Loss of the homologous recombination gene <i>rad51</i> leads to Fanconi anemia-like symptoms in zebrafish.

ABSTRACT: RAD51 is an indispensable homologous recombination protein, necessary for strand invasion and crossing over. It has recently been designated as a Fanconi anemia (FA) gene, following the discovery of two patients carrying dominant-negative mutations. FA is a hereditary DNA-repair disorder characterized by various congenital abnormalities, progressive bone marrow failure, and cancer predisposition. In this report, we describe a viable vertebrate model of RAD51 loss. Zebrafish rad51 loss-of-function mutants developed key features of FA, including hypocellular kidney marrow, sensitivity to cross-linking agents, and decreased size. We show that some of these symptoms stem from both decreased proliferation and increased apoptosis of embryonic hematopoietic stem and progenitor cells

SUBMITTER: Botthof JG 

PROVIDER: S-EPMC5465903 | biostudies-literature | 2017 May

REPOSITORIES: biostudies-literature