Cynthia D Downard1, Casey M Calkins2, Regan F Williams3, Elizabeth J Renaud4, Tim Jancelewicz3, Julia Grabowski5, Roshni Dasgupta6, Milissa McKee7, Robert Baird8, Mary T Austin9, Meghan A Arnold10, Adam B Goldin11, Julia Shelton12, Saleem Islam13. 1. Division of Pediatric Surgery, Hiram C. Polk, Jr., MD Department of Surgery, University of Louisville, 315 East Broadway, Suite 565, Louisville, KY, 40202, USA. C0down01@louisville.edu. 2. Division of Pediatric General and Thoracic Surgery, Department of Surgery, The Medical College of Wisconsin, Milwaukee, WI, USA. 3. Division of Pediatric Surgery, Department of Surgery, University of Tennessee Health Science Center, Memphis, TN, USA. 4. Division of Pediatric Surgery, Department of Surgery, Albany Medical Center, Albany, NY, USA. 5. Division of Pediatric Surgery, Department of Surgery, Ann and Robert Lurie Children's Hospital of Chicago, Chicago, IL, USA. 6. Division of Pediatric General and Thoracic Surgery, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, USA. 7. Pediatric Surgery, Phoenix, AZ, USA. 8. Division of Pediatric General and Thoracic Surgery, McGill University Health Center, Montreal Children's Hospital, Glen Site, Montreal, QC, Canada. 9. Departments of Surgical Oncology and Division of Pediatrics, The University of Texas MD Anderson Cancer Center, Houston, TX, USA. 10. Division of Pediatric Surgery, Department of Surgery, University of Michigan CS Mott Children's Hospital, Ann Arbor, MI, USA. 11. Division of Pediatric General and Thoracic Surgery, Seattle Children's Hospital, University of Washington School of Medicine, Seattle, WA, USA. 12. Division of Pediatric Surgery, Department of Surgery, University of Iowa Hospitals and Clinics, Iowa City, IA, USA. 13. Division of Pediatric Surgery, University of Florida, Gainesville, FL, USA.
Abstract
PURPOSE: Variation in management characterizes treatment of infants with a congenital pulmonary airway malformation (CPAM). This review addresses six clinically applicable questions using available evidence to provide recommendations for the treatment of these patients. METHODS: Questions regarding the management of a pediatric patient with a CPAM were generated. English language articles published between 1960 and 2014 were compiled after searching Medline and OvidSP. The articles were divided by subject area and by the question asked, then reviewed and included if they specifically addressed the proposed question. RESULTS: 1040 articles were identified on initial search. After screening abstracts per eligibility criteria, 130 articles were used to answer the proposed questions. Based on the available literature, resection of an asymptomatic CPAM is controversial, and when performed is usually completed within the first six months of life. Lobectomy remains the standard resection method for CPAM, and can be performed thoracoscopically or via thoracotomy. There is no consensus regarding a monitoring protocol for observing asymptomatic lesions, although at least one chest computerized tomogram (CT) should be performed postnatally for lesion characterization. An antenatally identified CPAM can be evaluated with MRI if fetal intervention is being considered, but is not required for the fetus with a lesion not at risk for hydrops. Prenatal consultation should be offered for infants with CPAM and encouraged for those infants in whom characteristics indicate risk of hydrops. CONCLUSIONS: Very few articles provided definitive recommendations for care of the patient with a CPAM and none reported Level I or II evidence. Based on available information, CPAMs are usually resected early in life if at all. A prenatally diagnosed congenital lung lesion should be evaluated postnatally with CT, and prenatal counseling should be undertaken in patients at risk for hydrops.
PURPOSE: Variation in management characterizes treatment of infants with a congenital pulmonary airway malformation (CPAM). This review addresses six clinically applicable questions using available evidence to provide recommendations for the treatment of these patients. METHODS: Questions regarding the management of a pediatric patient with a CPAM were generated. English language articles published between 1960 and 2014 were compiled after searching Medline and OvidSP. The articles were divided by subject area and by the question asked, then reviewed and included if they specifically addressed the proposed question. RESULTS: 1040 articles were identified on initial search. After screening abstracts per eligibility criteria, 130 articles were used to answer the proposed questions. Based on the available literature, resection of an asymptomatic CPAM is controversial, and when performed is usually completed within the first six months of life. Lobectomy remains the standard resection method for CPAM, and can be performed thoracoscopically or via thoracotomy. There is no consensus regarding a monitoring protocol for observing asymptomatic lesions, although at least one chest computerized tomogram (CT) should be performed postnatally for lesion characterization. An antenatally identified CPAM can be evaluated with MRI if fetal intervention is being considered, but is not required for the fetus with a lesion not at risk for hydrops. Prenatal consultation should be offered for infants with CPAM and encouraged for those infants in whom characteristics indicate risk of hydrops. CONCLUSIONS: Very few articles provided definitive recommendations for care of the patient with a CPAM and none reported Level I or II evidence. Based on available information, CPAMs are usually resected early in life if at all. A prenatally diagnosed congenital lung lesion should be evaluated postnatally with CT, and prenatal counseling should be undertaken in patients at risk for hydrops.
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