Prenatal diagnosis and management of congenital cystic adenomatoid malformation of the lung

Duncombe, Gregory; Dickinson, Jan E.; Kikiros, Colin

doi:10.1067/mob.2002.127460

Cited by 110 publications

(84 citation statements)

References 15 publications

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“…The incidence of hydrops in our cohort (8.6%) was similar to the 5–10% incidence of hydrops in some studies [11,12,13], but in contrast to the 30–40% incidence in the studies of Crombleholme et al [3] and Adzick et al [14]. The reason is likely that these latter studies are from two national referral centers for fetal surgery which see more high-risk or already hydropic fetuses.…”

Section: Discussioncontrasting

confidence: 51%

“…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: Discussioncontrasting

confidence: 33%

“…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: Discussioncontrasting

confidence: 51%

Section: Discussioncontrasting

confidence: 33%

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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“…Bronchopulmonary sequestration is characterized by a lung mass with nonfunctioning lung tissue receiving its blood supply from an anomalous systemic feeding artery, originating commonly from the aorta [5]. Microcystic CCAM is considered a pulmonary hamartoma with dysplastic lung tissue and lack of differentiation of the associated alveoli [6,7]. Mainstem bronchial atresia consists in a complete intrinsic obstruction of the fetal upper airway at the level of the main bronchi that involves all lung parenchyma as a consequence.…”

Section: Discussionmentioning

confidence: 99%

Fetal Bronchoscopy as a Useful Procedure in a Case with Prenatal Diagnosis of Congenital Microcystic Adenomatoid Malformation

Cruz‐Martínez¹,

Méndez²,

Perez-Garcilita³

et al. 2014

Fetal Diagn Ther

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Massive microcystic congenital cystic adenomatoid malformation (CCAM) and bronchial atresia are associated with a high perinatal mortality secondary to lung hypoplasia and cardiac dysfunction, and fetal intervention should be considered to improve prognosis. Therapeutic options include open fetal surgery with pulmonary resection, fetal sclerotherapy and fetoscopy. We present a case with a severely enlarged left lung without ultrasound signs of dilated airways compatible with the diagnosis of microcystic CCAM, hydrops and severe contralateral lung hypoplasia that was treated successfully at 30 weeks of gestation by fetal bronchoscopy, through which bronchial atresia was identified at the end of the left mainstem bronchi and permeabilized by laser ablation. After fetal surgery, weekly follow-up showed a progressive decrease in the affected lung size and an increase in the contralateral hypoplastic lung size, demonstrating normal dimensions of both lungs at 34 weeks of gestation, reversal of the mediastinal shift, and complete disappearance of hydrops. A healthy neonate was delivered uneventfully at term with no need for respiratory support, and the boy is now doing well at 15 months of age. This report demonstrates that in cases with prenatal diagnosis of large microcystic CCAM, fetal bronchoscopy can be used to refine the diagnosis of bronchial atresia and as a therapeutic tool with good outcome.

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“…It occurs in approximately 1 in every 35000 to 1 in 10,000 pregnancies. 6,12,13,16 Recent studies have shown that antenatally detected CCAMS offer a better prognosis and favourable outcome for the patients. 12,13,14,15,16 …”

Section: Introductionmentioning

confidence: 99%

The telltale Cinderella story of Congenital cystic adenomatoid malformation of the lungs in a child: a rare case report from DAKSHIN KANNADA

Shetty¹,

Thapa²,

Khandige³

2013

Int Jour of Biomed Res

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Prenatal diagnosis and management of congenital cystic adenomatoid malformation of the lung

Cited by 110 publications

References 15 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

Fetal Bronchoscopy as a Useful Procedure in a Case with Prenatal Diagnosis of Congenital Microcystic Adenomatoid Malformation

The telltale Cinderella story of Congenital cystic adenomatoid malformation of the lungs in a child: a rare case report from DAKSHIN KANNADA

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