Antenatal Diagnosis and Surgical Management of Congenital Cystic Adenomatoid Malformation of the Lung

Taguchi, Tomoaki; Suita, Sachiyo; Yamanouchi, Takeshi; Nagano, Miki; Shoji, Shuichi; Koyanagi, Takashi; Nakano, Hitoo

doi:10.1159/000264265

Cited by 37 publications

(21 citation statements)

References 6 publications

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“…In agreement with the published literature, our experience further confirms that CCAM is a pulmonary pathology with benign outcome, which is not usually associated with morphostructural, chromosomal or fetal growth defects [6,8,16,17]. The survival rate in our population was 13 of 15 cases (86.7%).…”

Section: Discussionsupporting

confidence: 92%

Congenital Cystic Adenomatoid Malformation of the Lung: Antenatal Ultrasound Findings and Fetal-Neonatal Outcome

Santis

Masini²,

Noia³

et al. 2000

Fetal Diagn Ther

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Seventeen cases of congenital cystic adenomatoid malformation of the lung (CCAM) are reported. They were followed up over a period of 1 month to 15 years. Diagnosis was made by prenatal ultrasound. Our purpose was to evaluate the fetal-neonatal outcome and the prognostic elements observable through ultrasound techniques, and to compare all types of CCAM. The outcome observed ranged from total prenatal resolution to postnatal spontaneous regression of the lesion, to complications due to the presence of nonimmune fetal hydrops (NIFH), intrauterine death and the necessity of surgical intervention. In our experience only hydrops represented a negative predictor of outcome since death occurred in all cases with this pathology. In the absence of NIFH, counselling should stress the prevalence of a positive outcome, even in cases of surgical intervention.

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Section: Discussionsupporting

confidence: 92%

Congenital Cystic Adenomatoid Malformation of the Lung: Antenatal Ultrasound Findings and Fetal-Neonatal Outcome

Santis

Masini²,

Noia³

et al. 2000

Fetal Diagn Ther

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“…In addition, close follow-up in utero with serial ultrasound is essential since these lesions may evolve and change appearance. As in 5 of our patients, others have noted regression of microcystic lesions [9,12,15,20]. Even if the appearance of the lesion (micro-vs. macrocystic) and polyhydramnios can have some prognostic value, the presence of associated anomalies and/or hydrops are the most important factors.…”

Section: Discussionsupporting

confidence: 69%

Outcome of the Prenatally Diagnosed Congenital Cystic Adenomatoid Lung Malformation: A Canadian Experience

et al. 2001

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Congenital cystic adenomatoid malformation of the lung (CCAM) is diagnosed by prenatal ultrasonography with an increasing frequency but controversy persists as to its prognosis and prenatal management. Method: A multi-institutional study of cases of CCAM diagnosed antenatally identified by ultrasonographers and by a review of hospital charts. Results: We obtained 48 cases from five centers. We estimate the incidence of CCAM at 1:25,000 to 1:35,000 pregnancies. The incidence of voluntary abortions was 15% (7/48), of spontaneous abortions 2% (1/41) and of postnatal death 10% (4/40). One of the postnatal deaths was from trisomy 18. Of the 7 aborted fetuses, 2 had multiple malformations and 1 had severe hydrops and oligohydramnios; the other 4 had a large mass with mediastinal displacement but without hydrops. When pregnancy was allowed to continue, 56% of the lesions regressed spontaneously, even though one third of these had initial progression. In 17 cases (42%) the mediastinal shift corrected itself, sometimes by simple growth of the fetus but most often by a decrease in the size of the lung mass. In 1 fetus, repeated needle decompressions followed by double-pigtail catheter drainage of large cysts allowed regression of hydrops. Despite this, neonatal death occurred from pulmonary hypoplasia. Conclusion: CCAM can lead to fetal or neonatal demise from hydrops, lung hypoplasia, prematurity or severe associated malformations, but has a good prognosis in the majority of cases.

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“…Possible mechanisms for this regression may include apoptosis and lesions outgrowing their vascular supply [50]. Regression of lung lesions associated with hydrops, while rare, has been reported [51,52].…”

Section: Prenatal Diagnosis and Natural Historymentioning

confidence: 99%

Congenital cystic lung disease: contemporary antenatal and postnatal management

2008

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Congenital cystic lung disease comprises a broad spectrum of rare but clinically significant developmental abnormalities, including congenital pulmonary adenomatoid malformations, bronchopulmonary sequestrations, bronchogenic cysts, and congenital lobar emphysema that result from perturbations in lung and airway embryogenesis. As congenital lung lesions are now more commonly recognized antenatally, mothers require accurate prenatal counseling and appropriate perinatal management. In light of long-term complications of infection and malignancy, there is growing consensus that infants with asymptomatic lesions should undergo elective excision of congenital pulmonary adenomatoid malformation (CPAM) or bronchopulmonary sequestration (BPS). This review will focus on advancements and current practice in the diagnosis and management of CPAM and BPS, identifying aspects of the literature that are confusing or controversial. Although our knowledge and pre- and postnatal management of lung lesions will continue to evolve and improve, there is a compelling need for a unified clinical and pathological classification system that creates a common platform for discussion, clinical management, and research.

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Antenatal Diagnosis and Surgical Management of Congenital Cystic Adenomatoid Malformation of the Lung

Cited by 37 publications

References 6 publications

Congenital Cystic Adenomatoid Malformation of the Lung: Antenatal Ultrasound Findings and Fetal-Neonatal Outcome

Congenital Cystic Adenomatoid Malformation of the Lung: Antenatal Ultrasound Findings and Fetal-Neonatal Outcome

Outcome of the Prenatally Diagnosed Congenital Cystic Adenomatoid Lung Malformation: A Canadian Experience

Congenital cystic lung disease: contemporary antenatal and postnatal management

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