Although globozoospermia (round-headed spermatozoa) is encountered as 0.1% of infertile population, familial appearance of globozoospermia is extremely limited. In this report, we aimed to describe 26 and 45-year-old male siblings with the clinical and pathologic details. Both light and electron microscopic examinations were performed for the diagnosis of complete globozoospermia. Of note, the karyotype analyses of the two siblings revealed 46+XY without any microdeletion on chromosome Y. In the available literature, there are only six more reports describing the familial globozoospermia. Although association with several aneuplodies has been reported, specific numerical or ultra structural abnormalities in somatic or sex chromosomes have not been clarified. In this sense, the low incidence of the familial globozoospermia may be the primary cause in order to fail to demonstrate any specific inheritance in such cases. However, documentation of siblings with globozoospermia may warrant strong and undetected genetic impact for the pathogenesis of the globozoospermia.

Keywords: Male infertility, teratozoospermia, sperm, sterility