* Corresponding Author
Congenital cystic adenomatoid malformation: a case report
https://doi.org/10.19106/JMedSci005303202111 Haryo Aribowo(1*), Divva Aurelia Azhalisita Amelinda(2), Ghifari Farandhi(3)
(1) Division of Cardiac, Thoracic, and Vascular Surgery, Department of Surgery, Faculty of Medicine, Public Health, and Nursing, Universitas Gadjah Mada/Dr. Sardjito General Hospital, Yogyakarta, Indonesia
(2) Division of Cardiac, Thoracic, and Vascular Surgery, Department of Surgery, Faculty of Medicine, Public Health, and Nursing, Universitas Gadjah Mada/Dr. Sardjito General Hospital, Yogyakarta, Indonesia
(3) Division of Cardiac, Thoracic, and Vascular Surgery, Department of Surgery, Faculty of Medicine, Public Health, and Nursing, Universitas Gadjah Mada/Dr. Sardjito General Hospital, Yogyakarta, Indonesia
(*) Corresponding Author
Abstract
defined by multiple cysts produced in the lung that occur during the fetal
period, with respiratory distress as presenting symptoms. Untreated CCAM
may lead to repeated lung infection and pneumothorax. Many surgical
techniques have been used to treat CCAM. However, those techniques showed
various results. Moreover, less studies were performed to evaluate the
effect of those surgical techniques in treating CCAM patients. We reported a
management of a rare pediatric case of CCAM referred to the Department of Thoracic and Cardiovascular Surgery, Faculty of Medicine, Public Health and
Nursing, Universitas Gadjah Mada/Dr. Sardjito General Hospital. The patient
was a 29-day-old male baby who presented with pneumothorax on the right
lung due to CCAM. The plan of treatment for the patient was lobectomy until
pneumonectomy on the affected lung. During the thoracotomy procedure,
we found that all lobes in the right lung were covered with fibrous tissue.
Based on this finding, a decortication procedure to remove the fibrous tissue
continued by a bullectomy procedure with the insertion of a chest tube were
performed. The patient continuously showed improvement in breathing and
wound healing, thus making the patient discharged from the hospital on the
nineteenth postoperative day. The cause of CCAM is thought to be congenital
abnormalities of the bronchiole epithelium that produce multiple cysts. Due to
its rarity and lack of research on CCAM, many CCAM patients are misdiagnosed/
underdiagnosed. Common surgical methods used in this patient are lobectomy
with continuation until pneumonectomy or bilobectomy, if necessary, to
prevent recurrence. Parenchymal saving methods can be considered because
they have the same outcome as lobectomy. As performed in this patient,
thoracotomy decortication continued with bullectomy is adequately capable of
alleviating respiratory distress symptoms and is thus described as successful.
Keywords
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1.Hamanaka R, Yagasaki H, Kohno M, Masuda R, Iwazaki M. Congenital cystic adenomatoid malformation in adults: Report of a case presenting with a recurrent pneumothorax and a literature review of 60 cases. Respir Med Case Rep 2018; 26:328-32.
https://doi.org/10.1016/j.rmcr.2018.02.002
2.Gornall AS, Budd JL, Draper E, Konje JC, Kurinczuk JJ. Congenital cystic adenomatoid malformation: accuracy of prenatal diagnosis, prevalence and outcome in a general population. Prenat Diagn 2003; 23(12):997-1002.
https://doi.org/10.1002/pd.739
3.Laberge JM, Flageole H, Pugash D, Khalife S, Blair G, Filiatrault D, et al. Outcome of the prenatally diagnosed congenital cystic adenomatoid lung malformation: a Canadian experience. Fetal Diagn Ther 2001; 16(3):178-86.
https://doi.org/10.1159/000053905
4.Sood M. Congenital Cystic Adenomatoid Malformation of the Lung: A Case Report. J Nepal Pediatr Surg 2011; 31(1):64-7.
https://doi.org/10.3126/jnps.v31i1.4162
5.Annam V, Korishetty SI, Yelikar BR, Hippargi SB, Shivalingappa DB. Bilateral congenital cystic adenomatoid malformation, stocker type III with associated findings and review of literature. Indian J Pathol Microbiol 2010; 53(2):331-3.
https://doi.org/10.4103/0377-4929.64324
6.Gajewska-Knapik K, Impey L. Congenital lung lesions: Prenatal diagnosis and intervention. Semin Pediatr Surg 2015; 24(4):156 -159.
https://doi.org/10.1053/j.sempedsurg.2015.01.012
7.Mon RA, Johnson KN, Ladino-Torres M, Heider A, Mychaliska GB, Treadwell MC, et al. Diagnostic accuracy of imaging studies in congenital lung malformations. Arch Dis Child Fetal Neonatal Ed 2019; 104(4):F372-F377.
https://doi.org/10.1136/archdischild-2018-314979
8.Račaitė J, Šumkovskaja A, Arlauskienė A, Pilypienė I, Landsbergytė-Bukauskienė E. Congenital cystic adenomatous malformation: a case report and a literature review. Acta Med Litu 2018; 25(2):95-100.
https://doi.org/10.6001/actamedica.v25i2.3762
9.Kotecha S, Barbato A, Bush A, Claus F, Davenport M, Delacourt C, et al. Antenatal and postnatal management of congenital cystic adenomatoid malformation. Paediatr Respir Rev 2012; 13(3):162-70; quiz 170-1.
https://doi.org/10.1016/j.prrv.2012.01.002
10.Chikkannaiah P, Kangle R, Hawal M. Congenital cystic adenomatoid malformation of lung: Report of two cases with review of literature. Lung India 2013; 30(3):215-8.
https://doi.org/10.4103/0970-2113.116272
11.Chowdhury MM, Chakraborty S. Imaging of congenital lung malformations. Semin Pediatr Surg 2015; 24(4):168-75.
https://doi.org/10.1053/j.sempedsurg.2015.02.001
12.Feng A, Cai H, Sun Q, Zhang Y, Chen L, Meng F. Congenital cystic adenomatoid malformation of the lung in adults: 2 rare cases report and review of the literature. Diagn Pathol 2012; 7:37.
https://doi.org/10.1186/1746-1596-7-37
13.Davies AR, Bapat V, Treasure T. Adult presentation of congenital cystic adenomatoid malformation: successful surgical management. J Thorac Cardiovasc Surg 2006; 132(6):1493-4.
https://doi.org/10.1016/j.jtcvs.2006.06.041
14.Alshamiri KM, Abbod HB. Congenital cystic adenomatoid malformation. Int J Pediatr Adolesc Med 2017; 4(4):159-60.
https://doi.org/10.1016/j.ijpam.2017.12.001
15.Lanza C, Bolli V, Galeazzi V, Fabrizzi B, Fabrizzi G. Cystic adenomatoid malformation in children: CT histopathological correlation. Radiol Med 2007; 112(4):612-9
https://doi.org/10.1007/s11547-007-0166-0
16.Stocker JT. Congenital pulmonary airway malformation : a new name for and an expanded classification of congenital cystic adenomatoid malformation of the lung. Histopathology 2002; 41(Suppl2):424-30.
https://doi.org/10.1186/1746-1596-7-37
17.Muller CO, Berrebi D, Kheniche A, Bonnard A. Is radical lobectomy required in congenital cystic adenomatoid malformation? J Pediatr Surg 2012; 47(4): 642-5.
https://doi.org/10.1016/j.jpedsurg.2011.08.002
18.Kim HK, Choi YS, Kim K, Shim YM, Ku GW, Ahn KM, et al. Treatment of congenital cystic adenomatoid malformation: should lobectomy always be performed? Ann Thorac Surg 2008; 86(1):249-53.
https://doi.org/10.1016/j.athoracsur.2008.01.036
19.Oh BJ, Lee JS, Kim JS, Lim CM, Koh Y. Congenital cystic adenomatoid malformation of the lung in adults: clinical and CT evaluation of seven patients. Res 2006; 11(4):496-501.
https://doi.org/10.1111/j.1440-1843.2006.00878.x
20.Sakamoto K, Takei H, Nishii T, Maehara T, Omori T, Tajiri M, et al. Staple line coverage with absorbable mesh after thoracoscopic bullectomy for spontaneous pneumothorax. Surg Endosc 2004; 18(3):478-81.
https://doi.org/10.1007/s00464-003-8918-y
DOI: https://doi.org/10.19106/JMedSci005303202111
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