Abstract
BackgroundHemophagocytic lymphohistiocytosis is a rare, potentially fatal syndrome of immune hyperactivation. Here we describe a ganglionar tuberculosis evolving to hemophagocytic lymphohistiocytosis following adjuvant immunotherapy in a melanoma patient.Case presentationA 76-year-old Caucasian male with melanoma started with fever, diffuse petechiae, splenomegaly, anemia, thrombocytopenia, hypofibrinogenemia, and hyperferritinemia 2 months following completion of adjuvant treatment with nivolumab. Positron emission tomography scan showed significant hypermetabolism in cervical, supraclavicular, mediastinal, and abdominal lymph nodes. Bone marrow aspiration demonstrated no alterations, except for a hypercellular pattern. Dexamethasone and intravenous immunoglobulin were started owing to suspicion of hemophagocytic lymphohistiocytosis. Core biopsy of the infracarinal lymph node revealed a chronic granulomatous inflammation and caseous necrosis, with positivity for Mycobacterium tuberculosis by polymerase chain reaction, and treatment for ganglionar tuberculosis was started.ConclusionThis case highlights the challenges involving programmed cell death 1 blockade in high-risk melanoma, in which infections, lymphoproliferative disorders, and sarcoidosis can mimic disease progression and trigger immune-related adverse events.
Highlights
Hemophagocytic lymphohistiocytosis is a rare, potentially fatal syndrome of immune hyperactivation
TB is most often characterized by pulmonary involvement, while extrapulmonary disease accounts for only 20% of all TB manifestations, and its association with immunosuppression such as human immunodeficiency virus (HIV), hepatitis, diabetes, alcohol abuse, drug addiction, and transplant recipients is well documented [9]
The only risk factor associated with Mycobacterium tuberculosis (Mtb) infection is Brazil’s endemic area, where it accounts for the highest number of TB cases in the Americas according to the World Health Organization (WHO) [10]
Summary
This case highlights the challenges involving clinical use of anti-PD-1 agents, especially in the context of high-risk cutaneous melanoma, in which infections, lymphoproliferative disorders, and even sarcoidosis can mimic disease progression. IrAE is becoming more evident owing to the increased use of immunotherapy, but many clinical conditions are still complex, especially when associated with infectious diseases. To our knowledge, this is the first case report of an Mtb infection evolving to HLH after anti-PD-1 treatment for high-risk melanoma
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