Congenital pulmonary airway malformation and bronchopulmonary sequestration in infants - a rare diagnosis

Authors

  • Joana Cachão Department of Pediatrics, Hospital de São Bernardo, Centro Hospitalar de Setúbal
  • Isabel Raminhos Department of Pediatrics, Hospital de São Bernardo, Centro Hospitalar de Setúbal
  • José D. Martins Department of Pediatrics Cardiology, Hospital de Santa Marta, Centro Hospitalar de Lisboa Central
  • Ivan Bravio Department of Cardiothoracic Surgery. Hospital de Santa Marta, Centro Hospitalar de Lisboa Central

DOI:

https://doi.org/10.25753/BirthGrowthMJ.v29.i4.18079

Keywords:

bronchopulmonary sequestration, congenital cystic adenomatoid malformation, infant

Abstract

Herein is described the case of a two-month-old female infant with recurrent respiratory infections, admitted to the Pediatric Emergency Department with productive cough, respiratory distress, and fever. Chest radiography showed an heterogenous hypotransparent image in the left pulmonary field and contralateral deviation of the mediastinum.
Angiotomography revealed bilateral bronchopulmonary sequestration and a systemic vessel contributing to area irrigation. The patient was submitted to vessel occlusion and lacquering, left inferior lobectomy, and resection of a right lower lobe segment. Histological examination confirmed intralobar bronchopulmonary sequestration and type 2 congenital pulmonary airway malformation in the left lung, and extralobar bronchopulmonary sequestration in the right lung.
With this report, the authors intend to emphasize the importance of clinical suspicion of congenital anomalies in infants with recurrent respiratory infections.

Downloads

Download data is not yet available.

References

Andrade CF, Ferreira HPC, Fischer GB. Malformações pulmonares congênitas. J Bras Pneumol. 2011; 37:259-71.

Ursini WP, Ponce CC. Congenital pulmonary airway malformation. Autops Case Rep. 2018; 8:1-6.

Pryce DM. Lower accessory pulmonary artery with intralobar sequestration of lung. J Pathol Bacteriol. 1946; 58:457-67.

Watanabe S, Suzuki T, Hara F, Yasui T, Uga N, Naoe A. Intralobar pulmonary sequestration complicated by gastric duplication. Journal of Pediatric Surgery Case Reports. 2017; 17:42-5.

Sousa A, Costa J, Silva LJ. Sequestro pulmonar com diagnóstico pré-natal. Caso clínico. Acta Pediatr Port. 2007; 38:117-9.

Ch’in KY, Tang MY. Congenital adenomatoid malformation of one lobe of a lung with general anasarca. 1949; 48:221-9.

Leblanc C, Baron M, Desselas E, Phan MH, Rybak A, Thouvenin G, et al. Congenital pulmonary airway malformations: state-of-the-art review for pediatrician’s use. Eur J Pediatr. 2017; 176:1559-71.

Stocker JT, Madewell JE, Drake RM. Congenital cystic adenomatoid malformation of the lung. Classification and morphologic spectrum. Hum Pathol. 1977; 8:155-71.

Sfakianaki AK, Copel JA. Congenital Cystic Lesions of the Lung: Congenital Cystic Adenomatoid Malformation and Bronchopulmonary Sequestration. Rev Obstet Gynecol. 2012; 5:85-93.

Wong KKY, Flake AW, Tibboel D, Rottier RJ, Tam PKH. Congenital pulmonary airway malformation: advances and controversies. Lancet Child Adolesc Health. 2018; 2:290-7.

Hung JH, Shen SH, Guo WY, Chen CY, Chao KC, Yang MJ, et al. Prenatal Diagnosis of Pulmonary Sequestration by Ultrasound and Magnetic Resonance Imaging. J Chin Med Assoc. 2008; 71:53-7.

Bolde S, Pudale S, Pandit G, Ruikar K, Ingle SB. Congenital pulmonary airway malformation: A report of two cases. World J Clin Cases. 2015; 3:470-3.

Couluris M, Schnapf BM, Barness EG. Intralobar pulmonary sequestration associated with a congenital pulmonary airway malformation type II. Fetal and Pediatric Pathology. 2007; 26:207-12.

Malik R, Pandya VK, Agrawal G, Jain M. Congenital Cystic Adenomatoid Malformation: A Case Report. Ind J Radiol Imag. 2006; 16:859-60.

Slood M. Congenital Cystic Adenomatoid Malformation of the Lung: A Case Report. J Nepal Paediatr Soc. 2011; 31:64-7.

Downloads

Published

2020-11-26

How to Cite

1.
Cachão J, Raminhos I, Martins JD, Bravio I. Congenital pulmonary airway malformation and bronchopulmonary sequestration in infants - a rare diagnosis. REVNEC [Internet]. 2020Nov.26 [cited 2024Mar.29];29(4):200-3. Available from: https://revistas.rcaap.pt/nascercrescer/article/view/18079

Issue

Section

Case Reports

Most read articles by the same author(s)