Skip to main page content
U.S. flag

An official website of the United States government

Dot gov

The .gov means it’s official.
Federal government websites often end in .gov or .mil. Before sharing sensitive information, make sure you’re on a federal government site.

Https

The site is secure.
The https:// ensures that you are connecting to the official website and that any information you provide is encrypted and transmitted securely.

Access keys NCBI Homepage MyNCBI Homepage Main Content Main Navigation
Review
. 2017 Aug;28(7-8):377-382.
doi: 10.1007/s00335-017-9711-x. Epub 2017 Jul 29.

Phenotyping first-generation genome editing mutants: a new standard?

Affiliations
Review

Phenotyping first-generation genome editing mutants: a new standard?

Lydia Teboul et al. Mamm Genome. 2017 Aug.

Abstract

The unprecedented efficiency of the CRISPR/Cas9 system in genome engineering has opened the prospect of employing mutant founders for phenotyping cohorts, thus accelerating research projects by circumventing the requirement to generate cohorts using conventional two- or three-generation crosses. However, these first-generation mutants are often genetic mosaics, with a complex and difficult to define genetic make-up. Here, we discuss the potential benefits, challenges and scientific validity of such models.

PubMed Disclaimer

References

    1. Ablain J, Durand EM, Yang S, Zhou Y, Zon LI. A CRISPR/Cas9 vector system for tissue-specific gene disruption in zebrafish. Dev Cell. 2015;32(6):756–764. doi: 10.1016/j.devcel.2015.01.032. - DOI - PMC - PubMed
    1. Davies FC, Hope JE, McLachlan F, Nunez F, Doig J, Bengani H, Smith C, Abbott CM. Biallelic mutations in the gene encoding eEF1A2 cause seizures and sudden death in F0 mice. Sci Rep. 2017;7:46019. doi: 10.1038/srep46019. - DOI - PMC - PubMed
    1. Dickinson ME, Flenniken AM, Ji X, Teboul L, Wong MD, White JK, Meehan TF, Weninger WJ, Westerberg H, Adissu H, Baker CN, Bower L, Brown JM, Caddle LB, Chiani F, Clary D, Cleak J, Daly MJ, Denegre JM, Doe B, Dolan ME, Edie SM, Fuchs H, Gailus-Durner V, Galli A, Gambadoro A, Gallegos J, Guo S, Horner NR, Hsu CW, Johnson SJ, Kalaga S, Keith LC, Lanoue L, Lawson TN, Lek M, Mark M, Marschall S, Mason J, McElwee ML, Newbigging S, Nutter LM, Peterson KA, Ramirez-Solis R, Rowland DJ, Ryder E, Samocha KE, Seavitt JR, Selloum M, Szoke-Kovacs Z, Tamura M, Trainor AG, Tudose I, Wakana S, Warren J, Wendling O, West DB, Wong L, Yoshiki A, International Mouse Phenotyping Consortium. Jackson Laboratory. Infrastructure. Nationale. PHENOMIN , Institut. Clinique de la Souris (ICS) Charles River Laboratories. MRC. Harwell. Toronto Centre for. Phenogenomics. Wellcome. Trust Sanger Institute. RIKEN. BioResource. Center. MacArthur DG, Tocchini-Valentini GP, Gao X, Flicek P, Bradley A, Skarnes WC, Justice MJ, Parkinson HE, Moore M, Wells S, Braun RE, Svenson KL, de Angelis MH, Herault Y, Mohun T, Mallon AM, Henkelman RM, Brown SD, Adams DJ, Lloyd KC, McKerlie C, Beaudet AL, Bućan M, Murray SA. High-throughput discovery of novel developmental phenotypes. Nature. 2016;537(7621):508–514. doi: 10.1038/nature19356. - DOI - PMC - PubMed
    1. Ertzeid G, Storeng R. The impact of ovarian stimulation on implantation and fetal development in mice. Hum Reprod. 2001;16(2):221–225. doi: 10.1093/humrep/16.2.221. - DOI - PubMed
    1. Guimier A, Gabriel GC, Bajolle F, Tsang M, Liu H, Noll A, Schwartz M, El Malti R, Smith LD, Klena NT, Jimenez G, Miller NA, Oufadem M, Moreau de Bellaing A, Yagi H, Saunders CJ, Baker CN, Di Filippo S, Peterson KA, Thiffault I, Bole-Feysot C, Cooley LD, Farrow EG, Masson C, Schoen P, Deleuze JF, Nitschké P, Lyonnet S, de Pontual L, Murray SA, Bonnet D, Kingsmore SF, Amiel J, Bouvagnet P, Lo CW, Gordon CT. MMP21 is mutated in human heterotaxy and is required for normal left-right asymmetry in vertebrates. Nat Genet. 2015;47(11):1260–1263. doi: 10.1038/ng.3376. - DOI - PMC - PubMed

LinkOut - more resources