Background <p>Congenital pulmonary airway malformation (CPAM) and bronchopulmonary sequestration (BPS) are rare congenital lung malformations that occur independently or as hybrid lesions. While typically diagnosed in childhood, their occurrence in adults is uncommon.</p> Case presentation <p>We report a 25-year-old female with chest pain and hemoptysis. Imaging studies revealed multiloculated cystic lesions in the right lower lobe with an anomalous arterial supply, consistent with a hybrid CPAM-BPS. A multidisciplinary approach was employed, including preoperative embolization of the aberrant artery followed by thoracoscopic lobectomy. There were no complications, and histopathology confirmed type 2 CPAM with intralobar sequestration. The patient remained asymptomatic at a six-month follow-up. To our knowledge, this is one of the first documented adult cases with hybrid CPAM-BPS lesions who underwent preoperative arterial embolization followed by thoracoscopic lobectomy.</p> Conclusions <p>Hybrid CCAM-BPS is a scarce pathology in adults, and few cases have been reported. A high index of suspicion is indicated to diagnose such rare lesions. A combination of preoperative endovascular embolization and thoracoscopic lobectomy is a safe and feasible treatment approach.</p>

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Hybrid Congenital Pulmonary Airway Malformation (CPAM) and Bronchopulmonary Sequestration (BPS) in an adult female: a combined surgical approach of a rare case

  • Maha Z. Alamri,
  • Ahmed G. Elkhouly,
  • Adeeb Bazuhair,
  • Mohammed Saleh Ishaq

摘要

Background

Congenital pulmonary airway malformation (CPAM) and bronchopulmonary sequestration (BPS) are rare congenital lung malformations that occur independently or as hybrid lesions. While typically diagnosed in childhood, their occurrence in adults is uncommon.

Case presentation

We report a 25-year-old female with chest pain and hemoptysis. Imaging studies revealed multiloculated cystic lesions in the right lower lobe with an anomalous arterial supply, consistent with a hybrid CPAM-BPS. A multidisciplinary approach was employed, including preoperative embolization of the aberrant artery followed by thoracoscopic lobectomy. There were no complications, and histopathology confirmed type 2 CPAM with intralobar sequestration. The patient remained asymptomatic at a six-month follow-up. To our knowledge, this is one of the first documented adult cases with hybrid CPAM-BPS lesions who underwent preoperative arterial embolization followed by thoracoscopic lobectomy.

Conclusions

Hybrid CCAM-BPS is a scarce pathology in adults, and few cases have been reported. A high index of suspicion is indicated to diagnose such rare lesions. A combination of preoperative endovascular embolization and thoracoscopic lobectomy is a safe and feasible treatment approach.