Here we focus on the DPY19L2 locus (12q14.2) which has recently been shown to be linked with Globozoospermia [17], a rare syndrome of male infertility [18] characterized by the presence of 100% round, acrosomeless spermatozoa in the patient's ejaculate (MIM #102530). This evidence concerns the gene DPY19L2 and Globozoospermia.