Evaluation of Newborn Infants with Prenatally Diagnosed Congenital Pulmonary Airway Malformation: A Single-Center Experience
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Purpose: Congenital pulmonary airway malformation (CPAM)－a rare developmental anomaly－affects the lower respiratory tract in newborns. By comparing the reliability of diagnostic tools and identifying predictive factors for symptoms, we provide comprehensive clinical data for the proper management of CPAM. Methods: We reviewed the medical records of 66 patients with prenatally diagnosed CPAM delivered at Severance Children’s Hospital between January 2005 and July 2017. Results: We enrolled 33 boys and 33 girls. Their mean gestational age and birth weight were 38.8 weeks and 3,050 g, respectively. Prenatal ultrasonography and postnatal radiography, lung ultrasonography, and chest computed tomography (CT) showed inconsistent findings. Chest CT showed superior sensitivity (100%) and positive predictive value (90%). Among the 66 patients, 59 had postnatally confirmed CPAM, three had pulmonary sequestration, one had cystic teratoma, and one had a normal lung. Of the 59 patients with CPAM, 21 (35%; mean age, 23.4 months) underwent surgery, including 15 who underwent video-assisted thoracoscopy. Twenty-five and 12 patients exhibited respiratory symptoms at birth and during infancy, respectively. Apgar scores and mediastinal shift on radiography were significantly associated with respiratory symptoms at birth. However, none of the factors could predict respiratory symptoms during infancy. Conclusion: Radiography or ultrasonography combined with chest CT can confirm an unclear or inconsistent lesion. Apgar scores and mediastinal shift on radiography can predict respiratory symptoms at birth. However, symptoms during infancy are not associated with prenatal and postnatal factors. Chest CT combined with periodic symptom monitoring is important for diagnosing and managing patients with prenatally diagnosed CPAM and to guide appropriate timing of surgery.
Abstract
Purpose: Congenital pulmonary airway malformation (CPAM)－a rare developmental anomaly－affects the lower respiratory tract in newborns. By comparing the reliability of diagnostic tools and identifying predictive factors for symptoms, we provide comprehensive clinical data for the proper management of CPAM. Methods: We reviewed the medical records of 66 patients with prenatally diagnosed CPAM delivered at Severance Children’s Hospital between January 2005 and July 2017. Results: We enrolled 33 boys and 33 girls. Their mean gestational age and birth weight were 38.8 weeks and 3,050 g, respectively. Prenatal ultrasonography and postnatal radiography, lung ultrasonography, and chest computed tomography (CT) showed inconsistent findings. Chest CT showed superior sensitivity (100%) and positive predictive value (90%). Among the 66 patients, 59 had postnatally confirmed CPAM, three had pulmonary sequestration, one had cystic teratoma, and one had a normal lung. Of the 59 patients with CPAM, 21 (35%; mean age, 23.4 months) underwent surgery, including 15 who underwent video-assisted thoracoscopy. Twenty-five and 12 patients exhibited respiratory symptoms at birth and during infancy, respectively. Apgar scores and mediastinal shift on radiography were significantly associated with respiratory symptoms at birth. However, none of the factors could predict respiratory symptoms during infancy. Conclusion: Radiography or ultrasonography combined with chest CT can confirm an unclear or inconsistent lesion. Apgar scores and mediastinal shift on radiography can predict respiratory symptoms at birth. However, symptoms during infancy are not associated with prenatal and postnatal factors. Chest CT combined with periodic symptom monitoring is important for diagnosing and managing patients with prenatally diagnosed CPAM and to guide appropriate timing of surgery.
참고문헌 (26)
- Kulwa E, "Congenital cystic adenomatoid malformation in the fetus: a hypothesis of its development", Fetal Diagn Ther, 20, pp.472~474 (2005)
- Laje P, "Postnatal management and outcome of prenatally diagnosed lung lesions", Prenat Diagn, 28, pp.612~618 (2008)
- 서영은, "조기수술이 필요한 폐 선천성 낭포성 샘 모양 기형아의 특성과 수술 전 중재적 시술", Neonatal medicine, 20(1), pp.81~89 (2013), 대한신생아학회
- Stanton M., "The argument for a non-operative approach to asymptomatic lung lesions", Semin Pediatr Surg, 24, pp.183~186 (2015)
- Stocker JT, "Congenital cystic adenomatoid malformation of the lung. Classification and morphologic spectrum", Hum Pathol, 8, pp.155~171 (1977)
- Xia B, "Surgical treatment of congenital cystic adenomatoid malformation: a retrospective study of single tertiary center experience", J Matern Fetal Neonatal Med, 30, pp.416~419 (2017)
- Leblanc C, "Congenital pulmonary airway malformations: stateof-the-art review for pediatrician's use", Eur J Pediatr, 176, pp.1559~1571 (2017)
- Turan O, "Prenatally detected congenital cystic adenomatoid malformation and postnatally diagnosed trisomy 13: case report and review of the literature", Turk J Pediatr, 53, pp.337~341 (2011)
- Kitaichi M, Yousem S., "Symposium 24: non-neoplastic lung disease", Histopathology, 41(Suppl 2), pp.424~458 (2002)
- Thakkar HS, "Antenatally detected congenital pulmonary airway malformations: the Oxford experience", Eur J Pediatr Surg, 27, pp.324~329 (2017)
- Shanmugam G, "Congenital lung malformations:antenatal and postnatal evaluation and management", Eur J Cardiothorac Surg, 27, pp.45~52 (2005)
- Hardee S, "Congenital cystic lung lesions: evolution from in-utero detection to pathology diagnosis. A multidisciplinary approach", Pediatr Dev Pathol, 20, pp.403~410 (2017)
- Pacharn P, "Congenital lung lesions: prenatal MRI and postnatal findings", Pediatr Radiol, 43, pp.1136~1143 (2013)
- Ruchonnet-Metrailler I, "Neonatal outcomes of prenatally diagnosed congenital pulmonary malformations", Pediatrics, 133, pp.e1285~e1291 (2014)
- Gallardo AM, "Antenatal ultrasound diagnosis and neonatal results of the congenital cystic adenomatoid malformation of the lung", Rev Chil Pediatr, 89, pp.224~230 (2018)
- Shamas AG, "Congenital cystic adenomatoid malformation of the lung (CCAM), a retrospective clinical audit and literature review in a tertiary centre in Scotland over a period of 14 years", J Obstet Gynaecol, 37, pp.19~24 (2017)
- Crombleholme TM, "Cystic adenomatoid malformation volume ratio predicts outcome in prenatally diagnosed cystic adenomatoid malformation of the lung", J Pediatr Surg, 37, pp.331~338 (2002)
- Shirota C, "Usefulness of fetal magnetic resonance imaging for postnatal management of congenital lung cysts: prediction of probability for emergency surgery", BMC Pediatr, 18, pp.105~ (2018)
- Kantor N, "Symptom development in originally asymptomatic CPAM diagnosed prenatally: a systematic review", Pediatr Surg Int, 34, pp.613~620 (2018)
- Morini F, "Erratum to: current management of congenital pulmonary airway malformations: a "European Pediatric Surgeons' Association"Survey", Eur J Pediatr Surg, 28, pp.e1~ (2018)
- Derderian SC, "Favorable outcomes in high-risk congenital pulmonary airway malformations treated with multiple courses of maternal betamethasone", J Pediatr Surg, 50, pp.515~518 (2015)
- Kapralik J, "Surgical versus conservative management of congenital pulmonary airway malformation in children: a systematic review and meta-analysis", J Pediatr Surg, 51, pp.508~512 (2016)
- Polites SF, "Thoracoscopic vs open resection of congenital cystic lung disease: utilization and outcomes in 1120 children in the United States", J Pediatr Surg, 51, pp.1101~1105 (2016)
- Rothenberg SS, "Two decades of experience with thoracoscopic lobectomy in infants and children: standardizing techniques for advanced thoracoscopic surgery", J Laparoendosc Adv Surg Tech A, 25, pp.423~428 (2015)
- Ghosh M, "Pleuropulmonary blastoma developing in a case of misinterpreted congenital pulmonary airway malformation: a case report", Fetal Pediatr Pathol, 37, pp.377~386 (2018)
- Delacourt C, "Shall all congenital cystic lung malformations be removed? the case in favour", Paediatr Respir Rev, 14, pp.169~170 (2013)