Prenatal Diagnosis and Evaluation of Sonographic Predictors for Intervention and Adverse Outcome in Congenital Pulmonary Airway Malformation

Hellmund, Astrid; Berg, Christoph; Geipel, A.; Bludau, Meike; Heydweiller, Andreas; Bachour, H.; Müller, Andreas; Müller, Annette; Gembruch, Ulrich

doi:10.1371/journal.pone.0150474

Cited by 64 publications

(50 citation statements)

References 59 publications

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“…The CVR is the most common standard assessment of a fetal CLL . Other US measurements, such as the mass‐to‐thorax ratio and lung‐to‐head ratio, have been assessed but were not found to be easier to obtain or superior to the CVR . All of these measurements require considerable technical expertise and depend on accurate measurements of the lesion size throughout gestation.…”

Section: Discussionmentioning

confidence: 99%

Utility of Fetal Cardiac Axis and Cardiac Position Assessment in Predicting Neonatal Respiratory Morbidity in Fetal Congenital Lung Lesions

Tuzović

Copel

Stitelman

et al. 2019

J of Ultrasound Medicine

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Objectives-To assess the diagnostic performance of the fetal cardiac axis (CA) and/or cardiac position (CP) versus the congenital pulmonary malformation volume ratio (CVR) in predicting any and severe neonatal respiratory morbidity in fetal congenital lung lesions.Methods-This work was an 11-year retrospective cohort study. The sensitivity, specificity, positive predictive value, and negative predictive value of CA and/or CP assessment in prediction of respiratory morbidity were calculated before 24 weeks' gestation and between 24 and 32 weeks and compared to CVR cutoffs obtained from the literature.Results-Fifty-three patients were included. CA and/or CP abnormalities were present in 45% and 38% of patients before 24 weeks and between 24 and 32 weeks and were significantly more common in left-versus rightsided lesions (60% versus 17%; P = .003). The sensitivity, specificity, positive predictive value, and negative predictive value of an abnormal CA and/or CP for any and severe respiratory morbidity were 0.67, 0.61, 0.33, and 0.86 and 0.8, 0.58, 0.17, and 0.97 before 24 weeks and 0.75, 0.73, 0.45, and 0.91 and 0.8, 0.67, 0.20, and 0.97 between 24 and 32 weeks, respectively. An abnormal CA and/or CP had higher sensitivity for any respiratory morbidity compared to the CVR at 0.5 and 0.8 cutoffs both before 24 weeks and between 24 and 32 weeks (P < .05).Conclusions-An abnormal CA and/or CP before 24 weeks and between 24 and 32 weeks has higher sensitivity for the detection of any respiratory morbidity at birth compared to the CVR at both 0.5 and 0.8 cutoffs. A normal CA and CP have a high negative predictive value for excluding any respiratory morbidity at birth both before 24 weeks and between 24 and 32 weeks.

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Section: Discussionmentioning

confidence: 99%

Utility of Fetal Cardiac Axis and Cardiac Position Assessment in Predicting Neonatal Respiratory Morbidity in Fetal Congenital Lung Lesions

Tuzović

Copel

Stitelman

et al. 2019

J of Ultrasound Medicine

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“…However, a comparative prospective study including noncomplicated BPS cases with severe pulmonary compression but no fetal fluid effusions, with and without fetal intervention, is warranted to address this issue. It is important to note that although the usefulness of the LHR in assessing lung growth was first described and validated for fetuses with congenital diaphragmatic hernia, a recent study including fetuses with lung masses has demonstrated its clinical utility in predicting adverse perinatal outcomes [20].…”

Section: Discussionmentioning

confidence: 99%

Thoracic Changes after Full Laser Ablation of the Feeding Artery in Fetuses with Bronchopulmonary Sequestration

Cruz‐Martínez

Nieto-Castro²,

Martínez‐Rodríguez³

et al. 2017

Fetal Diagn Ther

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Objective: To assess longitudinal intrathoracic changes after fetal laser surgery in fetuses with bronchopulmonary sequestration (BPS) with hydrops and/or hydrothorax. Study Design: The presence of intrafetal fluid effusions, the lung mass volume ratio (congenital pulmonary airway malformation volume ratio [CVR]), and the observed/expected lung-to-head circumference ratio (O/E-LHR) of both lungs were evaluated in a cohort of BPS fetuses with hydrops and/or hydrothorax treated with full laser ablation of the feeding artery (FLAFA). The longitudinal changes in intrafetal fluid effusions, lung mass volume, and pulmonary growth were analyzed by survival and multilevel analysis against days after FLAFA. Results: FLAFA was successfully performed in 15 cases at a median gestational age of 26.9 weeks. A complete disappearance of the hydrops and hydrothorax was observed a median interval of 7.5 and 21 days after the fetal intervention, respectively. A progressive decrease in the CVR and an increment in the size of both lungs were observed after FLAFA. The O/E-LHR of the lung contralateral and ipsilateral to the side of the BPS became normal on average 8 and 10 weeks after FLAFA, respectively. Conclusion: Fetal laser surgery with FLAFA promotes disappearance of all fetal fluid effusions, a lung mass regression sequence, and a normalization of pulmonary growth.

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“…The role of MRI in this setting remains controversial [31,32]. Less than half of prenatally diagnosed patients were followed by a calculated CVR despite multiple studies now demonstrating its prognostic value, both in terms of predicting hydrops risk as well as determining likelihood of respiratory distress at birth, among other outcome measures [12,[33][34][35][36].…”

Section: Accepted Manuscriptmentioning

confidence: 99%

Development of a multi-institutional registry for children with operative congenital lung malformations

Kunisaki

Saito

Fallat

et al. 2020

Journal of Pediatric Surgery

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Introduction: The purpose of this study was to develop a multi-institutional registry to characterize the demographics, management, and outcomes of a contemporary cohort of children undergoing congenital lung malformation (CLM) resection. Methods: After central reliance IRB approval, a web-based, secure database was created to capture retrospective cohort data on pathologically-confirmed CLMs performed between 2009-2015 within a multi-institutional research collaborative. Results: Eleven children's hospitals contributed 506 patients. Among 344 prenatally diagnosed lesions, the congenital pulmonary airway malformation volume ratio was measured in 49.1%, and fetal MRI was performed in 34.3%. One hundred thirty-four (26.7%) children had respiratory symptoms at birth. Fifty-eight (11.6%) underwent neonatal resection, 322 (64.1%) had surgery at 1-12 months, and 122 (24.3%) had operations after 12 mos. The median age at resection was 6.7 months (interquartile range, 3.6-11.4). Among 230 elective lobectomies performed in asymptomatic patients, thoracoscopy was successfully utilized in 102 (44.3%), but there was substantial variation across centers. The most common lesions were congenital pulmonary airway malformation (n=234, 47.3%) and intralobar bronchopulmonary sequestration (n=106, 21.4%). Conclusion: This multicenter cohort study on operative CLMs highlights marked disease heterogeneity and substantial practice variation in preoperative evaluation and operative management. Future registry studies are planned to help establish evidencebased guidelines to optimize the care of these patients.

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Prenatal Diagnosis and Evaluation of Sonographic Predictors for Intervention and Adverse Outcome in Congenital Pulmonary Airway Malformation

Cited by 64 publications

References 59 publications

Utility of Fetal Cardiac Axis and Cardiac Position Assessment in Predicting Neonatal Respiratory Morbidity in Fetal Congenital Lung Lesions

Utility of Fetal Cardiac Axis and Cardiac Position Assessment in Predicting Neonatal Respiratory Morbidity in Fetal Congenital Lung Lesions

Thoracic Changes after Full Laser Ablation of the Feeding Artery in Fetuses with Bronchopulmonary Sequestration

Development of a multi-institutional registry for children with operative congenital lung malformations

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