Life-Threatening Congenital Cystic Adenomatoid Malformation in the Premature Neonate

Chong, Young‐Jun; Rhee, Yun Ee; Han, Sung Joon; Cho, Hyun Jin; Kang, Shin Kwang; Kang, Min-Woong

doi:10.5090/kjtcs.2016.49.3.210

Cited by 10 publications

(12 citation statements)

References 8 publications

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“…2 In severe cases, patients develop respiratory failure even in the neonatal period, but others have a benign clinical course, with the manifestation of chronic obstructive pulmonary disease or recurrent pneumonia as an adult. [3][4][5]8 In our case, the lung malformation was responsible for the early acute respiratory failure and death.…”

Section: Discussionmentioning

confidence: 96%

“…1 Congenital pulmonary airway malformations (CPAMs) are a heterogeneous group of cystic and non-cystic lung lesions that result from early airway maldevelopment with the incidence ranging between 1:10,000 and 1:35,000 newborns. [2][3][4][5][6] This entity was first classified by Ch'in and Tang 7 in 1949 as congenital cystic adenomatoid malformation (CCAM). Although originally described as "congenital cystic adenomatoid malformation," this entity now has the designation of "congenital pulmonary airway malformation" because the lesions described as "cystic" were present in only three of the five subtypes and "adenomatoid" in only one subtype.…”

Section: Discussionmentioning

confidence: 99%

“…1 The symptoms and prognosis depend on the degree of pulmonary involvement. [3][4][5]8 The clinical presentation spectrum varies from asymptomatic cases, recurrent pneumonia, or respiratory insufficiency. 2 In severe cases, patients develop respiratory failure even in the neonatal period, but others have a benign clinical course, with the manifestation of chronic obstructive pulmonary disease or recurrent pneumonia as an adult.…”

Section: Discussionmentioning

confidence: 99%

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Congenital Pulmonary Airway Malformation

Mehta

2018

Imaging in Pediatrics

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Congenital pulmonary airway malformation (CPAM) is a developmental malformation of the lower respiratory tract. We report the case of a male newborn diagnosed with cystic lung disease during prenatal ultrasound. A cesarean section was performed at the 32nd gestational week because of premature rupture of the membranes, and soon after the delivery the newborn developed respiratory failure and died. The aim of this study is to report an autopsy case because of its rarity, and to briefly discuss the CPAM subtypes and differential diagnosis of cystic lung diseases of childhood.

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Section: Discussionmentioning

confidence: 96%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

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Congenital Pulmonary Airway Malformation

Mehta

2018

Imaging in Pediatrics

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“…Approximately 99% of CPAM can be diagnosed by fetal ultrasound in 18-20 weeks of gestation (8). Postnatal sensitivity of chest X-ray is 60% while the sensitivity of CT or MRI is 100% (8).…”

Section: Introductionmentioning

confidence: 99%

“…Approximately 99% of CPAM can be diagnosed by fetal ultrasound in 18-20 weeks of gestation (8). Postnatal sensitivity of chest X-ray is 60% while the sensitivity of CT or MRI is 100% (8). The lesion is usually unilateral (85%), with no difference in incidence of macrocystic or microcystic forms, but are also reported cases affecting bilateral or an entire lung (7,9).…”

Section: Introductionmentioning

confidence: 99%

Congenital Pulmonary Airway Malformation – Case Presentation

D¹,

Craiova²,

Dop³

et al. 2017

Ro J Pediatr.

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Congenital pulmonary airway malformation (CPAM) is a rare cystic lesion, whose prognosis depends on the extent of the lesion, the association of other congenital abnormalities, the associated pulmonary pathology and the possibility of curative surgery. We present the case of a 5-month-old infant diagnosed with bilateral CPAM, based on clinical and imaging investigations, with acute pulmonary manifestations and without other associated congenital anomalies.

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Clinical Characteristics of Congenital Pulmonary Airway Malformation of The Lungs: A Single-Center Study

Yoo,

Kim,

Kim

et al. 2023

jmds

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Objective: Congenital pulmonary airway malformation (CPAM) is a rare developmental abnormality of the lungs. We investigated clinical characteristics of CPAM based on patient age at diagnosis. Methods: In this retrospective study, we analyzed the medical records of 51 patients diagnosed with CPAM at Pusan National University Hospital between January 2000 and December 2019. Results: We investigated 39 children and 12 adults. The mean age at the diagnosis of the patients was 15.9 ± 14.3 years. The mean ages of children and adults at the time of diagnosis of CPAM were 6.8 ± 5.8 years and 31.2 ± 10.2 years, respectively. Among the 51 patients investigated, 20 (39%) were asymptomatic and 31 (61%) showed clinical symptoms, such as dry cough, recurrent respiratory infections, and dyspnea at the time of diagnosis. Notably, clinical symptoms at diagnosis were observed in 28 children (72%) and in only 3 adults (25%) (P = 0.006), and children were more symptomatic than adults. Children with large cysts tended to be more symptomatic than those with small cysts (P < 0.001). Combined anomalies were detected in 12 patients (23%). Patients with cystic lesions in the right lower lobe of the lung showed a higher prevalence of combined anomalies (P =0.015). Surgical resection was performed in 40 patients (78%), and all patients showed good prognosis. Conclusion: This study revealed that the presence of clinical symptoms of CPAM differed between children and adults, depending on the age at diagnosis and that patients with cysts in the right lower lobe of the lungs tended to show a higher prevalence of combined anomalies. (Allergy Asthma Respir Dis 2021, 9: 21–26)

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Life-Threatening Congenital Cystic Adenomatoid Malformation in the Premature Neonate

Cited by 10 publications

References 8 publications

Congenital Pulmonary Airway Malformation

Congenital Pulmonary Airway Malformation

Congenital Pulmonary Airway Malformation – Case Presentation

Clinical Characteristics of Congenital Pulmonary Airway Malformation of The Lungs: A Single-Center Study

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