Congenital pulmonary
adenomatoid malformation (CPAM) is a congenital lung malformation that is
distinguished by abnormal airway patterning during branching morphogenesis. It
may lead to significant morbidity and mortality in infants due to complications
such as pulmonary infections, lung hypoplasia, respiratory distress, and fetal
hydrops. The diagnosis is usually prenatal thanks to morphological ultrasound;
in postnatal, the anomaly may remain asymptomatic or show respiratory signs or
complications. The postnatal diagnosis is based on computed tomography. In this
article, we report a case of congenital pulmonary
adenomatoid malformation in a 7-month-old patient who presented respiratory
distress that is due to voluminous infected pleural effusion. The diagnosis of
CPAM was suspected at the initial CT and confirmed at the control CT that was
realized after treatment of the infection. The therapeutic management of CPAM is
depending on the severity of complications and its evolution.
References
[1]
Hansell, D.M., Bankier, A.A., MacMahon, H., et al. (2008) Fleischner Society: Glossary of Terms for Thoracic Imaging. Radiology, 246, 697-722.
https://doi.org/10.1148/radiol.2462070712
[2]
Azizkhan, R.G. and Crombleholme, T.M. (2008) Congenital Cystic Lung Disease: Contemporary Antenatal and Postnatal Management. Pediatric Surgery International, 24, 643-657. https://doi.org/10.1007/s00383-008-2139-3
[3]
Stocker, J.T. (2002) Congenital Pulmonary Airway Malformation—A New Name for and an Expanded Classification of Congenital Cystic Adenomatoid Malformation of the Lung. Histopathology, 41, 424-430.
[4]
Stocker, J.T., Madewell, J.E. and Drake, R.M. (1977) Congenital Cystic Adenomatoid Malformation of the Lung: Classification and Morphologic Spectrum. Human Pathology, 8, 155-171. https://doi.org/10.1016/S0046-8177(77)80078-6
[5]
Volpe, M.V., Pham, L., Lessin, M., Ralston, S.J., et al. (2003) Expression of Hoxb-5 during Human Lung Development and in Congenital Lung Malformations. Birth Defects Research Part A: Clinical and Molecular Teratology, 67, 550-556.
https://doi.org/10.1002/bdra.10086
[6]
Liechty, K.W., Crombleholme, T., Quinn, T.M., Cass, D.L., et al. (1999) Elevated Platelet-Derived Growth Factor-B in Congenital Cystic Adenomatoid Malformations Requiring Fetal Resection. Journal of Pediatric Surgery, 34, 805-809.
https://doi.org/10.1016/S0022-3468(99)90377-9
[7]
Cloutier, M.M., Schaeffer, D.A. and Hight, D. (1993) Congenital Cystic Adenomatoid Malformation. Chest, 103, 761-764. https://doi.org/10.1378/chest.103.3.761
[8]
Bianchi, D.W., Crombleholme, T.M. and D’Alton, M.E. (2000) Cystic Adenomatoid Malformation. In: Fetology: Diagnosis and Management of the Fetal Patient, Chapter 37, McGraw-Hill, New York.
[9]
Pinkerton, H.J. and Oldham, K.T. (2005) Principles and Practice of Pediatric Surgery. Chapter 61, Volume 2, Springer, Berlin, Heidelberg.
[10]
Pacharn, P., Kline-Fath, B., Calvo-Garcia, M., Linam, L.E., Rubio, E.I., Salisbury, S. and Brody, A.S. (2013) Congenital Lung Lesions: Prenatal MRI and Postnatal Findings. Pediatric Radiology, 43, 1136-1143. https://doi.org/10.1007/s00247-013-2668-3
[11]
An, P., Wang, Y., Feng, W., et al. (2019) Congenital Cystic Adenomatoid Malformation Volume Ratio in Prenatal Assessment of Prognosis of Fetal Pulmonary Sequestrations. Current Medical Science, 39, 658-662.
https://doi.org/10.1007/s11596-019-2088-1
[12]
Annunziata, F., Bush, A., Borgia, F., et al. (2019) Congenital Lung Malformations: Unresolved Issues and Unanswered Questions. Frontiers in Pediatrics, 7, 239.
https://doi.org/10.3389/fped.2019.00239
[13]
Kapralik, J., Wayne, C., Chan, E. and Nasr, A. (2016) Surgical versus Conservative Management of Congenital Pulmonary Airway Malformation in Children: A Systematic Review and Meta-Analysis. Journal of Pediatric Surgery, 51, 508-512.
https://doi.org/10.1016/j.jpedsurg.2015.11.022
[14]
Frick, A.E., Decaluwe, H., Weynand, B., et al. (2019) Invasive Mucinous Adenocarcinoma of the Lung Arising in a Type1 Congenital Pulmonary Airway Malformation in a 68-Year-Old Patient: A Case Report. Acta Chirurgica Belgica, 2, 1-6.
