Sertoli Cell Only Syndrome (SCOS), the severest form of male infertility with high homogeneity, is a disorder characterized by azoospermia in semen and complete absence of germ cells in seminiferous tubules in a single testicular biopsy.The etiologic factors of the idiopathic SCOS are poorly understood.We performed whole exome sequencing (WES) in 20 sporadicallyidiopathic SCOS and found several genes that their genetic variants are repeatedly present in patients but not in control human population.