Combined Pre- and Postnatal Minimally Invasive Approach to Complicated Pulmonary Sequestrations

Abstract

Pulmonary sequestration (PS) is mostly asymptomatic but there is a proportion of fetuses that develop hydrops, leading to fetal or neonatal death. Fetal treatments are available, but postnatal management of the residual lesions is not uniformly defined. We present two cases of combined pre- and postnatal minimally invasive approach to complicated extra-lobar PS.

Patient 1 presented with complicated PS at 31 weeks of gestation. Ultrasound-guided laser coagulation of the anomalous artery was successful. The patient was born asymptomatic at 38 weeks. Neonatal magnetic resonance imaging (MRI) showed a residual mass, confirmed by computed tomography (CT) at 6 months. No systemic artery was described, but perfusion was present. We decided for thoracoscopic resection. A residual artery was identified and sealed. Patient 2 presented with complicated PS at 25 weeks of gestation, underwent laser coagulation of the anomalous artery and was born asymptomatic at 38 weeks. Neonatal MRI showed persistence of the lesion, confirmed by CT scan at 4 months. We proceeded with thoracoscopic resection. A residual vessel was ligated. The patients 1 and 2 are now 24 and 21 months old, respectively, and healthy.

Prenatal treatment of complicated PS is a life-saving procedure.

Postnatal thoracoscopic resection of the residual lesion is feasible and safe; we believe it is the best course of treatment to grant the complete excision of the malformation.

Keywords

pulmonary sequestration - fetal therapy - thoracoscopy - laser coagulation - congenital lung malformation

Publication History

Received: 22 November 2019

Accepted: 16 May 2020

Article published online:
18 September 2020

• References

• 1 Riley JS, Urwin JW, Oliver ER. , et al. Prenatal growth characteristics and pre/postnatal management of bronchopulmonary sequestrations. J Pediatr Surg 2018; 53 (02) 265-269
• 2 Biyyam DR, Chapman T, Ferguson MR, Deutsch G, Dighe MK. Congenital lung abnormalities: embryologic features, prenatal diagnosis, and postnatal radiologic-pathologic correlation. Radiographics 2010; 30 (06) 1721-1738
• 3 Zhang H, Tian J, Chen Z. , et al. Retrospective study of prenatal diagnosed pulmonary sequestration. Pediatr Surg Int 2014; 30 (01) 47-53
• 4 Stoiber B, Moehrlen U, Kurmanavicius J. , et al. Congenital lung lesion: prenatal course, therapy and predictors of perinatal outcome. Ultraschall Med 2017; 38 (02) 158-165
• 5 Witlox RS, Lopriore E, Oepkes D, Walther FJ. Neonatal outcome after prenatal interventions for congenital lung lesions. Early Hum Dev 2011; 87 (09) 611-618
• 6 Adzick NS. Management of fetal lung lesions. Clin Perinatol 2003; 30 (03) 481-492
• 7 Baud D, Windrim R, Kachura JR. , et al. Minimally invasive fetal therapy for hydropic lung masses: three different approaches and review of the literature. Ultrasound Obstet Gynecol 2013; 42 (04) 440-448
• 8 Watson J, Campbell S. Antenatal evaluation and management in nonimmune hydrops fetalis. Obstet Gynecol 1986; 67 (04) 589-593
• 9 Im SS, Rizos N, Joutsi P, Shime J, Benzie RJ. Nonimmunologic hydrops fetalis. Am J Obstet Gynecol 1984; 148 (05) 566-569
• 10 Adzick NS. Open fetal surgery for life-threatening fetal anomalies. Semin Fetal Neonatal Med 2010; 15 (01) 1-8
• 11 Cavoretto P, Molina F, Poggi S, Davenport M, Nicolaides KH. Prenatal diagnosis and outcome of echogenic fetal lung lesions. Ultrasound Obstet Gynecol 2008; 32 (06) 769-783
• 12 Oepkes D, Devlieger R, Lopriore E, Klumper FJCM. Successful ultrasound-guided laser treatment of fetal hydrops caused by pulmonary sequestration. Ultrasound Obstet Gynecol 2007; 29 (04) 457-459
• 13 Ruano R, de A Pimenta EJ, Marques da Silva M, Maksoud JG, Zugaib M. Percutaneous intrauterine laser ablation of the abnormal vessel in pulmonary sequestration with hydrops at 29 weeks' gestation. J Ultrasound Med 2007; 26 (09) 1235-1241
• 14 Cruz-Martinez R, Méndez A, Dueñas-Riaño J. , et al. Fetal laser surgery prevents fetal death and avoids the need for neonatal sequestrectomy in cases with bronchopulmonary sequestration. Ultrasound Obstet Gynecol 2015; 46 (05) 627-628
• 15 Ruano R, da Silva MM, Salustiano EMA, Kilby MD, Tannuri U, Zugaib M. Percutaneous laser ablation under ultrasound guidance for fetal hyperechogenic microcystic lung lesions with hydrops: a single center cohort and a literature review. Prenat Diagn 2012; 32 (12) 1127-1132
• 16 Nicolini U, Cerri V, Groli C, Poblete A, Mauro F. A new approach to prenatal treatment of extralobar pulmonary sequestration. Prenat Diagn 2000; 20 (09) 758-760
• 17 Bermúdez C, Pérez-Wulff J, Bufalino G, Sosa C, Gómez L, Quintero RA. Percutaneous ultrasound-guided sclerotherapy for complicated fetal intralobar bronchopulmonary sequestration. Ultrasound Obstet Gynecol 2007; 29 (05) 586-589
• 18 Mallmann MR, Geipel A, Bludau M. , et al. Bronchopulmonary sequestration with massive pleural effusion: pleuroamniotic shunting vs intrafetal vascular laser ablation. Ultrasound Obstet Gynecol 2014; 44 (04) 441-446
• 19 Macchini F, Gentilino V, Morandi A, Leva E. Thoracoscopic removal of retained thoracoamniotic shunt catheters in newborns. J Laparoendosc Adv Surg Tech A 2014; 24 (11) 827-829
• 20 Bullard KM, Harrison MR. Before the horse is out of the barn: fetal surgery for hydrops. Semin Perinatol 1995; 19 (06) 462-473
• 21 Macchini F, Borzani I, Cavalli S. , et al. Thoracoscopic resection of congenital lung malformation: looking for the right preoperative assessment. Eur J Pediatr Surg 2019 (e-pub ahead of print). Doi: 10.1055/s-0039-1696669