Case series of hybrid lung lesion combining Congenital Pulmonary Airway Malformation (CPAM) and pulmonary Sequestration (PS), with Bilateral Pulmonary Sequestration and Literature Review

Introduction: Bilateral Pulmonary Sequestration (PS) is an exceptionally uncommon variant, characterized by non-functioning lung tissue supplied by systemic arteries on both sides, Hybrid lesions combining PS and Congenital Pulmonary Airway Malformation (CPAM) represent a complex subset of these anomalies.

Objective: A case report of CPAM, with bilateral PS and a literature review of hybrid lung lesion, bilateral PS.

Method: The first case report in the Middle East of CPAM and Bilateral PS.

Case Report: A 3-year-old boy medically free, presented with persistent fever despite received antibiotics and unremarkable physical examination, a chest x-ray showed a picture of necrotizing pneumonia treated with dual antimicrobials, With persistent radiological abnormalities, extensive workup was done with a contrast CT chest showed multiple cystic lesions with variable densities on the left side with blood supply coming from the lower descending aorta,  represent a hybrid condition as CPAM and extra lobar PS, there was another blood supply going from the descending thoracic to the right side, which represents an intrathoracic PS or arteriovenous malformation type. He underwent surgical resection of the left lower lobe lobectomy.

There is only one published case report of a similar presentation of a 2-year-old case of bilateral pulmonary varices associated with hybrid lung lesion PS and CPAM, While bilateral PS was reported in 36 cases.

Conclusion:  Understanding these complex congenital anomalies is crucial for appropriate patient care and surgical planning. Management approach ranges from conservative to surgical intervention, emphasizing the need for a multidisciplinary approach in diagnosis and treatment. Recognizing these unusual presentations is crucial for appropriate patient care and surgical planning in both pediatric and adult populations.

Dr. Hanaa Banjar, is a Professor of Pediatrics at Alfaisal University since 2013, Riyadh. She is also A consultant pediatric Pulmonology and the Director of Pediatric Pulmonary fellowship program at King Faisal Specialist Hospital and research Center, Riyadh, Saudi Arabia.

Dr. Banjar received her Residency in Pediatrics from The University of Ottawa, Ontario, Canada and her Pulmonary Fellowship from McGill University, Montreal, Quebec, Canada.

As an expert in pediatric pulmonary medicine, especially in cystic fibrosis, Pulmonary Hypertension, and Non-CF Bronchiectasis, she serves as the principle investigator of the cystic Fibrosis registry of up to 400 patients and the principle author of 90 articles.