Department of Respiratory Medicine, Tokyo Dental College Ichikawa General Hospital, 5-11-13, Sugano, Ichikawa, Chiba, 272-0824, Japan.
BMC Pulm Med. 2018 Mar 27;18(1):53. doi: 10.1186/s12890-018-0617-5.
Allergic bronchopulmonary aspergillosis (ABPA) is an allergic pulmonary disease comprising a complex hypersensitivity reaction to Aspergillus fumigatus. Clinical features of ABPA are wheezing, mucoid impaction, and pulmonary infiltrates. Oral corticosteroids and anti-fungal agents are standard therapy for ABPA, but long-term use of systemic corticosteroids often causes serious side effects.
A 64-year-old woman was diagnosed with ABPA based on a history of bronchial asthma (from 40 years of age), elevated total IgE, the presence of serum precipitating antibodies and elevated specific IgE antibody to A. fumigatus, and pulmonary infiltration. Bronchoscopy showed eosinophilic mucoid impaction. Systemic corticosteroid therapy was initiated, and her symptoms disappeared. Peripheral eosinophilia and pulmonary infiltration recurred five months after cessation of corticosteroid treatment. Systemic corticosteroids were re-initiated and itraconazole was added as an anti-fungal agent. The patient was free of corticosteroids, aside from treatment with a short course of systemic corticosteroids for asthma exacerbation, and clinically stable with itraconazole and asthma treatments for 3 years. In 2017, she experienced significant deterioration. Laboratory examination revealed marked eosinophilia (3017/μL) and a chest computed tomography (CT) scan demonstrated pulmonary infiltration in the left upper lobe and mucoid impaction in both lower lobes. The patient was treated with high-dose inhaled corticosteroid/long-acting beta-agonist, a long-acting muscarinic antagonist, a leukotriene receptor antagonist, and theophylline; spirometry revealed a forced expiratory volume in 1 s (FEV) of 1.01 L. An uncontrolled asthma state was indicated by an Asthma Control Test (ACT) score of 18. Mepolizumab, 100 mg every 4 weeks, was initiated for the treatment of severe bronchial asthma with ABPA exacerbation. Bronchial asthma symptoms dramatically improved, and ACT score increased to 24, by 4 weeks after mepolizumab treatment. Peripheral eosinophil count decreased to 174/μL. Spirometry revealed improvement of lung function (FEV: 1.28 L). A chest CT scan demonstrated the disappearance of pulmonary infiltration and mucoid impaction.
To our knowledge, this is the first case of ABPA to be treated with mepolizumab. Dramatic improvements were observed in symptoms, lung function, peripheral eosinophil counts, and chest images. Mepolizumab could serve as an alternative treatment with the potential to provide a systemic corticosteroid-sparing effect.
变应性支气管肺曲霉病(ABPA)是一种过敏性肺部疾病,由对烟曲霉的复杂过敏反应引起。ABPA 的临床特征包括喘息、黏液嵌塞和肺部浸润。口服皮质类固醇和抗真菌药物是 ABPA 的标准治疗方法,但长期使用全身皮质类固醇常导致严重的副作用。
一名 64 岁女性,因支气管哮喘(40 岁起)、总 IgE 升高、血清沉淀抗体阳性和特异性 IgE 抗体对烟曲霉升高,以及肺部浸润,被诊断为 ABPA。支气管镜检查显示嗜酸性黏液嵌塞。开始全身皮质类固醇治疗,症状消失。皮质类固醇治疗停止后 5 个月,外周嗜酸性粒细胞增多和肺部浸润再次出现。重新开始全身皮质类固醇治疗,并加用伊曲康唑作为抗真菌药物。除因哮喘加重接受短期全身皮质类固醇治疗外,患者无需使用皮质类固醇,并且在伊曲康唑和哮喘治疗下 3 年临床状况稳定。2017 年,她病情显著恶化。实验室检查显示嗜酸性粒细胞显著增多(3017/μL),胸部 CT 扫描显示左上肺浸润和双下肺黏液嵌塞。患者接受高剂量吸入皮质类固醇/长效β激动剂、长效毒蕈碱拮抗剂、白三烯受体拮抗剂和茶碱治疗;肺量测定显示 1 秒用力呼气量(FEV)为 1.01L。哮喘控制测试(ACT)评分为 18,表明存在未控制的哮喘状态。由于严重支气管哮喘伴 ABPA 恶化,开始使用美泊利单抗,每 4 周 100mg。支气管哮喘症状显著改善,美泊利单抗治疗 4 周后 ACT 评分增加至 24。外周嗜酸性粒细胞计数降至 174/μL。肺量测定显示肺功能改善(FEV:1.28L)。胸部 CT 扫描显示肺部浸润和黏液嵌塞消失。
据我们所知,这是首例使用美泊利单抗治疗 ABPA 的病例。症状、肺功能、外周嗜酸性粒细胞计数和胸部图像均有显著改善。美泊利单抗可作为一种替代治疗方法,具有潜在的全身皮质类固醇节省效应。