Perinatally diagnosed asymptomatic congenital cystic adenomatoid malformation: to resect or not?

Aziz, Dalal; Langer, Jacob C.; Tuuha, Sascha E.; Ryan, Greg; Ein, Sigmund H.; Kim, Peter C.W.

doi:10.1016/j.jpedsurg.2003.11.021

Cited by 168 publications

(132 citation statements)

References 16 publications

(20 reference statements)

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“…The prevalence of the composite adverse postnatal outcome was 15%, and so the majority of infants (85%) had an uncomplicated course. Similarly, other studies of postnatal follow-up after prenatal diagnosis of CCAM have also reported good outcomes [7,11,15,16,17,18,19,20,21,22,23]. Although the prevalence of asthma in our cohort is similar to estimates in the general population [24], children with the composite adverse postnatal outcome were more likely to have asthma.…”

Section: Discussionsupporting

confidence: 82%

“…When we repeated the association between the CVR and composite adverse postnatal outcome only for those cases where the CVR was calculated in the second or early third trimester (<28 weeks) (n = 47), it was still statistically significant (Mann-Whitney test, z = 2.52, p = 0.01). On the other hand, the study’s strengths include a sample size that is larger than other postnatal follow-up studies of CCAM and a median age of follow-up comparable to these other studies [7,11,15,16,17,18,19,20,21,22,23]. …”

Section: Discussionmentioning

confidence: 99%

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Prediction of Pediatric Outcome after Prenatal Diagnosis and Expectant Antenatal Management of Congenital Cystic Adenomatoid Malformation

Yong

Dadelszen²,

Carpara³

et al. 2012

Fetal Diagn Ther

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Objective: To determine whether the congenital cystic adenomatoid malformation (CCAM) volume ratio (CVR) is associated with fetal and postnatal outcome after prenatal diagnosis and antenatal expectant management in a provincial tertiary referral center that does not offer fetal surgery. Methods: Retrospective cohort of 71 consecutive cases of prenatally diagnosed CCAM meeting study criteria (1996–2004). CVR was calculated on the initial ultrasound at the referral center, and associated with hydrops (Fisher’s exact test) and a composite adverse postnatal outcome consisting of death, intubation for respiratory distress, extracorporeal membrane oxygenation, non-elective surgery for symptomatology, or respiratory infection requiring hospital admission (Mann-Whitney test). Results: A CVR >1.6 was significantly associated with hydrops (p = 0.003). In addition, the CVR was significantly associated with the composite adverse postnatal outcome (p = 0.004) at a mean age of follow-up of 41 months (range <1–117 months). For CVR and postnatal outcome, the area-under-the-curve receiver operating characteristic was 0.81 (95% CI 0.69–0.93, p = 0.006), and choosing a CVR cut-off of <0.56, the negative predictive value was 100% (95% CI 0.85–1.00). Conclusion: In a provincial referral center with antenatal expectant management of CCAM, the CVR was associated with hydrops and postnatal outcome, with a CVR <0.56 predictive of good prognosis after birth.

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Section: Discussionsupporting

confidence: 82%

Section: Discussionmentioning

confidence: 99%

Prediction of Pediatric Outcome after Prenatal Diagnosis and Expectant Antenatal Management of Congenital Cystic Adenomatoid Malformation

Yong

Dadelszen²,

Carpara³

et al. 2012

Fetal Diagn Ther

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“…Some authors adopt an early resection strategy to avoid the onset of symptoms [4,15,98,99] since the safety of lung resection during the infantile period has been proven [41,98,100], while others adopt a conservative strategy, recognising the surgical risks and the potential of overtreatment [97,101,102,103], and surgery is only purposed after the patient becomes symptomatic with recurrent infections or pneumothorax [100,104]. Additionally, some authors recognise the potential for spontaneous resolution: in a study of 56 children with an antenatal diagnosis of CPAM, two CPAMs spontaneously resolved postnatally and ten resolved antenatally [104].…”

Section: Managementmentioning

confidence: 99%

Prenatal and Postnatal Management of Congenital Pulmonary Airway Malformation

2016

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Congenital pulmonary airway malformation (CPAM) is one of the most common lung lesions detected prenatally. Despite the research efforts made in the past few years, controversy and lack of clarity in the literature still exist regarding nomenclature, classification, pathogenesis and the management of CPAM. Therefore, it is of greatest importance to delineate the natural history of CPAMs and to create a consensus to guide the management and follow-up of these lesions. This review will focus on classification systems, highlighting the most recent advancements in pathogenesis, and current practice in the prenatal diagnosis of CPAM. Strategies of prenatal management and postnatal management will be reviewed. Long-term follow-up, including lung cancer risk, is discussed and an outcome perspective is presented.

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“…Postnatal CCAM complications range from lesions that are completely asymptomatic to severe respiratory distress with neonatal death [3] . The ratio of asymptomatic to symptomatic neonates is 3 to 1 [22] .…”

Section: Discussionmentioning

confidence: 99%

Successful Intrauterine Therapy for Congenital Cystic Adenomatoid Malformation of the Lung

Isnard

Köhler

et al. 2007

Fetal Diagn Ther

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Congenital cystic adenomatoid malformation (CCAM) of the lung is a rare abnormality which is amenable to be diagnosed by prenatal ultrasonography. In general, CCAM associated with non-immune hydrops has a poor prognosis unless a fetal intervention is performed. In some series almost 100% of either prenatal or early neonatal deaths are observed without intervention. Recently the cystic adenomatoid malformation volume ratio (CVR) has been proposed as an index to predict the development of hydrops in this condition. If the CVR is >1.6, the risk of hydrops is approximately 75%. We report a case of CCAM (macrocystic type) of the left lung referred to our center at 21 weeks of gestation. Fetal therapy was considered owing to enlargement of the CCAM volume, severe mediastinal shift, right lung compression, polyhydramnios and ascites from 21 to 26 weeks. Thoracoamniotic shunting was performed successfully at 26 weeks with resolution of the cystic mass. At 37 weeks a male infant was born vaginally weighing 3,210 g with Apgar scores of 5 and 10. Resection of the mass was performed uneventfully on day 3. The infant is currently 22 months of age, asymptomatic and in good condition.

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Perinatally diagnosed asymptomatic congenital cystic adenomatoid malformation: to resect or not?

Cited by 168 publications

References 16 publications

Prediction of Pediatric Outcome after Prenatal Diagnosis and Expectant Antenatal Management of Congenital Cystic Adenomatoid Malformation

Prediction of Pediatric Outcome after Prenatal Diagnosis and Expectant Antenatal Management of Congenital Cystic Adenomatoid Malformation

Prenatal and Postnatal Management of Congenital Pulmonary Airway Malformation

Successful Intrauterine Therapy for Congenital Cystic Adenomatoid Malformation of the Lung

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