A Rare Case of Hemophagocytic Lymphohistiocytosis Triggered by Intravesical Bacillus Calmette-Guérin (BCG) for Bladder Cancer.
Mizuki Nakayama, Toyoshi Yanagihara, Makoto Fujimoto, Natsumi Kushima, Yu Okabe, Mikiko Aoki, Kaori Koga, Keiji Yokoyama, Yuka Sakaki, Takato Ikeda, Naoki Hamada, Makoto Hamasaki, Masaki Fujita
Abstract
Open AccessIntravesical bacillus Calmette-Guérin (BCG) therapy is widely used for non-muscle-invasive bladder cancer but can rarely lead to severe systemic complications. Hemophagocytic lymphohistiocytosis (HLH) is a life-threatening hyperinflammatory syndrome characterized by fever, cytopenias, and organ dysfunction. We describe a 63-year-old man who developed HLH after re-induction BCG therapy. He presented with fever, hepatosplenomegaly, pancytopenia, and liver and kidney dysfunction. Laboratory tests revealed hyperferritinemia and elevated soluble interleukin-2 receptor levels. Liver biopsy showed non-caseating granulomas without acid-fast bacilli, and bone marrow biopsy confirmed hemophagocytosis. Urine culture yielded Mycobacterium tuberculosis complex, consistent with prior BCG instillation. Treatment with high-dose corticosteroids, intravenous immunoglobulin, and BCG-active antibiotics resulted in rapid recovery. This case illustrates that HLH is a rare but serious complication of intravesical BCG, and emphasizes the importance of early recognition and combined immunosuppressive and anti-mycobacterial therapy.