Perinatal results of conservative management in fetal bronchopulmonary sequestration. Report of a case

Abstract

Introduction. Congenital bronchopulmonary malformations are uncommon but potentially life-threatening abnormalities in fetuses and newborns. They require urgent diagnostic evaluation during the prenatal and neonatal period. In 5.9% of cases, the lesion resolves spontaneously. The scarcity of evidence regarding their natural evolution has led to divergences in the management strategies for prenatal and asymptomatic neonatal cases, resulting in a constantly controversial topic. Prenatal regression is common in cases diagnosed before birth, and most children without symptoms beyond the neonatal period will remain symptom-free during childhood. The lack of statistical information in our country and center, as well as the scarcity of comparative studies on the different available therapeutic methods, make it difficult to formulate unique prenatal therapeutic protocols. We present a clinical case treated from the prenatal period at the University Hospital of Guayaquil in 2023 with an expectant approach to the fetus and resolution of maternal comorbidities, resulting in a favorable perinatal outcome. The spontaneous regression of mild polyhydramnios to severe oligohydramnios in 4 weeks, simultaneous with the resolution of fetal pleural effusion and the rearrangement of previously displaced fetal mediastinal structures by the tumor and tension hydrothorax, is noteworthy. Clinical case. 32-year-old patient, multipara, who was diagnosed late with a pregnancy at 30.1 weeks of gestation complicated by maternal syphilis and fetal bronchopulmonary sequestration (BPS) with pleural effusion and severe displacement of the fetal mediastinum to the right, without fetal hemodynamic compromise. Diagnoses confirmed by obstetric ultrasounds, serial fetal Doppler, and fetal magnetic resonance imaging. The patient develops mild polyhydramnios and is managed expectantly until 37 weeks when, after spontaneous resolution of the hydrothorax, progresses to severe oligohydramnios and is delivered by segmental cesarean section. The newborn undergoes thoracotomy with resection of the lung mass at 7 days old and the diagnosis is confirmed by pathology. Conclusion: We present a clinical case of a rare fetal pathology in all its types worldwide and underdiagnosed in our setting, attended late at 30.1 weeks of gestation with multiple maternal-fetal comorbidities and spontaneous improvement during the prenatal period. It was managed expectantly mainly due to the diagnostic and therapeutic challenges faced, in the absence of a fetal medicine and surgery service in the area. Despite this, expectant fetal management is a valid option internationally, with very favorable outcomes in the majority of cases without hydrops or other signs of fetal cardiac failure. Comparative studies on different prenatal management methods are lacking, partly due to the rarity of the pathology. In our setting, the dispersion of cases and late prenatal diagnosis with limited access to subspecialists in maternal-fetal medicine and fetal surgery may negatively impact perinatal outcomes.