Emory University School of Medicine, Atlanta, GA, USA.
Children's Healthcare of Atlanta, GA, USA.
J Investig Med High Impact Case Rep. 2022 Jan-Dec;10:23247096221077836. doi: 10.1177/23247096221077836.
Many pediatric rheumatic diseases can be safely managed with biologic therapy. Severe allergic reactions to these medications are uncommon. We report the case of a 2-year-old male with systemic-onset juvenile idiopathic arthritis and secondary macrophage activation syndrome (MAS), whose treatment was complicated by severe allergic reactions to biologics, including drug reaction with eosinophilia and systemic symptoms (DRESS)/drug-induced hypersensitivity reaction (DIHR) likely due to anakinra, and anaphylactoid reaction to intravenous tocilizumab. These required transition to canakinumab, cyclosporine, and corticosteroids, with later development of interstitial lung disease and MAS flare needing transition from canakinumab to tofacitinib, which led to disease control. Whether lung disease is a manifestation of DRESS/DIHR to canakinumab remains unclear. High index of suspicion of hypersensitivity reactions for timely diagnosis and drug discontinuation is critical, especially in patients with active disease who might be at increased risk of these adverse events.
许多儿科风湿性疾病可以用生物疗法安全地治疗。这些药物的严重过敏反应并不常见。我们报告了一例 2 岁男性全身型幼年特发性关节炎和继发巨噬细胞活化综合征（MAS），他的治疗因对生物制剂的严重过敏反应而复杂化，包括可能由阿那白滞素引起的药物反应伴嗜酸性粒细胞增多和全身症状（DRESS）/药物引起的过敏反应（DIHR），以及对静脉注射托珠单抗的类过敏反应。这需要转换为康那单抗、环孢素和皮质类固醇，后来发展为间质性肺病和 MAS 发作，需要从康那单抗转换为托法替尼，这导致疾病得到控制。肺部疾病是否是对康那单抗的 DRESS/DIHR 的表现尚不清楚。高度怀疑过敏反应以进行及时诊断和停药至关重要，特别是在患有活动性疾病的患者中，他们可能有更高的发生这些不良事件的风险。
