Whole exome sequencing (WES) performed in three patients with globozoospermia revealed a previously undescribed pathogenic nucleotide sequence variant in exon 6 of the DPY19L2 gene (n = 1), a variant of unknown significance in the DPY19L2 gene (n = 1), and a previously undescribed nucleotide sequence variant of unknown clinical significance in exon 3 of the SPATA16 gene (n = 1). This evidence concerns the gene DPY19L2 and Globozoospermia.