rik, connected anatomical system, Software, tandem MS, Data Base, WMS2, Electroimmunoblottings, Separation, PNAS-141, holin, Separations, BaP, Engineering, HEK293, AP-2beta, HEK-293, mKIAA0772, Bap37, Protein Gene Products, plan specification, Computer Programs, Gene Proteins, Applications Softwares, Bap-37, Modern Man, SSKS, Bap, BAP, liquid chromatography tandem mass spectrometry, Peptid, assay, Cytokinin B, reversed"],"pubmed_abstract_synonyms":["LC3/Atg8, Lysophagy, DmelCG5178, Deaf Mutism, DmErk, SNDI, atg8, Microtubule-associated protein, Complete deafness (finding), A4, Visible Light, Prelingual, MAP1 Microtubule-Associated Protein, AI790512, Deafness NOS (finding), ref2p, C130039E17Rik, responsivity, OSBPL10 protein, HOW, 3, Whole Transcriptome, Transcriptome Sequencing, Kinase, RAFT1, 7, SEM, DmelCG6251, A, C, D, E, Fetal Hairs, kus, beta1tub, ganglion cochlearis, M, vibrissa, Tissue, Misfolding diseases, stru, T, Dsor2, l(3)S053606, inhibition of homeostatic process, CD, ORP10 protein, e, decreased, \"deafness\" EXACT [MTH:345], classical Hodgkin lymphoma, beta1Tub, RMD-1, Transitory, \"Bilateral deafness (finding)\" EXACT [SNOMEDCT_2005_07_31:162344009], YKR003W protein, 1110017I01Rik, Act, G-Actin, intracellular, nodular sclerosis classical Hodgkin lymphoma, CG4943, accumulated, rapamycin, ERK-A, Proteinopathy, screening, TAF60/62, Clicking Tinnitus, dpERK, lamina medullaris accessoria, PROTEIN KINASE PINOID, Actin/BAP47, UNQ391/PRO726, Protein Folding Disorder, ref, act, Hairs, DmMAPK, Light, dp-ERK, Clicking, partial functionality, TAFII60, Leudet's Tinnitus, TAFII62, AU042359, LIGHT, 1110001K06Rik, OSBP-related protein 8, pMAPK, Cellular, FLOWERING TIME CONTROL PROTEIN FCA ALPHA, Epithelial Microtubule-Associate Protein, Mutant, PINOID, 30-dimethoxy-15, Hearing Loss Permanent, Microtubule-Associated, OSBP-related protein 4, OSBP-related protein 1, Complete Transcriptome, DmelCG4027, 4R)-4-hydroxy-3-methoxycyclohexyl]propan-2-yl}-19, \"Deafness NOS\" EXACT [SNOMEDCT_2005_07_31:194432002], CHDS7, gamma-Actin, l(2)41Ac, Electron, DYN4, beta1-tub, Hodgkin lymphoma, act79B, Bilateral Deafness, Osbp protein, CT34260, light quantum, dpERk, folding diseases, DL4180C, Protein Misfolding disorders, Ft, Cells, Whole Exome Sequencing, Thrombospondin receptor, \"Complete deafness (finding)\" EXACT [SNOMEDCT_2005_07_31:8531006], Mouse, ORP1S protein, Specbeta, Proteostasis Dysfunction, nodular sclerosis Hodgkin lymphoma, Act5c, atypia, oxysterol binding protein, Spiral Ganglia, Proteaphagy, Spontaneous Oto Acoustic Emission, Nerve, 6330400K15Rik, mini-ICE, IBSN, Dm Fat, 62kDa, dTAF[[II]]262, B-spec, Gene Products, Diets, atg8a, Cellular Autophagy, Mus musculus domesticus, Scarb3, Vascular Origin, atypical, MAM, GP3B, Hodgkin's disease, alpha-Actin, Erk, Exome Sequencings, Bx34, 35R)-1, Tissues, Lichtquant, dysfunction, Ringing in ears, lamella pallidi incompleta, OSBPL1B protein, Protein folding, accessory medullary lamina of pallidum, Act57, tor, Tpr, BTF2 p62, TAF[[II]]60, Sequencing, erk, TAF[[II]]62, anon-EST:fe1B3, CG18290, quill, folding disease, T10, rll, dTAF62, T11, CG5870, Associated Protein, T17, l(2)06950, 42A, DmelCG3352, Induced Tinnitus, findings, ORP5 protein, Complete Exome Sequencings, MDB, DmelCG32672, disorders, MFT1, BcDNA.LD22582, Pulsatile, l(2)gd2, selective autophagy, defective, mTOR, Dsmurf, Transitory Hearing Losses, i168, OSBPL1A protein, CD258, l(3)S090417, Protein Misfolding Disorders, ME5, l(2)06949, Tinnitus of Vascular Origin, dSmurf, truncus encephali, Epithelial Microtubule Associate Protein, \"Deafness NOS (finding)\" EXACT [SNOMEDCT_2005_07_31:267678002], oxysterol binding protein-like 7 protein, native protein, OSBPL1 protein, PASIV, Rapamycin, I2190A, Smurf ubiquitin ligase, Macro Autophagy, Hearing, Act42, C130070J12Rik, PAS IV, Platelet collagen receptor, CG12559, Act88-F, HVEM-L, dpERK1, E-MAP-115, Truncus Cerebrus, b spectrin, House Mice, sirolimus, MFT, mus87E, microchaeta, Tub, Extreme, Photoradiations, neuronal cell, Folding Disease, OSBPL7 protein, F Actin, 21R, coat hair, CG10360, Bap47, l(2)79/18, OSH6 protein, Objective Tinnitus, oxysterol, setulae, FKBP12-rapamycin complex-associated protein, Mus domesticus, Map1lc3, protein, l(2)ft, l(2)k03905, l(1)G0420, SR2-1, OSIL, Noise Induced Tinnitus, IMP2, DSmurf, Protein folding disorder, OSBPL8 protein, Noise Induced, 23S, oxysterol binding protein-like 1B, protein aggregate, Microtubule Associated Protein 4, Microtubule Associated Protein 7, fat tissue, l(3)j5D5, Microtubule Associated Protein 1, Microtubule Associated Protein 2, Microtubule Associated Protein 3, 24B, rsd, CYLDI, 24E, N Actin, Whole Transcriptome Sequencing, membrane region, DERK-A, E(sina)7, Fatty acid translocase, Rl, CYLD1, Misfolding Disease, l(3)j5B5, Isoactin, TNFSF14, Microtubule Associated Protein, LGP120, Fetal, ER Phagy, Mpl3, disease management, CG4027, TG, dERK, Hodgkin's disease nodular sclerosis, DmelCG18290, Nerve Cell, IMP-2, dTOR, FK506-binding protein 12-rapamycin complex-associated protein 1, Sam68, D-smurf, D7Ertd649e, 26E, Membrane Tissue, taf60, Noise, BETA 56D, Auditory Ganglions, hearing impairment, BcDNA:LD22582, MAP1BLC3, Mapk, Ganglion, DmelCG10360, USPL2, 1010001C15Rik, mouse, OSBP protein, membranous organ component, CD36, CTSD, OSBPL5 protein, \"deafness\" EXACT [CSP2005:0977-5812], Electron Microscopy, Ref2P, ACTIN, Body Fat, p62B, Lamp-1, zbs559, OSBP2 protein, Reticulophagy, MapK, SZ1, oxysterol receptors, Autophagocytosis, brain stem, CG5178, C77871, Microtubule, Protein FAM82B, Hearing Loss, 28E, beta-tub, ERKa, Aggrephagy, Acquired, whisker, BcDNA:RE08694, 293, l(2R)EMS45-39, mice, DmelCG12559, FCAALL.331, Actin, beta56D, DmelCG9277, MICE, TAF[[II]]60/62, TAF[[II]], Treatments, domesticus, l(1)G0486, DpErk, l(1)G0245, disease, beta-Tub, ERKA, DMDA, l(2)fd, l(1)G0009, Faf, CG6251, 5092, DmelCG1548, A170, Fat, Microtubule-Associated Protein, ZIP, dTAF[[II]]60, Transitory Deafnesses, 1010001H21Rik, SCARB3, oxysterol binding protein 2, l(1)G0010, nerve cell, Chaperone-Assisted-Selective, P62, CHM1L, GPIIIB, electron, fat, oxysterol binding protein-like 9 protein, anon-EST:CL2c12, l(2)k17004, dTAF[[II]]62, Orp2, lamina medullaris incompleta pallidi, l(1)G0025, BcDNA:LD05816, MAP1 Microtubule Associated Protein, Deafness NOS (disorder), ATG8a, EAC, polypeptide chain, Homo sapiens, House, subnumerary, p60, betaspec, integral component of membrane, p62, dmTAF6, 18-dihydroxy-12-{(2S)-1-[(1S, actin-12, p68, UNQ771/PRO1565, ATG8E, DmelCG8151, Lanugo, reactivity, AU045898, betatub(56D), Fetal Hair, Swiss, \"Total Deafness\" EXACT [NCI2004_11_17:C36194], CATH, TAF60, AW743425, STONE14, Auditory, Deafness NOS, Transphosphorylase, nodular sclerosis of unspecified site, CG18732, CPSD, Protein Misfolding Disease, Extreme Hearing Loss, Loss, SPEC8, Disorder, E MAP 115, Glycoprotein IIIb, region of membrane, disorder, TAF, CT24817, SR3-4B, Autoregulation, Abbreviation, Hodgkin's nodular sclerosis, Ifm(3)7, Spec-beta, membrane, R74677, taf6, act 79B, protein complex, clone 2.39, ABRUPTUS, Tubulin, Pulsatile Tinnitus, AW822074, DmelCG5092, BEST:RE38067, Misfolding disorders, Hodgkin's lymphoma, Brain Stems, 2.7.11.1, MAP3 Microtubule-Associated Protein, TFIID 62, Platelet glycoprotein IV, ganglion spirale, \"Deafness NOS\" EXACT [MTHICD9_2006:389.9], nodular sclerosis NOS (morphologic abnormality), whole membrane, MAP2 Microtubule Associated Protein, LGMD2C., b-Spec, truncus encephalicus, AA589433, CDHR8, who, MAP3 Microtubule Associated Protein, Radiations, l(1)G0079, l(1)G0074, Permanent, transmembrane, Leukocyte differentiation antigen CD36, Complete Transcriptome Sequencing, negatron, Kinases, Su(Raf)2B, act 57A, HEK293, 28-tetraene-2, DrAtg8a, CG9348, AI327068, MAP2 Microtubule-Associated Protein, oxysterol-binding protein 2, Membrane, Tinnitus, Laboratory Mice, Protein Gene Products, NOS\" EXACT [SNOMEDCT_2005_07_31:15188001], activation of homeostatic process, D-ERK, Therapeutic, DmelCG1945, CG8274, SCARMD2, qkr[93F], Treatment, GPIV, TAF6, Spec, Actin5C, TEM, Transmission Electron Microscopy, 293 cell, DmelCG7478, BODYFAT, 2610315D21Rik, nucleocytoplasm, CT11259, ER-Phagy, Laboratory, \"Bilateral Deafness\" EXACT [NCI2004_11_17:C36193], Diffraction Microscopy, Microtubule Associated, sci, House Mouse, DmTOR, Hair, Mutations, DmelCG12051, protein polypeptide chains, Misfolding disorder, lamina pallidi incompleta, hnu, symptoms, How, T31E10.1, HEK 293, Human Embryonic Kidney 293, Mammalian target of rapamycin, Sem, Acquired Deafness, treatment, MAP-k, Leudet Tinnitus, cyt5C, Autophagies, CG9277, Bricd4, folding disorder, ABR, Tensor Palatini Induced, beta-particle, Epithelial MAP, proteins, VICKZ2, OSBPL6 protein, beta Actin, CG1548, act88F, sem, Atg8, BTF2, 20-pentone, e-, (1R, N-Actin, GP4, Smurf, Protein Folding Diseases, Nucleophagy, ATP Phosphotransferases, act87E, Acronym, Proteinopathies, oxysterol binding protein-like 11 protein, Radiation, dpErk, l(2)gd-1, OSF-6, Electron Diffraction, Protein folding disorders, alpha Isoactin, Exome, Vascular Origin Tinnitus, integral to membrane, Taf[[II]]60, Lack, Ribophagy, act42A, CG32672, AFFX-Dros-ACTIN_M_r_at, pMapK, actin, \"complete hearing loss\" EXACT [], University, Dysfunction, Misfolding Disorder, l(2)k07918, Act57A, Tensor Tympani Induced, Transmission Electron, xabr, DmERKA, Nerve Cells, HVEML, rl/MAPK, Caspase-14 subunit p10, Auditory Ganglion, Autophagy, ORP4 protein, CathD, ganglion spirale cochleae, Rapamycin and FKBP12 target 1, Swiss Mouse, l(3)76BDf, signs, l(2)gd-l, NSCHL, Cerebri, Caspase-14 subunit p19, Hypoacusis, Act57b, human, beta(-), DTB2, actin5C, Spiral, Subjective, i6, spiral ganglion, beta-Tub56D, CT3996, PDB3, 3.4.22.-, oxysterol binding protein like 5 protein, Ref(2)P, e(-), 12559, ACT5C, Lipophagy, impaired, Transitory Deafness, Tensor Tympani Induced Tinnitus, protein-containing complex, BDPLT10, hFat1, Total Deafness, 4930547K17Rik, LRRGT00047, EK2-1, Human, Ly113, CG30294, LAMPA, CDHF7, Transitory Hearing Loss, nodular sclerosis Hodgkin's lymphoma, Ref(2)p, PAS-4, CG3352, MAP1ALC3, TOR, TAFII60(62), Man, Acronyms, ERK, Spontaneous Oto Acoustic Emission Tinnitus, Deafnesses, Xenophagy, Complete, Transmission, ORP-2, BRWS1, Elektron, Protein Misfolding diseases, Proteostasis Deficiency, mus79B, beta1t, nodular sclerosis Hodgkin's disease, TPR, man, nodular sclerosis Classic Hodgkin lymphoma, CG32211, anon-EST:fe2D2, oxysterol binding protein-like 6 protein, S cerevisiae, Whole Exome, oxysterol-binding protein-related protein 8, oxysterol-binding protein-related protein 4, Whole, Protein Folding, negative regulation of homeostatic process, photon, nodular sclerosis, Folding Disorder, bodyfat, Neuron, TR2, AY 22989, ZIP3, Frap1, Act-5C, CPH, beta-tubulin56D, fur, Dmbeta1, 4922501H04Rik, Ringing-Buzzing-Tinnitus, Electron Diffraction Microscopy, Proteins, Truncus, Protein 7, 30S, FRAP1, FRAP2, qkr, deafness, Deaf-Mutism, LC3, Cell, Exome Sequencing, NSHD, Transitory Hearing, Tensor Palatini Induced Tinnitus, Cerebrus, Chaperone-Assisted-Selective Autophagy, beta-actin, NSHL, KH93F, sirolimusum, M32055, Complete Hearing Loss, regulation of homeostatic process, DmelCG5870, G Actin, DMDA1, beta[[1]]-tubulin, ORP1L protein, I 2190A, 32S, beta1, Nupc1, CDMT, HEK-293, mKIAA0772, FRAP, beta, Microtubule-Associated Protein 3, Gene Proteins, having decreased function, Microtubule-Associated Protein 7, Deficiency, CT16317, dpMAPK, adipose, OSBPL11 protein, Leudets, Microtubule-Associated Protein 1, MTOR, BETA, Microtubule-Associated Protein 2, l(2)fat, receptor, Corti's ganglion, BAP47, act 87E, LC3A, TFB1, Brainstem, Protein folding disease, Dmp62, Protein Folding Disease, DELTA AND GAMMA, beta-Actin, Protein Misfolding Disorder, CASP-14, betaTub1, folding disorders, Act5, 10, d60, BRICD4, Deafness, Truncus Cerebri, dysfunctional, 14, Membrane Tissues, T31E10_1, B1t, 17, act 88F, mRMD-1, diseases, oxysterol binding protein-like 5 protein, gamma Actin, Hearing loss, Ganglion of Corti, Taf-6, Prelingual Deafness, Cph1, CLN10, fatty tissue, present in fewer numbers in organism, foton, 21, Atg8/LC3, 23, 24, 26, Erk/Map kinase, 29, Man (Taxonomy), adipose system, DmelCG32211, Complete Exome Sequencing, 12S, Microtubule Associated Proteins, MAP3, Swiss Mice, MAP2, MAP1, Actx, MAP4, CG10293, TfBl, dtor, 1t, LC3b, DERK, LC3a, Protein Misfolding Diseases, act 42A, Ac5C, Therapies, homeostasis, STAP, associated, Misfolding disease, Deafness Permanent, dTor, ATP, gamma, 3R, 293 HEK, Therapy, Mechanistic target of rapamycin, 0904/17, l(1)G0330, aberrant, Modern, Erk1, PS1TP5BP1, Protein Misfolding, (-)-Rapamycin, ref(2)Po2, l(1)G0108, positive regulation of homeostatic process, MAPK, Mini-ICE, 15R, BRSS, CG7478, internal to cell, spiral ganglion of cochlea, Proteostasis Dysfunctions, Ringing Buzzing Tinnitus, Mutant Protein, CG10067, RAPT1, oxysterol binding protein-related protein 1, mapk, Ensconsin, 16E, Visible Radiations, Mus musculus, Proteostasis, Transphosphorylases, Visible Radiation, membrane of organ, Beta <eudicots>, DmelCG4943, Protein Misfolding disorder, l(1)G0117, Bilateral deafness (finding), lacks function of type, beta-actin/Bap47, oxysterol binding protein-like 8 protein, low functionality, DpERK, ErkA, Osi, \"Deafness, 36-dioxa-4-azatricyclo[30.3.1.0(4, Ganglia, anon-EST:Liang-2.39, Microscopy, FAT, Transcriptome Sequencings, betaTub, pERK, beta1-Tubulin, complete hearing loss, act57b, HEL-S-130P, Antibiotic AY 22989, Leudet's, Act87e, human being, GroupII, 18R, Complete Exome, 35-hexamethyl-11, dSmurf1, CG5092, Gene, CG12051, 115 kDa, Protein Folding Disorders, TYPE, ORP2, act57B, DAGA4, act57A, Pexophagy, Act88f, reduced, 19R, Ringing in the ears, Taf62, Mice, SCG3, 9S, oxysterol binding protein-like 10 protein, Phosphotransferase, 143060_f_at, Taf60, DmelCG8274, protoplasm, OXYB protein, ChM1L, WES, act5C, DmelCG10067, l(3)s2612, protoplast, AY 22-989, coat/ hair, 9)]hexatriaconta-16, beta[[1]] tubulin, F-Actin, CatD, 79/18, flat, Visible, Hypoacuses, decreased number, CG1945, \"Deafness NOS\" EXACT [SNOMEDCT_2005_07_31:155259007], Act42a, CD107a, d-smurf, auditory ganglion, DmelCG10293, DmERK-A, OSBPL9 protein, alpha Actin, ref(2)Pn, l(1)G0177, Disease, Macro-Autophagy, Protein Misfolding disease, Protein folding diseases, Spontaneous Oto-Acoustic Emission Tinnitus, AY 22 989, CT39192, ref(2)p, FRAP/TOR, dTAFII62, brainstem, Phosphotransferases, LGMD2C, dme-TAFII60, Permanents, vestibulocochlear ganglion cochlear component, AI196048, membrane tetraspanin-enriched microdomain, Ganglions, natural protein, Mus, Protein, I-2190A, Objective, betaSpec, TFIID, TFIIH, Atg8A, Cph-1, CG1534, E430023M04Rik, l(1)G0198, AI426861, Dietary, DmelCG12042, Photoradiation, Subjective Tinnitus, act 5C, hair, oxysterol receptor, CT24745, oxysterol binding protein-like 1A protein, Rapamycin target protein 1, EY2-2, Anx3, ORP8 protein, Who/How, Rapamune, Spontaneous Oto-Acoustic Emission, Brainstems, Hearing Impairment, TeM, LTg, alpha-Isoactin, Leudet, SBS, fatty depot, dTAF6, beta-spec, Act(88F), Modern Man, CG8151, Chaperone Assisted Selective Autophagy, l(2)24Da, response, Laboratory Mouse"],"name_synonyms":["ORP2, Orp2, C130070J12Rik, mKIAA0772, ORP-2."],"data_synonyms":["visualization, molecular function unknown, CG1849, RAD3D, l(1)LB9, l(3)05592, single-organism process, Process, D2-2, Processes, leg, Dm0688, gov, DmelCG1849, GS-X, bHLHb28, single organism process, Rnt, Emc, AA33, CG1007, ORP2, Orp2, lLB5, l(1)AA33, Biological Processes, Biological Phenomenon, DmelCG1007, Biological, Phenomena, 1, 2, cellular component unknown, C130070J12Rik, subcellular entity, Biological Process, Phenomenon, biological process, ABC29, Data Base, 0977/09, l(3)04322, Biologic Phenomena, MRP, ORP-2, Runt, 0203/10, ms(3)61CD, l(1)19Ea, cellular component, l(3)j4E11, mKIAA0772, ABCC, Biological., P235, MRP1, 0587/01, Ach, l(1)B2/13.1, RUN, Run, physiological process, GOV, l(3)61Da, 0094/26, molecular function, RAB16, cell or subcellular entity, Biologic, LB5"],"additional_accession":[]},"is_claimable":false,"name":"Identification of WT and mutant OSBPL2 interactors","description":"Pathogenic OSBPL2 mutations led to almost identical truncated proteins, which formed cytoplasmic aggregates, we performed a proteomic analysis to understand the function of mutant OSBPL2 via the identification of its interactome.","dates":{"publication":"2023-03-10","submission":"2020-09-16"},"accession":"PXD021514","cross_references":{"TAXONOMY":["NEWT:330879","NEWT:377960","NEWT:2042546","NEWT:259447","NEWT:295546","NCBITaxon:1280","NEWT:112503","NEWT:1129","NEWT:309807","NEWT:59753","NEWT:89184","NEWT:309800","NEWT:281395","NEWT:1211601","NEWT:876138","NEWT:44271","NEWT:193516","NEWT:1073882","NEWT:6819","NEWT:475174","NEWT:1117","NEWT:498257","NEWT:10036","NEWT:1590","NEWT:10034","NEWT:661410","NEWT:638632","NEWT:224326","NEWT:376619","NCBITaxon:79857","NEWT:1096976","NEWT:3765","NEWT:1589","NEWT:135622","NEWT:35786","NEWT:1580","NEWT:399784","NEWT:96731","NEWT:656064","NEWT:36630","NEWT:383379","NCBITaxon:12721","NEWT:10029","NEWT:913645","NEWT:641809","NEWT:1392488","NEWT:38815","NEWT:317447","NEWT:4688","NEWT:7719","NEWT:868565","NEWT:135674","NEWT:79329","NEWT:30069","NEWT:12637","NEWT:59729","NEWT:2164133","NEWT:295105","NEWT:108061","NEWT:60711","NEWT:224308","NEWT:3347","NEWT:160621","NEWT:212790","NEWT:110368","NEWT:1310161","NEWT:77133","NEWT:145481","NEWT:1310165","NEWT:29058","NCBITaxon:79824","NEWT:410658","NEWT:44688","NEWT:44689","NEWT:498211","NEWT:347256","NEWT:5518","NEWT:398007","NEWT:1527468","NEWT:498217","NEWT:498216","NEWT:11320","NEWT:246196","NEWT:246197","NEWT:145458","NEWT:44685","NEWT:161934","NEWT:1148","NEWT:5508","NEWT:3329","NEWT:419481","NEWT:5507","NEWT:410661","NEWT:1140","NCBITaxon:2157","NEWT:1143","NEWT:1287689","NEWT:1094343","NEWT:1462472","NEWT:1336795","NEWT:644042","NEWT:1182590","NEWT:3712","NEWT:3711","NEWT:3714","NEWT:2065263","NEWT:177437","NEWT:10418","NEWT:118698","NEWT:1616117","NEWT:118696","NEWT:34865","NEWT:52283","NEWT:284812","NEWT:8175","NEWT:43330","NEWT:1603293","NEWT:44664","NEWT:43346","NEWT:3702","NEWT:1245466","NEWT:3704","NEWT:244366","NEWT:1246791","NEWT:118694","NEWT:2850","NEWT:118691","NEWT:34871","NEWT:33548","NEWT:52299","NEWT:8165","NEWT:96794","NEWT:3708","NEWT:332648","NEWT:44670","NEWT:536231","NEWT:376219","NEWT:219813","NEWT:1510","NEWT:460519","NEWT:1515","NEWT:572307","NEWT:1432138","NEWT:1424507","NEWT:1194599","NEWT:272844","NEWT:1348799","NEWT:33995","NEWT:55784","NEWT:1303443","NEWT:485","NEWT:56636","NEWT:2853422","NEWT:1679718","NEWT:480","NEWT:67767","NEWT:46835","NEWT:109757","NEWT:582580","NEWT:1185650","NEWT:216778","NEWT:1502","NEWT:128017","NEWT:376686","NEWT:237610","NEWT:95486","NEWT:1883446","NEWT:1233435","NEWT:118223","NEWT:109760","NEWT:29031","NEWT:317047","NEWT:491","NEWT:460523","NEWT:235443","NEWT:1757312","NEWT:85694","NEWT:109786","NEWT:108458","NEWT:5936","NEWT:320637","NEWT:1078283","NEWT:3750","NEWT:983964","NEWT:11706","NEWT:32644","NEWT:527796","NEWT:499175","NEWT:109779","NEWT:3745","NEWT:1715989","NCBITaxon:4751","NEWT:3747","NEWT:1116234","NEWT:1255228","NEWT:410289","NEWT:373153","NEWT:1640278","NEWT:472","NEWT:940825","NEWT:529507","NEWT:1071661","NEWT:470","NEWT:5911","NEWT:748693","NCBITaxon:50557","NEWT:39251","NEWT:29491","NEWT:101841","NEWT:446","NEWT:153481","NEWT:2014887","NEWT:33952","NEWT:445","NEWT:153009","NEWT:261756","NEWT:166313","NEWT:63366","NEWT:63367","NEWT:215402","NEWT:1547","NEWT:906914","NEWT:9031","NEWT:27292","NEWT:1050722","NEWT:108931","NEWT:1293497","NEWT:1055524","NEWT:150475","NEWT:267872","NEWT:381666","NEWT:172269","NEWT:9534","NEWT:5180","NEWT:256737","NEWT:9541","NEWT:8694","NEWT:33936","NEWT:8692","NEWT:2903","NEWT:185579","NEWT:13076","NEWT:207340","NEWT:1006581","NEWT:33940","NEWT:550","NEWT:554","NEWT:451516","NEWT:1325291","NEWT:36185","NEWT:1054211","NEWT:1225786","NEWT:771870","NEWT:51338","NEWT:575412","NEWT:28112","NEWT:6493","NEWT:10832","NEWT:6494","NEWT:6491","NEWT:507601","NEWT:520","NEWT:186441","NEWT:643680","NEWT:214092","NCBITaxon:6157","NEWT:13095","NEWT:162425","NEWT:104105","NEWT:216257","NEWT:9986","NEWT:8654","NEWT:8658","NEWT:1268063","NEWT:8657","NEWT:9983","NEWT:8655","NEWT:5147","NEWT:28108","NEWT:28104","NEWT:407821","NEWT:568703","NCBITaxon:2","NEWT:568708","NEWT:986","NEWT:52641","NEWT:28532","NEWT:353152","NEWT:298176","NEWT:227377","NEWT:1774284","NEWT:40674","NEWT:1194669","NEWT:443906","NEWT:519","NEWT:2510939","NEWT:7394","NEWT:6063","NEWT:1328388","NEWT:105487","NEWT:1548728","NEWT:667127","NEWT:9557","NEWT:377586","NEWT:300641","NEWT:39655","NEWT:9554","NEWT:38323","NEWT:475932","NEWT:256318","NEWT:206411","NCBITaxon:6191","NEWT:229533","NEWT:214053","NEWT:28550","NEWT:80863","NEWT:90675","NEWT:52639","NEWT:52638","NEWT:57075","NEWT:1666905","NEWT:8697","NEWT:8695","NEWT:884204","NEWT:1045775","NEWT:1123869","NEWT:9544","NEWT:9545","NEWT:979","NEWT:7370","NEWT:83906","NEWT:99488","NEWT:1134506","NEWT:255470","NEWT:38783","NEWT:6426","NEWT:33090","NEWT:9935","NEWT:287889","NEWT:305959","NEWT:1028307","NEWT:13489","NEWT:29829","NEWT:92867","NEWT:92866","NCBITaxon:3055","NEWT:51750","NEWT:202950","NEWT:441772","NEWT:48106","NEWT:202956","NEWT:295027","NCBITaxon:11320","NEWT:632957","NEWT:9925","NCBITaxon:9606","NEWT:90690","NEWT:1436183","NEWT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