Focal segmental glomerulosclerosis (FSGS) occurring in association with cytomegalovirus (CMV) infection in a renal transplant patient with no previous history of FSGS has rarely been reported. We present a case of a 16‐year‐old renal transplant recipient who developed acute hepatitis, leukopenia, nephrotic syndrome, and progressive renal dysfunction in the setting of acute infection with CMV. The cytomegalovirus infection was successfully treated with IV ganciclovir followed by oral valganciclovir but renal function deterioration and massive proteinuria continued. Features of FSGS were found on two renal allograft biopsies. Plasmapheresis and cyclophosphamide treatment was instituted with no clear effect on disease progress.

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