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

Kiyoshi UemasuToru Nyunoya
Published in: Respirology case reports (2024)
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 folliculin gene, confirming the diagnosis of BHD.
Keyphrases
  • lung function
  • early onset
  • case report
  • chronic obstructive pulmonary disease
  • air pollution
  • drug induced
  • gene expression
  • single cell
  • dna methylation
  • transcription factor
  • palliative care
  • thoracic surgery