ArticleFrontiers in pediatrics2025
Hemophagocytic lymphohistiocytosis as the initial manifestation of Epstein-Barr virus-related T/NK-cell lymphoproliferative disorders in a pediatric patient: a case report and literature review.
Article in Frontiers in pediatrics, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.
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1 citing paper in PubMed.
- High infant mortality, eschar absence, and universal CNS involvement in scrub typhus-associated HLH: a case series and narrative synthesis of 91 pediatric cases.Tropical medicine and health · 2026Article
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Abstract
Epstein-Barr virus-associated T/Natural Killer-cell lymphoproliferative disorders (EBV-T/NK-LPDs) are rare diseases that predominantly affect children and young adults. We report a case of a 12-year-old child who initially presented with EBV-associated hemophagocytic lymphohistiocytosis (HLH) and later progressed to aggressive NK-cell leukemia (ANKL). Both EBV-HLH and ANKL belong to the spectrum of EBV-T/NK-LPDs, which also includes chronic active EBV infection (CAEBV) of T-cell and NK-cell types, systemic EBV-positive T-cell lymphoma of childhood, extranodal NK/T-cell lymphoma (ENKTL), and primary EBV-positive nodal T/NK-cell lymphoma. The patient initially presented with recurrent high fever, pancytopenia, hepatosplenomegaly, hypertriglyceridemia, and hypofibrinogenemia with a significantly elevated EBV-DNA load. Bone marrow examination indicated a few histiocytes and hemophagocytic cells. Subsequent increases in ferritin and soluble CD25 (sCD25) levels were further consistent with the diagnosis of EBV-HLH. No lymphadenopathy or nasal primary lesion was identified. Further BM flow cytometry and BM cell morphology examination indicated abnormal NK-cell infiltration, leading to the exclusion of ENKTL, primary EBV-positive nodal T/NK-cell lymphoma, and systemic EBV-positive T-cell lymphoma of childhood. A diagnosis of ANKL was highly suspected. This case highlights that when EBV infection triggers a series of complex EBV-T/NK-LPDs occurring sequentially or simultaneously, the differential diagnosis and treatment become difficult, which can easily lead to delays in diagnosis and treatment. Developing optimized diagnostic algorithms and evidence-based treatment strategies is essential to improve outcomes of patients.
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