Departemen Anestesiologi dan Terapi Intensif Fakultas Kedokteran

Universitas Sriwijaya

Case Series

Anesthesia in Congenital Cystic Adenomatous Malformation (CCAM)

Resection in Pediatric Patients with Co-Existing Disease

Hari Ciptadi, ,Yusni Puspita

Anesthesiology and Intensive Therapy Department
Medical Faculty of Sriwijaya University
dr. Mohammad Hoesin Hospital
Palembang, Indonesia

Background: Congenital Cystic Adenomatous Malformation (CCAM) or commonly known as

lobar emphysema is a rare congenital disease that is often diagnosed in first six months of
neonatal period. The prevalence is one in 30.000 pregnancies. The sign of this disease is
hyperinflation of alveoli which results in disruption of lung parenchyma and loss of its
elasticity. If this condition continues, lobar hyperinflation will occur and compress its
surrounding structures, causing mediastinal shift, and venous return disturbance. Disruption
in ventilation perfusion will happen which results in hypoxia. The current therapy is
definitive surgery through resection. Co-existing malnutrition and hypothyroid can
complicate anesthesia in these patients. Comprehensive anesthesia management is
fundamental for a successful resectomy and optimal outcome.
Objective : To present case series of CCAM with co-existing diseases with different
anesthesia management.
Case series: We report a case series of CCAM. The first was a case of CCAM type I,
unicystic, with co-existing malnutrition in a five months infant that had prior diagnosis of
spontaneous pneumothorax through thorax posteroinferior X-ray and CT-scan. The second
was the case of a nineteen month infant with CCAM type I, multicystic, and co-existing
hypothyroid and malnutrition. The third was the case of twenty months infant with CCAM
type , unicystic and co-existing malnutrition. The fourth patient was a five months old infant
with CCAM Type , multicystic, and malnutrition. All were planned to undergo resectomy.
Conclusion : In emergency cases, assisted ventilation is possible in patients with CCAM with
co-existing diseases in the presence of a surgeon.

Keywords: anesthesia management, congenital cystic adenomatous malformation,

hypothyroid, malnutrition, assisted ventilation

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