A large scale hearing loss screen reveals an extensive unexplored genetic landscape for auditory dysfunction
Authors: Bowl Michael R. , Simon Michelle M. , Ingham Neil J. , Greenaway Simon , Santos Luis , Cater Heather , Taylor Sarah , Mason Jeremy , Kurbatova Natalja , Pearson Selina , Bower Lynette R. , Clary Dave A. , Meziane Hamid , Reilly Patrick , Minowa Osamu , Kelsey Lois ,   The International Mouse Phenotyping Consortium, Allen Sue , Clementson-Mobbs Sharon , Codner Gemma , Fray Martin , Gardiner Wendy , Joynson Russell , Kenyon Janet , Loeffler Jorik , Nell Barbara , Parker Andrew , Quwailid Deen , Stewart Michelle , Walling Alison , Zaman Rumana , Chen Chao-Kung , Conte Nathalie , Matthews Peter , Relac Mike , Tudose Ilinca , Warren Jonathan , Le Marchand Elise , El Amri Amal , El Fertak Leila , Ennah Hamid , Ali-Hadji Dalila , Ayadi Abdel , Wattenhofer-Donze Marie , Moulaert David , Jacquot Sylvie , André Philippe , Birling Marie-Christine , Pavlovic Guillaume , Lalanne Valérie , Lux Aline , Riet Fabrice , Mittelhaeuser Christophe , Bour Raphael , Guimond Alain , Bam’Hamed Chaouki , Leblanc Sophie , Vasseur Laurent , Selloum Mohammed , Sorg Tania , Ayabe Shinya , Furuse Tamio , Kaneda Hideki , Kobayashi Kimio , Masuya Hiroshi , Miura Ikuo , Obata Yuichi , Suzuki Tomohiro , Tamura Masaru , Tanaka Nobuhiko , Yamada Ikuko , Yoshiki Atsushi , Berberovic Zorana , Bubshait Mohammed , Cabezas Jorge , Carroll Tracy , Clark Greg , Clarke Shannon , Creighton Amie , Danisment Ozge , Eskandarian Mohammad , Feugas Patricia , Gertsenstein Marina , Guo Ruolin , Hunter Jane , Jacob Elsa , Lan Qing , Laurin Valerie , Law Napoleon , MacMaster Sue , Miller David , Morikawa Lily , Newbigging Susan , Owen Celeste , Penton Patricia , Pereira Monica , Qu Dawei , Shang Xueyuan , Sleep Gillian , Sohel Khondoker , Tondat Sandra , Wang Yanchun , Vukobradovic Igor , Zhu Yingchun , Chiani Francesco , Di Pietro Chiara , Di Segni Gianfranco , Ermakova Olga , Ferrara Filomena , Fruscoloni Paolo , Gambadoro Aalessia , Gastaldi Serena , Golini Elisabetta , La Sala Gina , Mandillo Silvia , Marazziti Daniela , Massimi Marzia , Matteoni Rafaele , Orsini Tiziana , Pasquini Miriam , Raspa Marcello , Rauch Aline , Rossi Gianfranco , Rossi Nicoletta , Putti Sabrina , Scavizzi Ferdinando , Tocchini-Valentini Giuseppe D. , Beig Joachim , Bürger Antje , Giesert Florian , Graw Jochen , Kühn Ralf , Oritz Oskar , Schick Joel , Seisenberger Claudia , Amarie Oana , Garrett Lillian , Hölter Sabine M. , Zimprich Annemarie , Aguilar-Pimentel Antonio , Beckers Johannes , Brommage Robert , Calzada-Wack Julia , Fuchs Helmut , Gailus-Durner Valérie , Lengger Christoph , Leuchtenberger Stefanie , Maier Holger , Marschall Susan , Moreth Kristin , Neff Frauke , Östereicher Manuela A. , Rozman Jan , Steinkamp Ralph , Stoeger Claudia , Treise Irina , Stoeger Tobias , Yildrim Ali Önder , Eickelberg Oliver , Becker Lore , Klopstock Thomas , Ollert Markus , Busch Dirk H. , Schmidt-Weber Carsten , Bekeredjian Raffi , Zimmer Andreas , Rathkolb Birgit , Wolf Eckhard , Klingenspor Martin , Tocchini-Valentini Glauco P. , Gao Xiang , Bradley Allan , Skarnes William C. , Moore Mark , Beaudet Arthur L. , Justice Monica J. , Seavitt John , Dickinson Mary E. , Wurst Wolfgang , de Angelis Martin Hrabe , Herault Yann , Wakana Shigeharu , Nutter Lauryl M. J. , Flenniken Ann M. , McKerlie Colin , Murray Stephen A. , Svenson Karen L. , Braun Robert E. , West David B. , Lloyd K. C. Kent , Adams David J. , White Jacqui , Karp Natasha , Flicek Paul , Smedley Damian , Meehan Terrence F. , Parkinson Helen E. , Teboul Lydia M. , Wells Sara , Steel Karen P. , Mallon Ann-Marie , Brown Steve D. M. 
Published: October 2017

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.