Autopsy and Case Reports
https://www.autopsyandcasereports.org/article/doi/10.4322/acr.2018.022
Autopsy and Case Reports
Article / Autopsy Case Report

Congenital pulmonary airway malformation

Wlamir Pestana Ursini; Cesar Cilento Ponce

Downloads: 7
Views: 1845

Abstract

Congenital pulmonary airway malformation (CPAM) is a developmental malformation of the lower respiratory tract. We report the case of a male newborn diagnosed with cystic lung disease during prenatal ultrasound. A cesarean section was performed at the 32nd gestational week because of premature rupture of the membranes, and soon after the delivery the newborn developed respiratory failure and died. The aim of this study is to report an autopsy case because of its rarity, and to briefly discuss the CPAM subtypes and differential diagnosis of cystic lung diseases of childhood.

Keywords

Cystic Adenomatoid Malformation of Lung, Congenital, Respiratory Distress Syndrome, Newborn, Autopsy

References

Andrade CF , Ferreira HPC , Fischer GB . Congenital lung malformations . J Bras Pneumol . 2011 ; 37 ( 2 ): 259 - 71 . [https://doi.org/10.1590/S1806-37132011000200017] . [PMID:21537663]

Reis AR , Ribeiro FB , Schultz R . Congenital cystic adenomatoid malformation type I . Autops Case Rep . 2015 ; 5 ( 3 ): 21 - 6 . [https://doi.org/10.4322/acr.2015.019] . [PMID:26558243]

Chong Y , Rhee YJ , Han SJ , Cho HJ , Kang SK , Kang MW . Life-threatening congenital cystic adenomatoid malformation in the premature neonate . Korean J Thorac Cardiovasc Surg . 2016 ; 49 ( 3 ): 210 - 3 . [https://doi.org/10.5090/kjtcs.2016.49.3.210] . [PMID:27298802]

Sfakianaki AK , Copel JA . Congenital cystic lesions of the lung: congenital cystic adenomatoid malformation and bronchopulmonary sequestration . Rev Obstet Gynecol . 2012 ; 5 ( 2 ): 85 - 93 . [PMID:22866187]

David M , Lamas-Pinheiro R , Henriques-Coelho T . Prenatal and postnatal management of congenital pulmonary airway malformation . Neonatology . 2016 ; 110 ( 2 ): 101 - 15 . [https://doi.org/10.1159/000440894] .

Bolde S , Pudale S , Pandit G , Ruikar K , Ingle SB . Congenital pulmonary airway malformation: A report of two cases . World J Clin Cases . 2015 ; 3 ( 5 ): 470 - 3 . [https://doi.org/10.12998/wjcc.v3.i5.470] . [PMID:25984523]

Ch’in KY , Tang MY . Congenital adenomatoid malformation of one lobe of a lung with general anasarca . Arch Pathol (Chic) . 1949 ; 48 ( 3 ): 221 - 9 . [PMID:18137795]

Giubergia V , Barrenechea M , Siminovich M , Pena HG , Murtagh P . Congenital cystic adenomatoid malformation: clinical features, pathological concepts and management in 172 cases . J Pediatr (Rio J) . 2012 ; 88 ( 2 ): 143 - 8 . [https://doi.org/10.2223/JPED.2177] . [PMID:22543527]

Stocker JT , Madewell JE , Drake RM . Congenital cystic adenomatoid malformation of the lung. Classification and morphologic spectrum . Hum Pathol . 1977 ; 8 ( 2 ): 155 - 71 . [https://doi.org/10.1016/S0046-8177(77)80078-6] . [PMID:856714]

Durell J , Lakhoo K . Congenital cystic lesions of the lung . Early Hum Dev . 2014 ; 90 ( 12 ): 935 - 9 . [https://doi.org/10.1016/j.earlhumdev.2014.09.014] . [PMID:25448785]

Cataneo DC , Rodrigues OR , Hasimoto EN , Schmidt AF Jr , Cataneo AJ . Congenital lobar emphysema: 30-year case series in two university hospitals . J Bras Pneumol . 2013 ; 39 ( 4 ): 418 - 26 . [https://doi.org/10.1590/S1806-37132013000400004] . [PMID:24068262]

Biyyam DR , Chapman T , Ferguson MR , Deutsch G , Dighe MK . Congenital lung abnormalities: embryologic features, prenatal diagnosis, and postnatal radiologic-pathologic correlation . Radiographics . 2010 ; 30 ( 6 ): 1721 - 38 . [https://doi.org/10.1148/rg.306105508] . [PMID:21071385]

Mirra V , Werner C , Santamaria F . Primary ciliary dyskinesia: an update on clinical aspects, genetics, diagnosis, and future treatment strategies . Front Pediatr . 2017 ; 5 : 135 . [https://doi.org/10.3389/fped.2017.00135] . [PMID:28649564]

Knowles MR , Daniels LA , Davis SD , Zariwala MA , Leigh MW . Primary ciliary dyskinesia. Recent advances in diagnostics, genetics, and characterization of clinical disease . Am J Respir Crit Care Med . 2013 ; 188 ( 8 ): 913 - 22 . [https://doi.org/10.1164/rccm.201301-0059CI] . [PMID:23796196]

Jain M , Goss CH . Update in cystic fibrosis 2013 . Am J Respir Crit Care Med . 2014 ; 189 ( 10 ): 1181 - 6 . [https://doi.org/10.1164/rccm.201402-0203UP] . [PMID:24832742]

Williams H , Campbell P . Generalized bronchiectasis associated with deficiency of cartilage in the bronchial tree . Arch Dis Child . 1960 ; 35 ( 180 ): 182 - 91 . [https://doi.org/10.1136/adc.35.180.182] . [PMID:13844857]

Wayne KS , Taussig LM . Probable familial congenital bronchiectasis due to cartilage deficiency (Williams–Campbell syndrome) . Am Rev Respir Dis . 1976 ; 114 ( 1 ): 15 - 22 . [PMID:937831]

 

 

 

 

 

 

 


Publication date:
05/02/2018

5aea08b10e88254f5ca00810 autopsy Articles
Links & Downloads

Autops Case Rep

Share this page
Page Sections