*Purpose: Emphysematous cystitis (EC) is a rare urinary tract infection from E. coli or K. pneumoniae in diabetics or immunocompromised individuals, resulting in sepsis and death in up to 7% of cases. BK viremia has been associated with hemorrhagic cystitis in lung transplant recipients (LTRs).

*Methods: We describe a rare case of an LTR with BK viremia who successfully recovered from EC and urosepsis.

*Results: A 74-year-old bilateral LTR (July 2019) with history of diabetes, BK viruria, BK viremia, chronic kidney disease stage IIIB, and iron deficiency anemia presented to the emergency room in November 2021 with history of fever, dysuria, and abdominal discomfort. He was hemodynamically stable, and labs revealed anemia (6.8 g/dL), neutrophilia (94.6%), and acute kidney injury (creatinine: 3.7 mg/dL, eGFR: 16 mL/min/1.73m²). Urinalysis revealed a large amount of blood, red blood cells (>50/HPF), protein (100 mg/dL), white blood cells (>50/HPF), and few bacteria. Urine culture detected polymicrobial growth with K. pneumoniae, E. faecium, and E. coli. Blood culture was positive for K. pneumoniae bacteremia. Abdominal imaging revealed circumferential bladder wall thickening up to 9 mm in the greatest dimension, intraluminal and intramural gas within the urinary bladder wall, and extension of gas into the adjacent perivesicular venous plexus, suggesting EC (Figure 1). He was catheterized and admitted for intravenous fluids, vancomycin, ampicillin-sulbactam, linezolid, and glycemic control. Maintenance leflunomide and mycophenolate were discontinued; tacrolimus was continued at reduced goal. After 13 days, his cultures were negative, imaging changes resolved, and he was discharged home in stable condition. Of note, his last known BK virus load in August 2021 was 556,550 copies/mL of blood and 5.746 log copy/mL of urine; BKV DNA by quantitative PCR of serum was 520,000 copies/mL, and retroperitoneum ultrasound was negative for hydronephrosis.

*Conclusions: Although our patient had hematuria and high BK viral load despite leflunomide therapy, K. pneumoniae urosepsis, intramural bladder wall gas with circumferential thickening, and response to antibiotics favor the diagnosis of EC. Antibiotics, immunosuppression modulation, and glycemic control led to successful recovery. Surgical intervention with cystectomy or debridement may be needed in patients with severe necrotizing EC, bladder perforation, or those refractory to medical management. As far as we know, this is the first report of EC with urosepsis in an LTR with BK viremia.

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