A neonate with coexisting congenital cystic adenomatoid malformation of the lung and alveolar capillary dysplasia: A case report with review of literature

Roeleveld, Peter P.; Martin, J. N.; Chow, C. W.; Duke, Trevor

doi:10.1097/pcc.0b013e318166fa88

Cited by 9 publications

(6 citation statements)

References 24 publications

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“…Extrapulmonary findings are present in 50 to 80% of cases (10,14,21,25,30,33,34). Therefore, an infant with unexplained respiratory failure and structural abnormalities of the genitourinary, gastrointestinal, or cardiovascular systems should be evaluated for ACD/MPV (10,33).…”

Section: Physical and Laboratory Findingsmentioning

confidence: 99%

“…Therefore, an infant with unexplained respiratory failure and structural abnormalities of the genitourinary, gastrointestinal, or cardiovascular systems should be evaluated for ACD/MPV (10,33). Additionally, disruption of right-left asymmetry of the intrathoracic and/or intraabdominal organs has been reported in more than 25% of cases (10).…”

Section: Physical and Laboratory Findingsmentioning

confidence: 99%

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Alveolar Capillary Dysplasia

Bishop

Stankiewicz

Steinhorn

2011

Am J Respir Crit Care Med

210

220

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Section: Physical and Laboratory Findingsmentioning

confidence: 99%

Section: Physical and Laboratory Findingsmentioning

confidence: 99%

Alveolar Capillary Dysplasia

Bishop

Stankiewicz

Steinhorn

2011

Am J Respir Crit Care Med

210

220

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“…Furthermore, many congenital lung masses regress in utero. Derderian et al directly compared fetuses with CDH and CPAMs and found that patients with CDH develop PH much more frequently than those with CPAMs (88% vs 18% respectively), suggesting that mass effect is not the sole driver of pulmonary artery hypoplasia in patients with CDH . The lower incidence of PH in fetuses with lung masses relative to those with CDH may be related to the later onset of disease and frequent regression during fetal life.…”

Section: Discussionmentioning

confidence: 99%

In fetuses with congenital lung masses, decreased ventricular and atrioventricular valve dimensions are associated with lesion size and clinical outcome

et al. 2019

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Introduction:The clinical importance of mass effect from congenital lung masses on the fetal heart is unknown. We aimed to report cardiac measurements in fetuses with congenital lung masses and to correlate lung mass severity/size with cardiac dimensions and clinical outcomes.Methods: Cases were identified from our institutional database between 2009 and 2016. We recorded atrioventricular valve (AVVz) annulus dimensions and ventricular widths (VWz) converted into z scores, ratio of aortic to total cardiac output (AoCO), lesion side, and congenital pulmonary airway malformation volume ratio (CVR). Respiratory intervention (RI) was defined as intubation, extracorporeal membrane oxygenation (ECMO), or use of surgical intervention prior to discharge.Results: Fifty-two fetuses comprised the study cohort. Mean AVVz and VWz were below expected for gestational age. CVR correlated with ipsilateral AVVz (R S = −.59, P < .001) and ipsilateral VWz (−0.59, P < .001). Lower AVVz and AoCO and higher CVR were associated with RI. No patient had significant structural heart disease identified postnatally. Conclusion:In fetuses with left-sided lung masses, ipsilateral cardiac structures tend to be smaller, but in our cohort, there were no patients with structural heart disease. However, smaller left-sided structures may contribute to the need for RI that affects a portion of these fetuses.

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“…All ACD cases and 75% of the controls had TR velocities ≥ 3.2 m/sec ( of severe and fixed pulmonary hypertension attributed to "the deranged vascular endothelial growth factor signaling" have been previously described in ACD patients [14]. Furthermore, despite the presence of severe pulmonary hypertension and profound hypoxemia, neonates with ACD can be differentiated from others by relative initial sparing of lung parenchymal disease as exhibited by their relatively normal lung mechanics and compliance [1].…”

Section: Echocardiographicmentioning

confidence: 95%

Echocardiographic Predictors of Alveolar Capillary Dysplasia: A Case-Control Study

Arcinue¹,

Theodora²,

Noori³

et al. 2016

Enliven: J Anesth Crit Care Med

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ObjectiveTo evaluate early echocardiographic variables of neonates with alveolar capillary dysplasia (ACD). Study DesignA case-control study of five neonates with ACD and their matched controls on extracorporeal membrane oxygenation from 2000-2011. Two cardiologists prospectively reviewed first admission echocardiograms. Statistical tests included Kappa, T-test, Mann-Whitney, Fisher's exact, and Chi-square analysis. ResultsExcellent inter-rater reliability readings were achieved (Kappa 0.75-1). Tricuspid regurgitation (TR) velocities between the groups were not statistically significant; all ACD cases and 75% of the controls had TR velocities ≥ 3.2 m/sec. More ACD cases had moderate-severe TR jet (80% vs. none, p<0.01); moderate-severe right atrial enlargement (RAE) (100% vs. none, p<0.01); moderate-severe right ventricular (RV) dilatation (100% vs. none, p<0.01); and moderate-severely depressed RV function (60% vs. none, p<0.01). ACD patients were 12 times more likely to have a combination of moderate-severe RAE and moderate-severe TR jet compared to controls ). ConclusionThe combination of moderate-severe RAE and TR jet unresponsive to maximal interventions should alert clinicians of the possibility for ACD. We speculate that early echocardiographic predictors may identify at-risk neonates with fatal ACD and lead to early consideration of lung transplant.

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A neonate with coexisting congenital cystic adenomatoid malformation of the lung and alveolar capillary dysplasia: A case report with review of literature

Cited by 9 publications

References 24 publications

Alveolar Capillary Dysplasia

Alveolar Capillary Dysplasia

In fetuses with congenital lung masses, decreased ventricular and atrioventricular valve dimensions are associated with lesion size and clinical outcome

Echocardiographic Predictors of Alveolar Capillary Dysplasia: A Case-Control Study

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