BackgroundCytomegalovirus (CMV) infection remains one of the most common and feared complications of transplantation, justifying prophylaxis or preemptive strategies guided by donor and recipient CMV serostatus. In case of seronegative donor and recipient (D−/R−), no prophylaxis is recommended. Late-onset CMV disease is usually defined as occurring after prophylaxis discontinuation in D+/R− transplant patients.Case presentationWe are reporting the case of a D−/R− kidney Caucasian transplant recipient presenting with CMV primoinfection 12 years after renal transplant, and discuss the role of a secondary prophylaxis so late after transplantation.ConclusionsPrimary infections leading to late-onset CMV disease in transplant patients remain rare. Recurrent disease has been described in as many as one-third of these patients. A systematic secondary prophylaxis in this particular group of patients is questionable.