Aspergillus tracheobronchitis with Birt-Hogg-Dubè syndrome as a rare cause of chronic cough

Background 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. Case presentation: Herein, we presented a 45-year-old male pa...

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Bibliographic Details
Published in:BMC pulmonary medicine 2022-11, Vol.22 (1), p.1-420, Article 420
Main Authors: Huang, Lianrong, Yi, Fang, Zhan, Chen, Lai, Kefang
Format: Article
Language:English
Subjects:
Antibiotics
Aspergillus
Aspergillus tracheobronchitis
Asthma
Biopsy
Birt-Hogg-Dube syndrome
Birt-Hogg-Dubé syndrome
Bronchitis
Care and treatment
Case Report
Case reports
Chronic cough
Chronic infection
Computed tomography
Cough
Cystic fibrosis
Cysts
Diagnosis
Disease
Dyspnea
Etiology
Family medical history
Fungi
Gastroesophageal reflux
Genetic screening
Infections
Itraconazole
Mediastinum
Metagenomics
Multiple lung cysts
Mutation
Next-generation sequencing
Patients
Pulmonology
Sputum
Tracheobronchitis
Tumor suppressor genes
Tumors
Citations: Items that this one cites
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Summary:Background 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. Case presentation: 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. Conclusion 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. Keywords: Chronic cough, Multiple lung cysts, Aspergillus tracheobronchitis, Birt-Hogg-Dubé syndrome, Case report
ISSN:1471-2466
1471-2466
DOI:10.1186/s12890-022-02136-2