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Biallelic variants in KARS1are associated with neurodevelopmental disorders and hearing loss recapitulated by the knockout zebrafish

Authors :
Lin, Sheng-Jia
Vona, Barbara
Barbalho, Patricia G.
Kaiyrzhanov, Rauan
Maroofian, Reza
Petree, Cassidy
Severino, Mariasavina
Stanley, Valentina
Varshney, Pratishtha
Bahena, Paulina
Alzahrani, Fatema
Alhashem, Amal
Pagnamenta, Alistair T.
Aubertin, Gudrun
Estrada-Veras, Juvianee I.
Hernández, Héctor Adrián Díaz
Mazaheri, Neda
Oza, Andrea
Thies, Jenny
Renaud, Deborah L.
Dugad, Sanmati
McEvoy, Jennifer
Sultan, Tipu
Pais, Lynn S.
Tabarki, Brahim
Villalobos-Ramirez, Daniel
Rad, Aboulfazl
Ambrose, J.C.
Arumugam, P.
Bleda, M.
Boardman-Pretty, F.
Boustred, C.R.
Brittain, H.
Caulfield, M.J.
Chan, G.C.
Fowler, T.
Giess, A.
Hamblin, A.
Henderson, S.
Hubbard, T.J.P.
Jackson, R.
Jones, L.J.
Kasperaviciute, D.
Kayikci, M.
Kousathanas, A.
Lahnstein, L.
Leigh, S.E.A.
Leong, I.U.S.
Lopez, F.J.
Maleady-Crowe, F.
Moutsianas, L.
Mueller, M.
Murugaesu, N.
Need, A.C.
O‘Donovan, P.
Odhams, C.A.
Patch, C.
Perez-Gil, D.
Pereira, M.B.
Pullinger, J.
Rahim, T.
Rendon, A.
Rogers, T.
Savage, K.
Sawant, K.
Scott, R.H.
Siddiq, A.
Sieghart, A.
Smith, S.C.
Sosinsky, A.
Stuckey, A.
Tanguy, M.
Thomas, E.R.A.
Thompson, S.R.
Tucci, A.
Walsh, E.
Welland, M.J.
Williams, E.
Witkowska, K.
Wood, S.M.
Galehdari, Hamid
Ashrafzadeh, Farah
Sahebzamani, Afsaneh
Saeidi, Kolsoum
Torti, Erin
Elloumi, Houda Z.
Mora, Sara
Palculict, Timothy B.
Yang, Hui
Wren, Jonathan D.
Fowler, Ben
Joshi, Manali
Behra, Martine
Burgess, Shawn M.
Nath, Swapan K.
Hanna, Michael G.
Kenna, Margaret
Merritt, J. Lawrence
Houlden, Henry
Karimiani, Ehsan Ghayoor
Zaki, Maha S.
Haaf, Thomas
Alkuraya, Fowzan S.
Gleeson, Joseph G.
Varshney, Gaurav K.
Source :
Genetics in Medicine; October 2021, Vol. 23 Issue: 10 p1933-1943, 11p
Publication Year :
2021

Abstract

Pathogenic variants in Lysyl-tRNA synthetase 1 (KARS1) have increasingly been recognized as a cause of early-onset complex neurological phenotypes. To advance the timely diagnosis of KARS1-related disorders, we sought to delineate its phenotype and generate a disease model to understand its function in vivo.

Details

Language :
English
ISSN :
10983600 and 15300366
Volume :
23
Issue :
10
Database :
Supplemental Index
Journal :
Genetics in Medicine
Publication Type :
Periodical
Accession number :
ejs63385546
Full Text :
https://doi.org/10.1038/s41436-021-01239-1