biostudies-literatureBowl MRMedical Research CouncilNHGRI NIH HHSWellcome TrustNIH HHSBiotechnology and Biological Sciences Research Council886https://www.ebi.ac.uk/biostudies/studies/S-EPMC5638796biostudies-literatureUnknown8(1)The developmental and physiological complexity of the auditory system is likely reflected in the underlying set of genes involved in auditory function. In humans, over 150 non-syndromic loci have been identified, and there are more than 400 human genetic syndromes with a hearing loss component. Over 100 non-syndromic hearing loss genes have been identified in mouse and human, but we remain ignorant of the full extent of the genetic landscape involved in auditory dysfunction. As part of the International Mouse Phenotyping Consortium, we undertook a hearing loss screen in a cohort of 3006 mouse knockout strains. In total, we identify 67 candidate hearing loss genes. We detect known hearing loss genes, but the vast majority, 52, of the candidate genes were novel. Our analysis reveals a large and unexplored genetic landscape involved with auditory function.The full extent of the genetic basis for hearing impairment is unknown. Here, as part of the International Mouse Phenotyping Consortium, the authors perform a hearing loss screen in 3006 mouse knockout strains and identify 52 new candidate genes for genetic hearing loss.Nature communicationsA large scale hearing loss screen reveals an extensive unexplored genetic landscape for auditory dysfunction.PMC5638796U54 HG006370U54 HG006332U54 HG006364MC_QA137918MR/N012119/1BB/M02069X/1MC_U142684175G0300212UM1 HG006370U42 OD011174U42 OD011185U42 OD011175U54 HG006348U42 OD012210UM1 HG006348MC_UP_1502/1MC_UP_1502/3MC_U142684171UM1 OD023222098051UM1 OD0232211006991774734Rossi GMatteoni RSteinkamp RTreise IWakana SLe Marchand ERiet FTaylor SRossi NBeaudet ALSeisenberger CCodner GGambadoro ACater HPenton PLengger CFruscoloni PGardiner WSchmidt-Weber CMittelhaeuser CLa Sala GYamada IMcKerlie CRaspa MInternational Mouse Phenotyping ConsortiumLux AGarrett LLaurin VAndre PCreighton ABeckers JSelloum MZhu YWhite JMoreth KAyabe SSantos LSeavitt JAdams DJFlicek PZimprich ASorg TMiller DGreenaway SBradley APereira MReilly PGuimond AEskandarian MPearson SStoeger CZaman RKurbatova NWells SOllert MGailus-Durner VStoeger TAli-Hadji DMassimi MErmakova OJustice MJBusch DHJacob EChen CKTocchini-Valentini GDGolini EParker ADanisment OSvenson KLFray MSuzuki TBubshait MDi Segni GBeig JChiani FKenyon JGastaldi SWolf ETamura MGertsenstein MBekeredjian RBower LRLoeffler JOstereicher MABurger ATocchini-Valentini GPEickelberg OKuhn RObata YPasquini MKarp NPutti SLeblanc SBam'Hamed CIngham NJHerault YNutter LMJMarazziti DMarschall SFeugas PFlenniken AMTudose IMoore MMaier HZimmer AMallon AMBrown SDMBowl MRClark GSkarnes WCEl Amri AQuwailid DGuo RStewart MMorikawa LAguilar-Pimentel AHunter JOwen CNell BWalling AMatthews PCabezas JEnnah HYildrim AOConte NJoynson RMurray SAKaneda HLan QRozman JWattenhofer-Donze MMoulaert DMiura IMason JMinowa OWest DBQu DKlingenspor MTanaka NVasseur LEl Fertak LFerrara FNewbigging SBirling MCOritz OBrommage RLaw NJacquot SOrsini TBraun REAmarie OKelsey LAllen SFuruse TMandillo SSohel KMeehan TFPavlovic GClarke SBour RKobayashi Kde Angelis MHSimon MMSteel KPAyadi ALalanne VShang XMasuya HWang YBerberovic ZVukobradovic IMeziane HClementson-Mobbs SHolter SMRauch ATondat SGiesert FSleep GTeboul LMGao XCarroll TKlopstock TMacMaster SBecker LLloyd KCKDickinson MERathkolb BSmedley DCalzada-Wack JNeff FDi Pietro CClary DAWurst WParkinson HELeuchtenberger SYoshiki AGraw JFuchs HSchick JWarren JRelac MScavizzi FfalseA large scale hearing loss screen reveals an extensive unexplored genetic landscape for auditory dysfunction.The developmental and physiological complexity of the auditory system is likely reflected in the underlying set of genes involved in auditory function. In humans, over 150 non-syndromic loci have been identified, and there are more than 400 human genetic syndromes with a hearing loss component. Over 100 non-syndromic hearing loss genes have been identified in mouse and human, but we remain ignorant of the full extent of the genetic landscape involved in auditory dysfunction. As part of the International Mouse Phenotyping Consortium, we undertook a hearing loss screen in a cohort of 3006 mouse knockout strains. In total, we identify 67 candidate hearing loss genes. We detect known hearing loss genes, but the vast majority, 52, of the candidate genes were novel. Our analysis reveals a large and unexplored genetic landscape involved with auditory function.The full extent of the genetic basis for hearing impairment is unknown. Here, as part of the International Mouse Phenotyping Consortium, the authors perform a hearing loss screen in 3006 mouse knockout strains and identify 52 new candidate genes for genetic hearing loss.2017-01-01T00:00:00Z2017 Oct2021-03-18T09:08:27Z2019-03-27T02:58:49ZS-EPMC56387962902608910.1038/s41467-017-00595-4