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A case report of hemophagocytic lymphohistiocytosis occurring during ibrutinib chemotherapy with a literature review.

Medicine 2026 Vol.105(3) p. e45957

Hou X, Wang M, Miao S, Guo Y

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[RATIONALE] Ibrutinib, a Bruton tyrosine kinase inhibitor, is widely used in chronic lymphocytic leukemia (CLL).

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BibTeX ↓ RIS ↓
APA Hou X, Wang M, et al. (2026). A case report of hemophagocytic lymphohistiocytosis occurring during ibrutinib chemotherapy with a literature review.. Medicine, 105(3), e45957. https://doi.org/10.1097/MD.0000000000045957
MLA Hou X, et al.. "A case report of hemophagocytic lymphohistiocytosis occurring during ibrutinib chemotherapy with a literature review.." Medicine, vol. 105, no. 3, 2026, pp. e45957.
PMID 41559979

Abstract

[RATIONALE] Ibrutinib, a Bruton tyrosine kinase inhibitor, is widely used in chronic lymphocytic leukemia (CLL). Its association with secondary hemophagocytic lymphohistiocytosis (HLH) is exceedingly rare but potentially fatal.

[PATIENT CONCERNS] A 59-year-old male with CLL presented with recurrent fever and progressive splenomegaly.

[DIAGNOSES] Bone marrow biopsy confirmed CLL. Five months after ibrutinib (420 mg/day) initiation, progressive pancytopenia, hyperferritinemia, and hemophagocytosis on repeat marrow aspirate established HLH diagnosis.

[INTERVENTIONS] HLH-94 protocol (etoposide + dexamethasone) was initiated.

[OUTCOMES] The patient developed fungemia followed by massive cerebral infarction. Treatment was withdrawn due to deterioration, resulting in death.

[LESSONS] Ibrutinib may trigger HLH via immune dysregulation. Early recognition through vigilant monitoring of cytopenias, ferritin, and marrow hemophagocytosis is critical to improve survival.

MeSH Terms

Humans; Male; Lymphohistiocytosis, Hemophagocytic; Middle Aged; Adenine; Piperidines; Leukemia, Lymphocytic, Chronic, B-Cell; Fatal Outcome; Pyrimidines; Pyrazoles

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