The aim of this study is to evaluate the pregnancy outcomes of males with a 47, XYY karyotype following assisted reproductive treatment.A retrospective study was performed using data from infertile men with 47, XYY at a center for reproductive medicine in 2004 to 2017. Of the 19,842 infertile males treated, a total of 21 showed the 47, XYY karyotype and were included in the present study. Clinical variables were collected. Three men were under treatment with their partner before either in vitro fertilization (IVF) or intracytoplasmic sperm injection (ICSI).The incidence of 47, XYY in infertile men is 1/945 (21/19842). Most men are azoospermic or severely oligospermic. Three men and their partners underwent IVF or ICSI treatment with fresh ejaculate samples. The fertilization rate was 52.94% to 83.33%. The embryo cleavage rate was 50% to 90%. One man had abnormal sex hormonal levels and his partner had no clinical pregnancy. The other 2 couples had healthy baby boys.Live spermatozoa can be gathered and fertility is possible for infertile males with 47, XYY syndrome when IVF or ICSI treatment is used. It is recommended that genetic counseling is provided in such cases.