%0 Case Reports
%T Aspergillus tracheobronchitis with Birt-Hogg-Dubè syndrome as a rare cause of chronic cough.
%A Huang L
%A Yi F
%A Zhan C
%A Lai K
%J BMC Pulm Med
%V 22
%N 1
%D Nov 2022 16
%M 36384555
%F 3.32
%R 10.1186/s12890-022-02136-2
%X <B>BACKGROUND: </B>Aspergillus tracheobronchitis (ATB) is confined as a condition of chronic superficial infection of tracheobronchial tree. Its diagnosis is difficult due to atypical manifestations and low detective rate of Aspergillus thus far.<BR><B>METHODS: </B>Herein, we presented a 45-year-old male patient with a sole chronic productive cough for five years referred to our cough specialist clinic. Chest high-resolution computed tomography showed multiple lung cysts predominantly located in the subpleural lesions and near the mediastinum. Neither bacteria nor fungi were identified by sputum culture. However, metagenomic next-generation sequencing in sputum detected Aspergillus fumigatus DNA. The genetic testing of whole blood suggested the germline mutation of the tumor suppressor gene folliculin, supporting a diagnosis of Birt-Hogg-Dubé (BHD) syndrome. His productive cough symptom significantly improved after receiving itraconazole treatment for 2 months. After discontinuation of antifungal treatment, there was no relapse for four months follow-up. A diagnosis of ATB with BHD syndrome was eventually established in this patient.<BR><B>CONCLUSIONS: </B>ATB should be considered in any patient with prolonged unexplained productive cough. Next-generation sequencing technologies may be useful to identify ATB which is uncommon and easily ignored in clinical practice.