Logo image
A case report of Birt-Hogg-Dubé syndrome associated with severe airway obstruction in a 62-year-old female smoker
Journal article   Peer reviewed

A case report of Birt-Hogg-Dubé syndrome associated with severe airway obstruction in a 62-year-old female smoker

Kiyoshi Uemasu and Toru Nyunoya
Respirology case reports, Vol.12(3), pp.e01327-n/a
03/2024
DOI: 10.1002/rcr2.1327
PMID: 38523622
url
https://doi.org/10.1002/rcr2.1327View
Published (Version of record) Open Access

Abstract

Birt-Hogg-Dubé syndrome (BHD) typically does not manifest airway obstruction despite the presence of multiple lung cysts. However, the long-term effects of cigarette smoking on lung function among individuals with BHD are unknown. We report a case of a smoking individual diagnosed with BHD syndrome complicated by spontaneous pneumothorax and severe airway obstruction. The patient presented with chronic dyspnea and productive cough. Further work-up revealed severe obstructive airflow limitation, and multiple lung cysts in both lungs, accompanied centrilobular emphysematous changes. Genetic testing confirmed a heterozygous deletion of exons 6-8 in the gene, confirming the diagnosis of BHD.
centrilobular emphysema paraseptal emphysema folliculin chronic obstructive disease Birt–Hogg–Dubé syndrome

Details

Metrics

Logo image