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BACKGROUND AND OBJECTIVE: Congenital pulmonary malformations (CPM) are mostly recognized on prenatal ultrasound scans. In a minority of cases, they may impair breathing at birth. The factors predictive of neonatal respiratory distress are not well defined, but an understanding of these factors is essential for decisions concerning the need for the delivery to take place in a tertiary care center. The aim of this study was to identify potential predictors of respiratory distress in neonates with CPM. METHODS: We selected cases of prenatal diagnosis of hyperechoic and/or cystic lung lesions from RespiRare, the French prospective multicenter registry for liveborn children with rare respiratory diseases (2008–2013). Prenatal parameters were correlated with neonatal respiratory outcome. RESULTS: Data were analyzed for 89 children, 22 (25%) of whom had abnormal breathing at birth. Severe respiratory distress, requiring oxygen supplementation or ventilatory support, was observed in 12 neonates (13%). Respiratory distress at birth was significantly associated with the following prenatal parameters: mediastinal shift (P = .0003), polyhydramnios (P = .05), ascites (P = .0005), maximum prenatal malformation area (P = .001), and maximum congenital pulmonary malformation volume ratio (CVR) (P = .001). Severe respiratory distress, requiring oxygen at birth, was best predicted by polyhydramnios, ascites, or a CVR >0.84. CONCLUSIONS: CVR >0.84, polyhydramnios, and ascites increased the risk of respiratory complications at birth in fetuses with CPM, and especially of severe respiratory distress, requiring oxygen supplementation or more intensive intervention. In such situations, the delivery should take place in a tertiary care center.
BACKGROUND AND OBJECTIVE: Congenital pulmonary malformations (CPM) are mostly recognized on prenatal ultrasound scans. In a minority of cases, they may impair breathing at birth. The factors predictive of neonatal respiratory distress are not well defined, but an understanding of these factors is essential for decisions concerning the need for the delivery to take place in a tertiary care center. The aim of this study was to identify potential predictors of respiratory distress in neonates with CPM. METHODS: We selected cases of prenatal diagnosis of hyperechoic and/or cystic lung lesions from RespiRare, the French prospective multicenter registry for liveborn children with rare respiratory diseases (2008–2013). Prenatal parameters were correlated with neonatal respiratory outcome. RESULTS: Data were analyzed for 89 children, 22 (25%) of whom had abnormal breathing at birth. Severe respiratory distress, requiring oxygen supplementation or ventilatory support, was observed in 12 neonates (13%). Respiratory distress at birth was significantly associated with the following prenatal parameters: mediastinal shift (P = .0003), polyhydramnios (P = .05), ascites (P = .0005), maximum prenatal malformation area (P = .001), and maximum congenital pulmonary malformation volume ratio (CVR) (P = .001). Severe respiratory distress, requiring oxygen at birth, was best predicted by polyhydramnios, ascites, or a CVR >0.84. CONCLUSIONS: CVR >0.84, polyhydramnios, and ascites increased the risk of respiratory complications at birth in fetuses with CPM, and especially of severe respiratory distress, requiring oxygen supplementation or more intensive intervention. In such situations, the delivery should take place in a tertiary care center.
IMPORTANCEThe ability of computed tomography (CT) to distinguish between benign congenital lung malformations and malignant cystic pleuropulmonary blastomas (PPBs) is unclear.OBJECTIVE To assess whether chest CT can detect malignant tumors among postnatally detected lung lesions in children. DESIGN, SETTING, AND PARTICIPANTSThis retrospective multicenter case-control study used a consortium database of 521 pathologically confirmed primary lung lesions from January 1, 2009, through December 31, 2015, to assess diagnostic accuracy. Preoperative CT scans of children with cystic PPB (cases) were selected and age-matched with CT scans from patients with postnatally detected congenital lung malformations (controls). Statistical analysis was performed from January 18 to September 6, 2020. Preoperative CT scans were interpreted independently by 9 experienced pediatric radiologists in a blinded fashion and analyzed from January 24, 2019, to September 6, 2020.MAIN OUTCOMES AND MEASURES Accuracy, sensitivity, and specificity of CT in correctly identifying children with malignant tumors. RESULTS Among 477 CT scans identified (282 boys [59%]; median age at CT, 3.6 months [IQR, 1.2-7.2 months]; median age at resection, 6.9 months [IQR, 4.2-12.8 months]), 40 cases were extensively reviewed; 9 cases (23%) had pathologically confirmed cystic PPB. The median age at CT was 7.3 months (IQR, 2.9-22.4 months), and median age at resection was 8.7 months (IQR, 5.0-24.4 months). The sensitivity of CT for detecting PPB was 58%, and the specificity was 83%. High suspicion for malignancy correlated with PPB pathology (odds ratio, 13.5; 95% CI, 2.7-67.3; P = .002).There was poor interrater reliability (κ = 0.36 [range, 0.06-0.64]; P < .001) and no significant difference in specific imaging characteristics between PPB and benign cystic lesions. The overall accuracy rate for distinguishing benign vs malignant lesions was 81%. CONCLUSIONS AND RELEVANCEThis study suggests that chest CT, the current criterion standard imaging modality to assess the lung parenchyma, may not accurately and reliably distinguish PPB from benign congenital lung malformations in children. In any cystic lung lesion without a prenatal diagnosis, operative management to confirm pathologic diagnosis is warranted.
Introduction: To assess fetal vs. neonatal diagnoses, pregnancy outcomes and need for surgery in babies prenatally diagnosed with congenital pulmonary airway malformation (CPAM) or bronchopulmonary sequestration (BPS). Methods: Retrospective single-centre cohort study of fetuses with a prenatal diagnosis of CPAM or BPS between 2006 and 2014. Data collected included serial antenatal ultrasound information and neonatal/infant diagnoses and outcomes. Results: Initial ultrasound diagnosis (n = 63) was CPAM in 51 and BPS in 12: nineteen (30%) fetuses had mediastinal shift and 2 (3%) had hydrops. All neonates with known birth outcome (n = 56) were liveborn. Final diagnosis in 52 infants (83%) with neonatal imaging and/or histopathology confirmed CPAM in 17/44 (39%) and BPS in 6/9 (67%). Of 34 prenatally suspected but unconfirmed CPAM lesions: 10 had no lesion on neonatal imaging, one no neonatal imaging performed, five confirmed BPS, 11 other lung/thoracic lesions, seven were lost to follow-up. There was one infant death. 27/63 infants (43%) had post-natal surgery. More neonates requiring neonatal respiratory support/resuscitation had surgery compared to those who did not (67% vs. 29%, P = 0.008). Patients with suspected CPAM or BPS on both initial and final ultrasound were more likely to have post-natal surgical management than when a lesion was no longer visible on final antenatal ultrasound (68% vs. 23%, P = 0.001). Conclusions: Over 50% of antenatally suspected CPAM/BPS either regressed or had an alternate post-natal diagnosis. Perinatal outcome was good, with the majority of neonates/infants conservatively managed. Persistently visible antenatal lesion and need for neonatal respiratory support predicted ultimate surgical management.
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