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Secondary Leukemia in a Patient With EBV-HLH Carrying Heterozygous STXBP2 Variant
1Department of Hematology and Oncology, Chongqing Medical University Affiliated Children's Hospital, National Clinical Research Center for Child Health and Disorders, Ministry of Education Key Laboratory of Child Development and Disorders, Chongqing Key Laboratory of Pediatrics, Chongqing, China.
Hemophagocytic lymphohistiocytosis (HLH) is a symptom with severe systemic hyperinflammation. A 26-month-old male presented with Epstein-Barr virus associated HLH with a heterozygous variant of syntaxin-binding protein-2 (STXBP2). Complete remission was achieved with the HLH-2004 protocol, but the disease soon relapsed. Four weeks after re-installing HLH-2004 protocol, HLH was resolved. The cumulative dosage of etoposide was 2100 mg/m2. He developed acute promyelocytic leukemia 17 months later. The patient underwent standard chemotherapy and since remained in complete remission. In conclusion, a regular screening of malignance might be in necessity for the patients harboring gene variants associated with familial HLH.
Hemophagocytic lymphohistiocytosis (HLH) is a symptom with severe systemic hyperinflammation. A 26-month-old male presented with Epstein-Barr virus associated HLH with a heterozygous variant of syntaxin-binding protein-2 (STXBP2). Complete remission was achieved with the HLH-2004 protocol, but the disease soon relapsed. Four weeks after re-installing HLH-2004 protocol, HLH was resolved. The cumulative dosage of etoposide was 2100 mg/m2. He developed acute promyelocytic leukemia 17 months later. The patient underwent standard chemotherapy and since remained in complete remission. In conclusion, a regular screening of malignance might be in necessity for the patients harboring gene variants associated with familial HLH.
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