Strain-Dependent Modifier Genes Determine Survival in Zfp423 Mice

Author:

Alcaraz Wendy A,Liu Zheng,Valdes Phoebe,Chen Edward,Valdovino Gonzalez Alan G,Wade Shelby,Wong Cinny,Kim Eunnie,Chen Hsiang-Hua M,Ponn Alison,Concepcion Dorothy,Hamilton Bruce A

Abstract

Abstract Zfp423 encodes a transcriptional regulatory protein that interacts with canonical signaling and lineage pathways. Mutations in mouse Zfp423 or its human ortholog ZNF423 are associated with a range of developmental abnormalities reminiscent of ciliopathies, including cerebellar vermis hypoplasia and other midline brain defects. Null mice have reduced viability in most strain backgrounds. Here we show complete lethality on a C57BL/6J background, dominant rescue in backcrosses to any of 13 partner strains, with strain-dependent survival frequencies, and evidence for a BALB/c-derived survival modifier locus on chromosome 5. Survival data indicate both perinatal and postnatal periods of lethality. Anatomical data from a hypomorphic gene trap allele observed on both C57BL/6J and BALB/c congenic backgrounds shows an aggregate effect of background on sensitivity to Zfp423 loss rather than a binary effect on viability.

Publisher

Oxford University Press (OUP)

Subject

Genetics(clinical),Genetics,Molecular Biology

Reference33 articles.

1. Modifier genes and non-genetic factors reshape anatomical deficits in Zfp423-deficient mice.;Alcaraz;Hum. Mol. Genet.,2011

2. Zfp423 controls proliferation and differentiation of neural precursors in cerebellar vermis formation.;Alcaraz;Proc. Natl. Acad. Sci. USA,2006

3. The ciliopathies: an emerging class of human genetic disorders.;Badano;Annu. Rev. Genomics Hum. Genet.,2006

4. Multigenic control of Listeria monocytogenes susceptibility in mice.;Boyartchuk;Nat. Genet.,2001

5. Mapping quantitative trait loci in the case of a spike in the phenotype distribution.;Broman;Genetics,2003

同舟云学术

1.学者识别学者识别

2.学术分析学术分析

3.人才评估人才评估

"同舟云学术"是以全球学者为主线,采集、加工和组织学术论文而形成的新型学术文献查询和分析系统,可以对全球学者进行文献检索和人才价值评估。用户可以通过关注某些学科领域的顶尖人物而持续追踪该领域的学科进展和研究前沿。经过近期的数据扩容,当前同舟云学术共收录了国内外主流学术期刊6万余种,收集的期刊论文及会议论文总量共计约1.5亿篇,并以每天添加12000余篇中外论文的速度递增。我们也可以为用户提供个性化、定制化的学者数据。欢迎来电咨询!咨询电话:010-8811{复制后删除}0370

www.globalauthorid.com

TOP

Copyright © 2019-2024 北京同舟云网络信息技术有限公司
京公网安备11010802033243号  京ICP备18003416号-3