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


Congenital cystic adenomatoid malformation (CCAM) is a rare condition
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


congenital cystic adenomatoid malformation;bullectomy; pneumothorax; decortication; thoracotomy

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References


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DOI: https://doi.org/10.19106/JMedSci005303202111

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Journal of the Medical Sciences (Berkala Ilmu Kedokteran) by  Universitas Gadjah Mada is licensed under a Creative Commons Attribution-NonCommercial 4.0 International License.
Based on a work at http://jurnal.ugm.ac.id/bik/.