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Sauvageau, M.* ; Goff, L.A.* ; Lodato, S.* ; Bonev, B. ; Groff, A.F.* ; Gerhardinger, C.* ; Sanchez-Gomez, D.B.* ; Hacisuleyman, E.* ; Li, E.* ; Spence, M.* ; Liapis, S.C.* ; Mallard, W.* ; Morse, M.* ; Swerdel, M.R.* ; D'Ecclessis, M.F.* ; Moore, J.C.* ; Lai, V.* ; Gong, G.* ; Yancopoulos, G.D.* ; Frendewey, D.* ; Kellis, M.* ; Hart, R.P.* ; Valenzuela, D.M.* ; Arlotta, P.* ; Rinn, J.L.*

Multiple knockout mouse models reveal lincRNAs are required for life and brain development.

eLife 2:e01749 (2013)
DOI PMC
Creative Commons Lizenzvertrag
Open Access Gold as soon as Publ. Version/Full Text is submitted to ZB.
Many studies are uncovering functional roles for long noncoding RNAs (lncRNAs), yet few have been tested for in vivo relevance through genetic ablation in animal models. To investigate the functional relevance of lncRNAs in various physiological conditions, we have developed a collection of 18 lncRNA knockout strains in which the locus is maintained transcriptionally active. Initial characterization revealed peri- and postnatal lethal phenotypes in three mutant strains (Fendrr, Peril, and Mdgt), the latter two exhibiting incomplete penetrance and growth defects in survivors. We also report growth defects for two additional mutant strains (linc-Brn1b and linc-Pint). Further analysis revealed defects in lung, gastrointestinal tract, and heart in Fendrr(-/-) neonates, whereas linc-Brn1b(-/-) mutants displayed distinct abnormalities in the generation of upper layer II-IV neurons in the neocortex. This study demonstrates that lncRNAs play critical roles in vivo and provides a framework and impetus for future larger-scale functional investigation into the roles of lncRNA molecules. DOI: http://dx.doi.org/10.7554/eLife.01749.001.
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Publication type Article: Journal article
Document type Scientific Article
Corresponding Author
Keywords Brain Development ; Developmental Defect ; Knockout Mouse Models ; Lethality ; Long Noncoding Rnas
ISSN (print) / ISBN 2050-084X
e-ISSN 2050-084X
Journal eLife
Quellenangaben Volume: 2, Issue: , Pages: , Article Number: e01749 Supplement: ,
Publisher eLife Sciences Publications
Non-patent literature Publications
Reviewing status Peer reviewed