Lee Jin, Bae Kil Seong, Rhim Jung Woo, Lee Soo-Young, Jeong Dae Chul, Kang Jin Han
Department of Pediatrics, College of Medicine, The Catholic University of Korea, Seoul 06591, Republic of Korea.
Department of Pediatrics, Incheon St. Mary's Hospital, The Catholic University of Korea, Incheon 21431, Republic of Korea.
Children (Basel). 2024 Jun 21;11(7):755. doi: 10.3390/children11070755.
Macrophage activation syndrome (MAS) is potentially fatal; so, early diagnosis and timely treatment are essential. However, detecting MAS is sometimes challenging because its principal features can be observed in other pediatric diseases that cause severe inflammation. Cytokine storm due to immune dysregulation represents the clinical and laboratory features of MAS that are included in the diagnostic criteria. Most cases of MAS occur as an underlying condition worsens and progresses. Therefore, a patient with autoimmune or autoinflammatory disease who shows unexplained clinical deterioration despite appropriate management should be considered at high risk for MAS (i.e., occult MAS). The basic principles of treatment are control of triggering factors, supportive care, and relief of hyperinflammation. Systemic steroids and cyclosporine A are frequently used as a first-line treatment. For the treatment of refractory MAS, cytokine-specific biologic agents such as anakinra have recently become preferred over traditional immunosuppressive agents such as etoposide. MAS might be underrecognized in pediatric patients with infectious and inflammatory diseases due to its diverse clinical presentations. Clinical suspicion of MAS is of the utmost importance for early recognition of the disease.
巨噬细胞活化综合征(MAS)可能会致命,因此早期诊断和及时治疗至关重要。然而,检测MAS有时具有挑战性,因为其主要特征也可见于其他引起严重炎症的儿科疾病中。免疫失调导致的细胞因子风暴代表了MAS的临床和实验室特征,这些特征包含在诊断标准中。大多数MAS病例是在潜在疾病恶化和进展时发生的。因此,患有自身免疫性或自身炎症性疾病的患者,尽管接受了适当治疗仍出现无法解释的临床恶化,应被视为MAS的高危人群(即隐匿性MAS)。治疗的基本原则是控制触发因素、支持治疗和缓解过度炎症。全身用类固醇和环孢素A常被用作一线治疗药物。对于难治性MAS的治疗,近年来,细胞因子特异性生物制剂如阿那白滞素比依托泊苷等传统免疫抑制剂更受青睐。由于MAS临床表现多样,在患有感染性和炎症性疾病的儿科患者中可能未得到充分认识。临床怀疑MAS对于早期识别该疾病至关重要。