Congenital cystic adenomatoid malformation of the lung (CCAM): Report of 2 atypical cases
Keywords:
congenital cycsic adenomatoid malformation, CCAM, lung, neonates
Abstract
Congeni~ cystic adenomatoid malformation (CCAM) presents mainly inneonates, rarely in children beyond infancy, and has been reported in adults. Two females (aged 26 months and 34 days) who had CCAM in the right and left lower lobe, respectively, are reported. One of them presented with recurrent respiratory infection and the other as newbom with respiratory distress secondary to mediastinal displacement and pulmonary compression as a result of expanding cystic lesions. Both cases had different lesions from typical neonatal cases reported in the literature.
Single cyst was shown in the first case, but multiple cysts in other. The young child survived, but the baby died. All lesions had lining varied from pseudostratified columnar to cuboidal epithelium. Cartilage plates was found in the second case but not in the first. The absence of inflammation is typical in neonates' lesions, by contrast, all of our two patients had clinical and pathologic evidence of chronic inflammation. CCAM may be clinically silent in infancy and may present as pneumonia associated with cystic lesion on chest X-ray in childhood or la ter in life. To support the diagnosis, CT scanning is needed.
References
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2. Bale PM. Congenital cystic malformation of the lung: A form of congenital bronchiolar ("adenomatoid") malformation. Am J Clin Path 1979;71 : 411-20.
3. Neugebauer H, Stauber R. Air trapping from torsion of a congenital cyst of the lung. Eur J Pediatr Surg 1992;2: 233-5.
4. Kwittken J , Reiner L. Congenital cystic adcnomatoid malformation of the lung. Pediatrics 1962;30: 759-68.
5. Stocker JT, Madewell JE, Drake RM. Congenital cystic adenomatoid malformation of the lung. Classification and morphologic spectrum. Hum Palhol1977;8: 155-71.
6. Nuchtem JG, Harberg FJ. Congenital lung cyst. Semin Pediatr Surg 1994: 233-43.
7. Merenstein MAJ. Congenital cystic adenomatoid malformation of the lung. Report of a case and review of the literature. Arner J Dis Child 1969; 118: 772-6.
8. Avitabile AM, Greco MA, Hulnick DH, Feiner HD. Congenital cystic adenomatoid malformation of the lung in adults. The Am J of Surg Path 1984;8: 193-202.
9 . Salzberg AM, Krummel TM. Congenital malformations of the lower respiratory tract. In: Chernick, Kendig eds. Kendig's Disorders of the respiratory tract in children. Fifth edition. Philadelphia WB Sa under 1990;227 -67.
10. Raffensperger JG. Congenital malformation of the lung. In: Swensons's pediatric surgery. Fifth edition, Norwalk: Appleton & Lange 1990;743-53.
11. Pankaj S Martin MM, Eliot. Congenital malformation of the lung. In: Surgery of the newborn. Edinburg: Churchill Livingstone 1994: 431-49.
12. Lierl M. Congenital abnormalities. In: Hilmm BC ed. Pediatric Respiratory Disease. Diagnosis and Treatment. Philadelphia WB Saunders 1993;457-98.
13. Rowe MI, O'Neill JA, Grosfcld JL, Fonkalsrud EW, Coran AG. Congenital anomalies of the lung. In: Essentials of Pediatric Surgery. StLouis: Mosby 1995;365-72.
14. Shields TW. Parapneumonic empyema. In: Shields ed. General Thoracic Surgery. Fourth edition. William & Wilkins. Baltimore 1994;648-93.
Published
1999-08-30
How to Cite
1.
Nataprawira M, Kartasasmita C, Rosmayudi0, H. A, R. S, Wahyu T, Silitonga L, Hernowo B. Congenital cystic adenomatoid malformation of the lung (CCAM): Report of 2 atypical cases. PI [Internet]. 30Aug.1999 [cited 18Apr.2024];39(7-8):229-6. Available from: https://paediatricaindonesiana.org/index.php/paediatrica-indonesiana/article/view/1774
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Received 2018-02-13
Accepted 2018-02-13
Published 1999-08-30
Accepted 2018-02-13
Published 1999-08-30
