The phenotype of FancB-mutant mouse embryonic stem cells

Tae Moon Kim; Jun Ho Ko; Yong Jun Choi; Lingchuan Hu; Paul Hasty

doi:10.1016/j.mrfmmm.2011.03.010

The phenotype of FancB-mutant mouse embryonic stem cells

Tae Moon Kim, Jun Ho Ko, Yong Jun Choi, Lingchuan Hu, Paul Hasty

Barshop Institute

Research output: Contribution to journal › Article › peer-review

14 Scopus citations

Abstract

Fanconi anemia (FA) is a rare autosomal recessive disease characterized by bone marrow failure, developmental defects and cancer. There are multiple FA genes that enable the repair of interstrand crosslinks (ICLs) in coordination with a variety of other DNA repair pathways in a way that is poorly understood. Here we present the phenotype of mouse embryonic stem (ES) cells mutated for FancB. We found FancB-mutant cells exhibited reduced cellular proliferation, hypersensitivity to the crosslinking agent mitomycin C (MMC), increased spontaneous and MMC-induced chromosomal abnormalities, reduced spontaneous sister chromatid exchanges (SCEs), reduced gene targeting, reduced MMC-induced Rad51 foci and absent MMC-induced FancD2 foci. Since FancB is on the X chromosome and since ES cells are typically XY, FancB is an excellent target for an epistatic analysis to elucidate FA's role in ICL repair.

Original language	English (US)
Pages (from-to)	20-27
Number of pages	8
Journal	Mutation Research - Fundamental and Molecular Mechanisms of Mutagenesis
Volume	712
Issue number	1-2
DOIs	https://doi.org/10.1016/j.mrfmmm.2011.03.010
State	Published - Jul 1 2011

Keywords

FancD2
Fanconi anemia
G2 arrest
Interstrand crosslink
Rad51
Radial

ASJC Scopus subject areas

Molecular Biology
Genetics
Health, Toxicology and Mutagenesis

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10.1016/j.mrfmmm.2011.03.010

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title = "The phenotype of FancB-mutant mouse embryonic stem cells",

abstract = "Fanconi anemia (FA) is a rare autosomal recessive disease characterized by bone marrow failure, developmental defects and cancer. There are multiple FA genes that enable the repair of interstrand crosslinks (ICLs) in coordination with a variety of other DNA repair pathways in a way that is poorly understood. Here we present the phenotype of mouse embryonic stem (ES) cells mutated for FancB. We found FancB-mutant cells exhibited reduced cellular proliferation, hypersensitivity to the crosslinking agent mitomycin C (MMC), increased spontaneous and MMC-induced chromosomal abnormalities, reduced spontaneous sister chromatid exchanges (SCEs), reduced gene targeting, reduced MMC-induced Rad51 foci and absent MMC-induced FancD2 foci. Since FancB is on the X chromosome and since ES cells are typically XY, FancB is an excellent target for an epistatic analysis to elucidate FA's role in ICL repair.",

keywords = "FancD2, Fanconi anemia, G2 arrest, Interstrand crosslink, Rad51, Radial",

author = "Kim, {Tae Moon} and Ko, {Jun Ho} and Choi, {Yong Jun} and Lingchuan Hu and Paul Hasty",

note = "Funding Information: We thank Charnae Williams for technical expertise and Dr. Allan Bradley for the gift of the AB2.2 cells and the pu Δ tk selection cassette. This work was supported by the following NIH grants to PH: 1 RO1 CA123203-01A1 and P01 AG17242 .",

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AU - Ko, Jun Ho

AU - Choi, Yong Jun

AU - Hu, Lingchuan

AU - Hasty, Paul

N1 - Funding Information: We thank Charnae Williams for technical expertise and Dr. Allan Bradley for the gift of the AB2.2 cells and the pu Δ tk selection cassette. This work was supported by the following NIH grants to PH: 1 RO1 CA123203-01A1 and P01 AG17242 .

PY - 2011/7/1

Y1 - 2011/7/1

N2 - Fanconi anemia (FA) is a rare autosomal recessive disease characterized by bone marrow failure, developmental defects and cancer. There are multiple FA genes that enable the repair of interstrand crosslinks (ICLs) in coordination with a variety of other DNA repair pathways in a way that is poorly understood. Here we present the phenotype of mouse embryonic stem (ES) cells mutated for FancB. We found FancB-mutant cells exhibited reduced cellular proliferation, hypersensitivity to the crosslinking agent mitomycin C (MMC), increased spontaneous and MMC-induced chromosomal abnormalities, reduced spontaneous sister chromatid exchanges (SCEs), reduced gene targeting, reduced MMC-induced Rad51 foci and absent MMC-induced FancD2 foci. Since FancB is on the X chromosome and since ES cells are typically XY, FancB is an excellent target for an epistatic analysis to elucidate FA's role in ICL repair.

AB - Fanconi anemia (FA) is a rare autosomal recessive disease characterized by bone marrow failure, developmental defects and cancer. There are multiple FA genes that enable the repair of interstrand crosslinks (ICLs) in coordination with a variety of other DNA repair pathways in a way that is poorly understood. Here we present the phenotype of mouse embryonic stem (ES) cells mutated for FancB. We found FancB-mutant cells exhibited reduced cellular proliferation, hypersensitivity to the crosslinking agent mitomycin C (MMC), increased spontaneous and MMC-induced chromosomal abnormalities, reduced spontaneous sister chromatid exchanges (SCEs), reduced gene targeting, reduced MMC-induced Rad51 foci and absent MMC-induced FancD2 foci. Since FancB is on the X chromosome and since ES cells are typically XY, FancB is an excellent target for an epistatic analysis to elucidate FA's role in ICL repair.

KW - FancD2

KW - Fanconi anemia

KW - G2 arrest

KW - Interstrand crosslink

KW - Rad51

KW - Radial

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The phenotype of FancB-mutant mouse embryonic stem cells

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