Objective: To assess the outcome of 12 fetuses with bronchopulmonary sequestration (BPS) and massive pleural effusion after intrafetal vascular laser ablation (VLA). Methods: All fetuses with BPS and massive pleural effusion that were treated with intrafetal VLA during a 5-year period (2012-2016) were reviewed for safety, intrauterine course, and postnatal outcome. Results: In the study period, 12 fetuses with BPS were treated with VLA. In 7 (58.3%) fetuses, complete cessation of blood flow was achieved after the first VLA, while in 5 (41.7%) fetuses, residual perfusion of the feeding vessel was demonstrated at follow-up. A second intervention was successful in 4 of 5 (80%) fetuses. Overall, in 11 of 12 (91.7%) fetuses, complete coagulation of the feeding vessel could be achieved, followed by a reduction in size or complete resolution of the BPS. All 11 fetuses with successful prenatal intervention were live-born at a median gestational age of 39+1 (range, 37+5-41+2) weeks. Postnatally, 2 (18.2%) of the 11 newborns underwent sequestrectomy, as well as the preterm newborn on which a second fetal intervention was not feasible. Conclusion: VLA is an effective and safe treatment of BPS that appears to be of benefit in improving prognosis and decreasing the need for postnatal sequestrectomy.

1.
Achiron R, Hegesh J, Yagel S: Fetal lung lesions: a spectrum of disease. New classification based on pathologies, two-dimensional and color Doppler ultrasound. Ultrasound Obstet Gynecol 2004;24:107-114.
2.
Sade RM, Clouse M, Ellis FH Jr: The spectrum of pulmonary sequestration. Ann Thorac Surg 1974;18:644-658.
3.
Kitano Y, Sago H, Hayashi S, Kuroda T, Honna T, Morikawa N: Aberrant venous flow measurement may predict the clinical behavior of a fetal extralobar pulmonary sequestration. Fetal Diagn Ther 2008;23:299-302.
4.
Mallmann MR, Geipel A, Bludau M, Matil K, Gottschalk I, Hoopmann M, Müller A, Bachour H, Heydweiller A, Gembruch U, Berg C: Bronchopulmonary sequestration with massive pleural effusion: pleuroamniotic shunting vs intrafetal vascular laser ablation. Ultrasound Obstet Gynecol 2014;44:441-446.
5.
Cavoretto P, Molina F, Poggi S, Davenport M, Nicolaides KH: Prental diagnosis and outcome of echogenic fetal lung lesions. Ultrasound Obstet Gynecol 2008;32:769-783.
6.
Dolkart LA, Reimers FT, Helmuth WV, Porte MA, Eisinger G: Antenatal diagnosis of pulmonary sequestration: a review. Obstet Gynecol Surv 1992;47:515-520.
7.
Brus F, Nikkels PG, van Loon AJ, Okken A: Non-immune hydrops fetalis and bilateral pulmonary hypoplasia in a newborn infant with extralobar pulmonary sequestration. Acta Paediatrica 1993;82:416-418.
8.
Cruz-Martinez R, Mendez A, Dueñas Riaño J, Ordorica Flores R, Nieto Zermeño J, Malagon-Salazar P, Rebolledo-Fernandez C: Fetal laser surgery prevents fetal death and avoids the need for neonatal sequestrectomy in cases with bronchopulmonary sequestration. Ultrasound Obstet Gynecol 2015;46:627-628.
9.
Adzick NS: Management of fetal lung lesions. Clin Perinatol 2003;30:481-492.
10.
Anandakumar C, Biswas A, Wong YC, Chia D, Annapoorna V, Arulkumaran S, Ratnam S: Management of non-immune hydrops: 8 years' experience. Ultrasound Obstet Gynecol 1997;8:196-200.
11.
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;29:586-589.
12.
Cass DL, Cromblehome TM, Howel LJ, Stafford PW, Ruchelli ED, Adzick NS: Cystic lung lesions with systemic arterial blood supply: a hybrid of congenital cystic adenomatoid malformation and bronchopulmonary sequestration. J Pediatr Surg 1997;32:986-990.
13.
Salomon LJ, Audibert F, Dommergues M, Vial M, Frydman R: Fetal thoracoamniotic shunting as the only treatment for pulmonary sequestration with hydrops: favorable long-term outcome without postnatal surgery. Ultrasound Obstet Gynecol 2003;21:299-301.
14.
Hayashi S, Sago H, Kitano Y, Kuroda T, Honna T, Nakamura T, Ito Y, Kitagawa M, Natori M: Fetal pleuroamniotic shunting for bronchopulmonary sequestration with hydrops. Ultrasound Obstet Gynecol 2006;28:963-967.
15.
Oepkes D, Devlieger R, Lopriore E, Klumper FJCM: Successful ultrasound-guided laser treatment of fetal hydrops caused by pulmonary sequestration. Ultrasound Obstet Gynecol 2007;29:457-459.
16.
Witlox RSGM, Lopriore E, Walther FJ, Rikkers Mutsaerts ERVM, Klumper FJCM, Oepkes D: Single-needle laser treatment with drainage of hydrothorax in fetal bronchopulmonary sequestration with hydrops. Ultrasound Obstet Gynecol 2009;34:355-357.
17.
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;26:1235-1241.
18.
Rammos KS, Foroulis CN, Rammos CK, Andreou A: Prenatal interventional and postnatal surgical therapy of extralobar pulmonary sequestration. Interact Cardiovasc Thorac Surg 2010;10:634-635.
19.
Rodeck CH, Fisk NM, Fraser DI, Nicolini U: Long-term in utero drainage of fetal hydrothorax. N Engl J Med 1988;319:1135-1138.
20.
Hadchouel A, Benachi A, Revillon Y, Rousseau V, Martinovic J, Verkarre V, Dumez Y, Delacourt C: Factors associated with partial and complete regression of fetal lung lesions. Ultrasound Obstet Gynecol 2011;38:88-93.
21.
Ruano R, 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;32:1127-1132.
22.
Shanmugam G, MacArthur K, Pollock JC: Congenital lung malformations - antenatal and postnatal evaluation and management. Eur J Cardiothorac Surg 2005;27:45-52.
23.
Bratu I, Flageole H, Chen MF, DiLorenzo M, Yazbeck S, Laberge JM: The multiple facets of pulmonary sequestration. J Ped Surg 2001;36:784-790.
24.
Fievet L, Natale C, D´Journo XB, Coze S, Dubus JC, Guys JM, Thomas P, Da Lagausie P: Congenital pulmonary airway malformation and sequestration: two standpoints for a single condition. J Minim Access Surg 2015;11:129-133.
25.
Ahmetoglu A, Kosucu P, Imamoglu M, Reis A, Cay A, Gümele HR: Sclerosing haemangioma arising with extralobar pulmonary sequestration. Pediatr Radiol 2003;33:641-643.
26.
Senturk E, Pabuscu E, Sen S, Meteoglu I: A rare association; pulmonary blastoma developing on extrapulmonary sequestration basis. Interact Cardiovasc Thorac Surg 2010;11:110-111.
27.
Lawal L, Mikroulis D, Eleftheriadi S, Karros P, Bougioukas I, Bougioukas G: Adenocarcinoma in pulmonary sequestration. Asian Cardiovasc Thorac Ann 2001;19:433-435.
28.
Crombleholme TM, Coleman B, Hedrick H, Liechty K, Howell L, Flake AW, Johnson M, Adzick NS: Cystic adenomatoid malformation volume ratio predicts outcome in prenatally diagnosed cystic adenomatoid malformation of the lung. J Ped Surg 2002;37:331-338.
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