AbstractUterus transplantation is a repeatedly proven treatment for women with absolute uterine‐factor infertility, which is the congenital or acquired absence of the uterus, who desire to carry, and ultimately deliver, a child. No stranger to the field of transplant or obstetrics is cytomegalovirus. Cytomegalovirus is both a frequent complication after transplant, presenting as an opportunistic infection, and a common congenital disease in the newborn child from pregnancy. To date, there have been no reported cases of pregnancy following uterus transplantation from cytomegalovirus‐positive donors into cytomegalovirus‐negative recipients. We present a case report describing our experience of a cytomegalovirus‐negative recipient, transplanted with a uterus from a cytomegalovirus‐positive living donor, and subsequently diagnosed with active cytomegalovirus infection despite prophylactic treatment. She was treated for infection prior to embryo transfer and carried a healthy child to term. This case suggests transplanting a cytomegalovirus‐positive uterus into a negative donor is possible to do safely.