• Caihong Wang, Junke Qiu, Xiaoqing Huang, Jiekun Xu, and Lei Pan.
• Tuberculosis Intensive Care Unit, Zhejiang Provincial Hospital of Integrated Traditional Chinese and Western Medicine, Hangzhou, Zhejiang, China.
• Medicine (Baltimore). 2024 Jul 19; 103 (29): e39011e39011.

RationaleHemophagocytic lymphohistiocytosis (HLH) is a rare, life-threatening systemic inflammatory syndrome characterized by an overactive immune response. This hyperactivation can arise from genetic mutations, infections, malignancies, or autoimmune disorders. Medication-induced HLH is extremely rare and requires special attention.Patient ConcernsA 53-year-old female diagnosed with pulmonary and urinary tract tuberculosis. She underwent quadruple therapy, including isoniazid, rifampin, ethambutol, and pyrazinamide. Subsequently, she developed fever, hepatosplenomegaly, pancytopenia, hypertriglyceridemia, hypofibrinogenemia, hyperferritinemia, increased soluble CD25 levels, decreased natural killer cell activity, and hemophagocytosis, notably without eosinophilia. Her clinical symptoms were exacerbated by rifampin intake.DiagnosesPulmonary and left kidney tuberculosis, multiple organ failure, and rifampin-induced HLH.InterventionsAnti-tuberculosis regimen (isoniazid, pyrazinamide, ethambutol, and levofloxacin, excluding rifampin) combined with glucocorticoid therapy.OutcomesSatisfactory recovery with improved clinical symptoms, laboratory tests, and chest imaging studies.LessonsEarly correct diagnosis and appropriate management of HLH are essential to save the lives of affected patients. The potential severe side effects of rifampin should not be ignored.Copyright © 2024 the Author(s). Published by Wolters Kluwer Health, Inc.

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