Rodrigo Ruano, Eniola R. Ibirogba, Michelle A. Wyatt, Karthik Balakrishnan, M. Yasir Qureshi, Amy B. Kolbe, Joseph A. Dearani, R. Paul Boesch, Leal Segura, Katherine W. Arendt, Ellen Bendel-Stenzel, Shana S. Salik, Denise B. Klinkner
Mayo Clinic College of Medicine.
United States
Fetal Diagnosis and Therapy
Fetal Diagn Ther 2021; 48: 70-77
DOI: 10.1159/000510635
Abstract
Introduction: In utero interventions are performed in fetuses with “isolated” major congenital anomalies to improve neonatal outcomes and quality of life. Sequential in utero interventions to treat 2 anomalies in 1 fetus have not yet been described.
Case presentation: Here, we report a fetus with a large left-sided intralobar bronchopulmonary sequestration (BPS) causing mediastinal shift, a small extralobar BPS, and concomitant severe left-sided congenital diaphragmatic hernia (CDH). At 26-week gestation, the BPS was noted to be increasing in size with a significant reduction in right lung volume and progression to fetal hydrops. The fetus underwent ultrasound-guided ablation of the BPS feeding vessel leading to complete tumor regression. However, lung development remained poor (O/E-LHR: 0.22) due to the left-sided CDH, prompting fetal endoscopic tracheal occlusion therapy at 28-week gestation to allow increased lung growth. After vaginal delivery, the newborn underwent diaphragmatic repair with resection of the extralobar sequestration. He was discharged home with tracheostomy on room air at 9 months.
Discussion/conclusion: Sequential in utero interventions to treat 2 severe major anomalies in the same fetus have not been previously described. This approach may be a useful alternative in select cases with otherwise high morbidity/mortality. Further studies are required to confirm our hypothesis.
Category
Abnormal Systemic to Pulmonary Arterial Collaterals or Connections
Surgical and Catheter-mediated Interventions for Pulmonary Vascular Disease
Age Focus: Pediatric Pulmonary Vascular Disease
Fresh or Filed Publication: Filed (PHiled). Greater than 1-2 years since publication
Article Access
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