Beijing Children's Hospital Affiliated to Capital Medical University, National Center for Children's Health, No. 56 South Lishi Road, Beijing, 10045, China.
J Clin Immunol. 2018 Feb;38(2):155-158. doi: 10.1007/s10875-017-0473-8. Epub 2018 Jan 4.
Hypersensitive pneumonitis (HP) is a rare initial presentation of chronic granulomatous disease (CGD), especially in children. CGD presenting as HP may result from exposure to inhaled environmental antigens and be induced by excessive production of inflammatory cytokines due to loss of reactive oxygen species production. We herein describe a 2-year-old boy with CGD caused by a mutation in CYBB gene, who initially presented with HP. The patient developed dry cough, progressive dyspnea, and fever after playing in dusty air in a factory that produced plastic materials containing isocyanates. The patient's symptoms and radiological abnormalities did not improve after antigen avoidance, but disappeared after corticosteroid therapy. Because HP is uncommon in children, we should consider the possibility of CGD in children who present with HP. A prompt diagnosis of CGD is essential to enable initiation of prophylactic antibacterial and antifungal therapies.
过敏性肺炎（HP）是慢性肉芽肿病（CGD）的罕见首发表现，尤其是在儿童中。CGD 以 HP 表现出来可能是由于吸入环境抗原所致，并且由于活性氧产生的丧失而导致炎症细胞因子过度产生而引起。我们在此描述了一例由 CYBB 基因突变引起的 CGD 患儿，其最初表现为 HP。患儿在一家生产含有异氰酸酯的塑料材料的工厂的灰尘空气中玩耍后，出现干咳、进行性呼吸困难和发热。患者在避免抗原后症状和影像学异常无改善，但在皮质类固醇治疗后消失。由于 HP 在儿童中并不常见，因此我们应该考虑出现 HP 的儿童是否患有 CGD。及时诊断 CGD 对于启动预防性抗菌和抗真菌治疗至关重要。
