Department of Paediatric Haematology and Oncology, University Medical Center, Hamburg, Germany.
Strategic Direction Staff, Children's Hospital Giovanni XXIII, Azienda Ospedaliero Universitaria Consorziale Policlinico, Bari, Italy.
Acta Paediatr. 2021 Oct;110(10):2723-2728. doi: 10.1111/apa.15889. Epub 2021 May 19.
Familial haemophagocytic lymphohistiocytosis (FHL) is an inherited immune deficiency with defective cytotoxicity of natural killer cells and cytotoxic T lymphocytes. A highly stimulated, but ineffective immune response leads to severe hyperinflammation. Clinical and laboratory features are characteristic, but unspecific; thus awareness of FHL is important for early diagnosis. FHL is rapidly fatal without treatment. Standard-of-care therapy is etoposide and corticosteroids, followed by haematopoietic stem cell transplantation (HSCT). CONCLUSION: FHL has become a curable disease with present treatment. Additional cytokine-directed therapy still has to prove its value. Earlier HSCT and less toxic conditioning regimens will lead to improved cure rates.
家族性噬血细胞性淋巴组织细胞增生症(FHL)是一种遗传性免疫缺陷病,其特征为自然杀伤细胞和细胞毒性 T 淋巴细胞的细胞毒性缺陷。高度活跃但无效的免疫反应导致严重的过度炎症。临床和实验室特征具有特征性,但不具有特异性;因此,对 FHL 的认识对于早期诊断很重要。如果不治疗,FHL 会迅速致命。标准治疗方法是依托泊苷和皮质类固醇,随后进行造血干细胞移植(HSCT)。结论:目前的治疗方法使 FHL 成为一种可治愈的疾病。额外的细胞因子靶向治疗仍需证明其价值。更早的 HSCT 和毒性更小的预处理方案将提高治愈率。