Orderly segregation of chromosomes during meiosis requires that crossovers form between homologous chromosomes by recombination. Programmed DNA double-strand breaks (DSBs) initiate meiotic recombination. We identify ANKRD31 as a key component of complexes of DSB-promoting proteins that assemble on meiotic chromosome axes. Genome-wide, ANKRD31 deficiency causes delayed recombination initiation. In addition, loss of ANKRD31 alters DSB distribution because of reduced selectivity for sites that normally attract DSBs. Strikingly, ANKRD31 deficiency also abolishes uniquely high rates of recombination that normally characterize pseudoautosomal regions (PARs) of X and Y chromosomes. Consequently, sex chromosomes do not form crossovers, leading to chromosome segregation failure in ANKRD31-deficient spermatocytes. These defects co-occur with a genome-wide delay in assembling DSB-promoting proteins on autosome axes and loss of a specialized PAR-axis domain that is highly enriched for DSB-promoting proteins in wild type. Thus, we propose a model for spatiotemporal patterning of recombination by ANKRD31-dependent control of axis-associated DSB-promoting proteins.

Department of Biomedicine and Prevention Faculty of Medicine University of Rome Tor Vergata Via Montpellier n 1 00133 Rome Italy

Institute for Brain Research and Rehabilitation South China Normal University 510631 Guangzhou China

Institute of Human Genetics UMR 9002 CNRS Université de Montpellier 34396 Montpellier Cedex 5 France

Institute of Molecular Genetics Division BIOCEV Prumyslova 595 25250 Vestec Czech Republic

Institute of Physiological Chemistry Faculty of Medicine Technische Universität Dresden Fetscherstraße 74 01307 Dresden Germany

MRC Human Genetics Unit MRC Institute of Genetics and Molecular Medicine University of Edinburgh Western General Hospital Edinburgh EH4 2XU UK

Research Institute of Molecular Pathology 1030 Vienna Austria

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