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Published on: June 29, 2013
Long-Term Neurodevelopmental and Respiratory Outcome after Intrauterine Therapy for Fetal Thoracic Abnormalities
Ruben S G M Witlox1, Enrico Lopriore1, Monique Rijken1
1Division of Neonatology, Department of Pediatrics, Leiden University Medical Center, Leiden, The Netherlands.
Insights
Fetal therapy for thoracic abnormalities shows a 15% rate of severe neurodevelopmental impairment (NDI) in children, necessitating further research. Long-term outcomes require careful monitoring and multicenter studies for better understanding.
Area of Science:
- Pediatric Surgery
- Fetal Medicine
- Neonatal Outcomes
Background:
- Fetal therapy is employed for thoracic abnormalities like congenital cystic adenomatoid malformation and bronchopulmonary sequestration.
- Evaluating long-term neurodevelopmental and respiratory outcomes is crucial for these treated fetuses.
Purpose of the Study:
- To assess neurodevelopmental, motor, cognitive, and respiratory outcomes in children treated with fetal therapy for thoracic abnormalities.
- To evaluate behavioral outcomes using the Child Behavioral Checklist.
Main Methods:
- Children aged ≥18 months underwent neurodevelopmental assessments.
- Medical records were reviewed for respiratory outcomes.
- Parental reports via Child Behavioral Checklist assessed behavior.
Main Results:
- 64% perinatal survival was observed in 63 treated fetuses.
- 15% of 26 children showed severe neurodevelopmental impairment (NDI).
- 15% experienced severe respiratory sequelae; behavioral issues were within norms.
Conclusions:
- Severe NDI occurred in 15% of this high-risk cohort, exceeding rates in other fetal therapy series.
- Multicenter studies and registries are recommended for prospective data collection.
- Long-term outcome data at fixed time points are essential.
Introduction:
The aim of this study is to evaluate long-term neurodevelopmental and respiratory outcome after fetal therapy for fetal pleural effusion, congenital cystic adenomatoid malformation, and bronchopulmonary sequestration.
Methods:
Children ≥18 months of age underwent an assessment of neurologic, motor, and cognitive development. Medical records were reviewed to determine respiratory outcome. Behavioral outcome was assessed using the Child Behavioral Checklist.
Results:
Between 2001 and 2016, 63 fetuses with fetal hydrops secondary to thoracic abnormalities were treated at our center. Overall perinatal survival was 64% (40/63). Twenty-six children were included for follow-up (median age 55 months). Severe neurodevelopmental impairment (NDI) was detected in 15% (4/26). Three out of 4 children with severe NDI had associated causes contributing to the impairment. Overall adverse outcome, including perinatal mortality or NDI, was 55% (27/49). Fifteen percent (4/26) had severe respiratory sequelae. Parents did not report more behavioral problems than Dutch norms.
Discussion:
Our results suggest that severe NDI in this specific high-risk cohort occurs in 15%, which is above the range of the incidence of NDI reported in case series treated with other fetal therapies (5-10%). Large multicenter studies and an international web-based registry are warranted to prospectively gather outcome data at fixed time points.
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