Hosoki Koa, Fujisawa Takao, Masuda Sawako, Usui Satoko, Ito Hiroaki, Nagao Mizuho, Terada Akihiko, Iguchi Kousei, Ogawa Satoru, Nakatani Kaname, Takeuchi Kazuhiko
Department of Pediatrics, Mie National Hospital and Electron Microscopy Research Center, Mie University.
Arerugi. 2010 Jul;59(7):847-54.
We report a case of 18-old girl with primary ciliary dyskinesia (PCD) who had been diagnosed as asthma. Since birth, she had presented with unexplained productive cough, sputum, rhinorrhea, and stridor with situs solitus. Her familial history was negative for PCD. At 2 years of age, ciliary beat frequency and beat pattern were normal. She was diagnosed as rhinosinusitis, chronic secretory otitis media. At 3 years of age, she was diagnosed as asthma because of wheezing not associated with respiratory infection. Various asthma medications were then administered, including DSCG, inhaled corticosteroids, and salmeterol, but varying responses to the treatment were noted. Spirometry revealed persistent severe small airway obstruction. Beta2 agonist reversibility was recognized by impulse oscillation system, not with FEV1. At age of 18, chest CT disclosed bronchiectasis, and nasal nitric oxide concentration was very low, 98 ppb and a diagnostic approach for PCD was performed. Electron microscopic analysis of nasal cilia demonstrated defects of the outer and inner dynein arms, and the diagnosis of PCD was made. Mutations in DNAH1 and DNAI1 genes were found. The diagnosis of PCD is often difficult in the absence of situs inversus totalis. Recurrent wheeze with chronic rhinosinusitis, chronic otitis media, and brochiectasis may warrant detailed investigations for PCD, especially with nasal NO measurement.
我们报告一例18岁患有原发性纤毛运动障碍(PCD)的女孩,她曾被诊断为哮喘。自出生以来,她一直有不明原因的咳痰、流涕、鼻漏和喘鸣,内脏位置正常。她的家族史中无PCD。2岁时,纤毛摆动频率和摆动模式正常。她被诊断为鼻窦炎、慢性分泌性中耳炎。3岁时,因喘息与呼吸道感染无关而被诊断为哮喘。随后使用了各种哮喘药物,包括色甘酸钠、吸入性皮质类固醇和沙美特罗,但对治疗的反应各不相同。肺功能检查显示持续严重的小气道阻塞。β2激动剂的可逆性通过脉冲振荡系统得以确认,而非通过第一秒用力呼气容积(FEV1)。18岁时,胸部CT显示支气管扩张,鼻一氧化氮浓度非常低,为98 ppb,并进行了PCD的诊断方法。鼻纤毛的电子显微镜分析显示外动力臂和内动力臂存在缺陷,从而做出了PCD的诊断。发现了DNAH1和DNAI1基因的突变。在没有完全性内脏反位的情况下,PCD的诊断往往很困难。伴有慢性鼻窦炎、慢性中耳炎和支气管扩张的反复喘息可能需要对PCD进行详细检查,尤其是通过测量鼻一氧化氮。