THAP1: Role in Mouse Embryonic Stem Cell Survival and Differentiation

被引：24

作者：

Aguilo, Francesca ^{[1
,2
,10
]}

Zakirova, Zuchra ^{[3
]}

Nolan, Katie ^{[3
]}

Wagner, Ryan ^{[4
,5
]}

Sharma, Rajal ^{[1
]}

Hogan, Megan ^{[4
,5
]}

Wei, Chengguo ^{[6
]}

Sun, Yifei ^{[1
,2
]}

Walsh, Martin J. ^{[1
,7
]}

Kelley, Kevin ^{[4
]}

Zhang, Weijia ^{[6
]}

Ozelius, Laurie J. ^{[8
]}

Gonzalez-Alegre, Pedro ^{[9
]}

Zwaka, Thomas P. ^{[4
,5
]}

Ehrlich, Michelle E. ^{[2
,3
,7
]}

机构：

[1] Icahn Sch Med Mt Sinai, Dept Pharmacol Sci, New York, NY 10029 USA

[2] Icahn Sch Med Mt Sinai, Dept Pediat, New York, NY 10029 USA

[3] Icahn Sch Med Mt Sinai, Dept Neurol, New York, NY 10029 USA

[4] Icahn Sch Med Mt Sinai, Dept Cell Dev & Regenerat Biol, New York, NY 10029 USA

[5] Icahn Sch Med Mt Sinai, Black Family Stem Cell Inst, New York, NY 10029 USA

[6] Icahn Sch Med Mt Sinai, Dept Med Bioinformat Core, New York, NY 10029 USA

[7] Icahn Sch Med Mt Sinai, Dept Genet & Genom Sci, New York, NY 10029 USA

[8] Harvard Med Sch, Massachusetts Gen Hosp, Dept Neurol, Charlestown, MA 02129 USA

[9] Univ Penn, Perelman Sch Med, Childrens Hosp Philadelphia, Dept Neurol,Perelman Ctr Cellular & Mol Therapeut, Philadelphia, PA 19104 USA

[10] Umea Univ, Wallenberg Ctr Mol Med, Dept Med Biosci, S-90185 Umea, Sweden

来源：

STEM CELL REPORTS | 2017年 / 9卷 / 01期

关键词：

LARGE GENE LISTS; DYT6; DYSTONIA; IN-VITRO; PROTEIN; TRANSCRIPTION; EXPRESSION; MUTATIONS; PLURIPOTENCY; ENHANCERS; DOMAIN;

D O I：

10.1016/j.stemcr.2017.04.032

中图分类号：

Q813 [细胞工程];

学科分类号：

摘要：

THAP1 (THAP [Thanatos-associated protein] domain-containing, apoptosis-associated protein 1) is a ubiquitously expressed member of a family of transcription factors with highly conserved DNA-binding and protein-interacting regions. Mutations in THAP1 cause dystonia, DYT6, a neurologic movement disorder. THAP1 downstream targets and the mechanism via which it causes dystonia are largely unknown. Here, we show that wild-type THAP1 regulates embryonic stem cell (ESC) potential, survival, and proliferation. Our findings identify THAP1 as an essential factor underlying mouse ESC survival and to some extent, differentiation, particularly neuroectodermal. Loss of THAP1 or replacement with a disease-causing mutation results in an enhanced rate of cell death, prolongs Nanog, Prdm14, and/or Rex1 expression upon differentiation, and results in failure to upregulate ectodermal genes. ChIP-Seq reveals that these activities are likely due in part to indirect regulation of gene expression.

引用

页码：92 / 107

页数：16

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