Airway clearance ameliorated the control of diffuse panbronchiolitis accompanied by asthma and leukocytoclastic vasculitis
Airway clearance ameliorated the control of diffuse panbronchiolitis accompanied by asthma and leukocytoclastic vasculitis
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气道清除改善了伴有哮喘和白细胞破碎性血管炎的弥漫性全细支气管炎的控制
DOI：
10.1016/j.alit.2020.07.003
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发表时间：
2021
影响因子：
6.8
通讯作者：
et al.
中科院分区：
文献类型：
--
作者：
Tashima N;Matsumoto H;et al.
哮喘偶尔并发支气管扩张或弥漫性泛细支气管炎（DPB），这使得哮喘或哮喘样症状难以控制，可能是由于亚临床感染或细菌定植。1在此，我们报告了一例难治性哮喘合并DPB或迁延性细菌性细支气管炎的病例，该病例对大环内酯类药物治疗耐药，并导致皮肤白细胞破碎性血管炎（CLV）。在Acapella®（一种振荡式正压呼气装置，可促进气道清除）诱导后，所有状况均显著改善。一名21岁的非吸烟女性，在5岁时发生哮喘，接受ICS、LABA和白三烯受体拮抗剂治疗，直到18岁时，她患上肺炎球菌肺炎和急性鼻窦炎。这些急性感染用抗生素成功治疗;然而，排痰性咳嗽和轻度鼻窦炎持续存在。根据症状、体征和胸部计算机断层扫描（CT）结果，既往医生诊断患者为DPB合并症，符合以下三个主要标准：持续咳嗽、咳痰和劳力性呼吸困难;既往慢性鼻窦炎复发史;胸部平片上双侧弥漫性小结节影或胸部CT图像上小叶中心结节影;以及以下三个次要标准中的至少两个：粗裂纹; FEV 1/用力肺活量（FVC）< 70%和动脉氧分压< 80 mmHg;日本厚生省（MHLW）提出的冷凝集素滴度64。2实验室检查结果显示，白色血细胞（WBC）计数为6030/μL（嗜酸性粒细胞9.5%），C反应蛋白（CRP）水平为1.5 mg/dL，总IgE为721 IU/mL，对吸入性过敏原（包括屋尘螨、雪松、曲霉菌和链格孢菌）有多种致敏作用，冷凝集素滴度为128，抗中性粒细胞胞质自身抗体结果为阴性。肺功能检查显示FEV 1为2.22 L（预测值的72%），FVC为2.81 L（预测值的81%）。培养铜绿假单胞菌，在痰中观察到明显的嗜酸性炎症。患者从18岁开始接受红霉素（EM）或克拉霉素的低剂量大环内酯类药物治疗，未能改善其症状。然而，奥马珠单抗略微减少排痰性咳嗽。在20岁时，出现了可触及的紫癜和水肿，双腿疼痛，导致基于皮肤活检诊断为CLV。
Asthma is occasionally complicated with bronchiectasis or diffuse panbronchiolitis (DPB), which makes asthma or asthmalike symptoms difficult to control, potentially owing to subclinical infection or bacterial colonization. 1 Here, we present a case of difficult-to-treat asthma comorbid with DPB or protracted bacterial bronchiolitis that was resistant to macrolide therapy and resulted in cutaneous leukocytoclastic vasculitis (CLV). All conditions dramatically improved after the induction of Acapella®, an oscillating positive expiratory device, which facilitates airway clearance. A 21-year-old non-smoker woman who developed asthma at 5 years of age was treated with ICSs, LABAs, and leukotriene receptor antagonists until 18 years of age, when she suffered from pneumococcal pneumonia and acute sinusitis. These acute infections were successfully treated with antibiotics; however, productive cough and mild sinusitis persisted. Former doctors diagnosed the patient with co-occurrence of DPB based on the symptoms, signs, and chest computed tomography (CT) findings that satisfied the following three major criteria: persistent cough, sputum, and exertional dyspnea; past history of recurrent chronic sinusitis; bilateral diffuse small nodular shadows on a plain chest radiograph or centrilobular nodular shadows on the chest CT images; and at least two of the following three minor criteria: coarse crackles; FEV1/forced vital capacity (FVC)< 70% and partial pressure of arterial oxygen< 80 mmHg; cold agglutinin titer 64 as proposed by the Ministry of Health and Welfare of Japan (MHLW). 2 Laboratory results showed a white blood cell (WBC) count of 6030/μL (eosinophils 9.5%), C-reactive protein (CRP) level of 1.5 mg/dL, total IgE of 721 IU/mL with multiple sensitization to inhaled allergens, including house dust mites, cedar, Aspergillus, and Alternaria, titer of cold agglutinin of 128, and negative result for anti-neutrophil cytoplasmic autoantibodies. Pulmonary function tests revealed FEV1 of 2.22 L (72% of predicted) and FVC of 2.81 L (81% of predicted). Pseudomonas aeruginosa was cultured, and prominent neutrophilic inflammation was observed in the sputum. Low-dose macrolide therapy with erythromycin (EM) or clarithromycin, which had been prescribed to the patient from 18 years of age, failed to improve her symptoms. However, omalizumab slightly reduced productive cough. At 20 years of age, palpable purpura and edema appeared with pain in both legs, leading to the diagnosis of CLV based on skin biopsy.