Post-transplant lymphoproliferative disorder (PTLD) occurs in ~5% of solid organ and hematopoietic stem cell transplant recipients. We report a unique presentation of PTLD in the bladder of a lung transplant recipient. Our patient was a 62-year-old female who received a bilateral lung transplant for chronic obstructive pulmonary disease. She presented with fever, left-sided flank pain and foul-smelling urine consistent with urosepsis. An abdominal and pelvic computerized tomography revealed an irregular and nodular bladder wall thickening suspicious for urothelial neoplasm. Cystoscopy revealed multiple bladder masses and biopsy demonstrated non-Hodgkin lymphoma consistent with PTLD. She was treated with a reduction in immunosuppression followed by chemotherapy and achieved remission. PTLD in the lung transplant recipients has been described in the gut, respiratory tract, skin, liver and kidney but not in the bladder. This case highlights the need for maintaining a high clinical vigilance even when transplant recipients present with seemingly benign clinical complaints.