Pleural loculated empyema masking a CPAM 3 in a newborn infant: A case report with breef literature review

Abstract

Congenital pulmonary airway malformations (CPAMs) are a heterogeneous group of hamartomatous cystic and noncystic lung lesions that result from early airway maldevelopment. Usually they are distinguished according to Stocker's classification in type 0, 1, 2, 3 and 4.We present the case of a 2 weeks old baby who was admitted to hospital with RDS symptoms and left pleural effusion: X rays and CT were suggestive for a pulmonary cystic lesion with pleural complications. Because of the persistence of pleural empyema and the development of a pneumothorax the baby underwent surgery. The histological examination revealed a type 3 CPAM associated with pleural loculated empyema.According to this case, in newborns with RDS loculated pleural empyema may mimick pulmonary cystic lesions; a treatment-resistant pleural empyema or pyopneumothorax in a newborn can recognize a CPAM 3 as a probable underlying condition, even in the absence of lung suppurative changes; CPAM 3 involving only two lung segments can have an excellent prognosis after surgical excision.

A rare case of multiloculated thoracic empyema in a neonate

“…In neonate, loculated thoracic empyema may be hard to differentiate with congenital pulmonary airway malformation (CPAM), which also gives appearance of multiple lucent areas on chest x-ray. This emphasizes the importance of comprehensive radiological examinations to confirm the true diagnoses of multiloculated thoracic empyema, as CT scan will clearly show the presence of fluid within the pleural cavity as in thoracic empyema, while in CPAM it will show multiple cystic areas with pulmonary artery supply [ 11 , 12 ].…”

An Incidental Finding of Congenital Pulmonary Airway Malformation Type 3 During a Forensic Autopsy for a Sudden Infant Death

“…[4][5][6][7] It has an incidence ranging from 1 in 5000 to 35,000 live births and ranging from 1 in 10,000 to 35,000 in newborns. 1,4,[7][8][9][10] As previously stated, this type of lung disorder was formerly named congenital cystic airway malformation by Ch'in and Tang in 1949. 1,9,11 It was a rather poorly chosen name, as only 2 of the 5 major subtypes have a cystic pattern, and only 1 other type has an adenomatoid appearance.…”

“…Radiologically, CPAMs are classified as large cyst CPAMs (types 1 and 4), small cyst CPAMs (type 2), or microcystic (solid-appearing) CPAMs (type 3). 5,9,12,18 An overview of the imaging characteristics of each type can be found in Table 2. After birth, the imaging modality of choice for the first evaluation is a chest x-ray; the lesions can be further evaluated using CT or magnetic resonance imaging scan.…”

“…1 Symptoms, clinical findings, and prognoses depend on the subtype and the degree of pulmonary involvement. 6,9 An overview of these classifications can be found in Table 1. Asymptomatic cases at birth have also been described.…”

“…In such a case, the most probable differential diagnosis is CPAM type 3. 9,18 As for cystic lesions, bronchogenic cyst and pneumatoceles should be considered as differential diagnoses. The latter is caused by infection, typically Staphylococcus, whereas the former is seen in older children or adolescents and usually concerns a single cyst, located centrally or in the mediastinum.…”