Ultrasound Obstet Gynecol 2015; 46: 627–632 Published online 30 September 2015 in Wiley Online Library (wileyonlinelibrary.com).
Letters to the Editor
Fetal laser surgery prevents fetal death and avoids the need for neonatal sequestrectomy in cases with bronchopulmonary sequestration Bronchopulmonary sequestration (BPS) is a lung mass that has no tracheobronchial connection and receives its blood supply from an aberrant systemic feeding artery, originating from the descending aorta. The perinatal prognosis depends on the presence of hydrops, massive fetal pleural effusion and/or lung hypoplasia1 . However, virtually all fetuses, whether they have complicated or uncomplicated lesions, require postnatal surgery with thoracic sequestrectomy to prevent respiratory morbidity during childhood1 . For fetuses with hydrops and/or massive pleural effusion, percutaneous ultrasound-guided laser ablation of the feeding artery has been attempted as an alternative to improve survival1 – 4 but its impact in decreasing the need for postnatal surgery is uncertain. We describe our experience of eight fetuses with isolated BPS complicated by hydrops and/or massive pleural effusion which underwent fetal surgery with full laser ablation of the feeding artery (FLAFA) as described previously2 . Briefly, FLAFA was performed under maternal local anesthesia and fetal anesthesia, injected under ultrasound guidance into the fetal leg or arm. An 18-G needle was introduced percutaneously, directly into the amniotic cavity through the maternal abdominal wall. Under ultrasound guidance, the needle was directed through the fetal thorax to the lung mass and advanced 2 mm to the main systemic feeding artery. The 600-nm laser fiber was inserted through the needle and, at a power of 25 Watts, contacted the aberrant artery for 5–10 s, achieving coagulation (Figure 1). If power Doppler
demonstrated residual flow to the lung mass on fetal follow-up, a repeat procedure was performed until there was complete cessation of blood flow to the mass. Fetal laser surgery was performed at a median gestational age of 27.1 (range, 20.6–31.7) weeks. No complications such as fetal bleeding, placental abruption, uterine bleeding, uterine contractions requiring active maternal tocolysis, or premature rupture of membranes were observed. Blood supply to the mass was disrupted completely with one intervention in five cases, while in three (42.9%) cases flow in the feeding artery reappeared within 72 h after surgery and therefore a second fetal intervention was required. After fetal surgery, a progressive decrease in the lung mass size, an increase in both pulmonary dimensions and disappearance of fetal effusions were observed in all cases (Figure 2). All cases were delivered liveborn at term, at a median gestational age of 38.9 (range, 37.6–39.7) weeks resulting in a perinatal survival rate of 100%. The pediatric surgeons determined the need for neonatal surgery based on the presence of mediastinal shift, respiratory morbidity or persistence of blood supply to the lung mass on computed tomographic angiography (CTA). In all cases, a remaining small avascular mass without mediastinal shift was observed by fetal ultrasound before delivery and by neonatal CTA. All neonates were asymptomatic; none required mechanical ventilation or oxygen support, and consequently no need for neonatal sequestrectomy. In agreement with cases reported previously1 – 4 , our case series provides evidence that successful FLAFA induces a necrotic process in the solid lesion with progressive lung mass regression and disappearance of hydrops, thus preventing fetal demise and even avoiding the need for neonatal surgery. However, it is of note
Figure 1 Ultrasound images of fetal thorax showing bronchopulmonary sequestration (BPS). (a) Sagittal view showing massive pleural effusion, ascites and a large and hyperechogenic lung mass (BPS) with a systemic feeding artery arising from the descending aorta. (b) Transverse view with color Doppler revealing BPS with feeding artery and showing an 18-G needle containing the laser fiber during fetal surgery. (c) Complete disappearance of the feeding artery is observed at the end of fetal surgery.
Copyright © 2015 ISUOG. Published by John Wiley & Sons Ltd.
LETTERS TO THE EDITOR
Letters to the Editor
628
Figure 2 Transverse ultrasound images of fetal thorax at level of four-chamber view, showing intrathoracic sequence of changes: (a) before fetal intervention; (b) 1 week after fetal intervention; and (c) 4 weeks after fetal intervention. Note regression in size of lung mass ( ), associated with increasing dimensions of both lungs and decrease in fetal pleural effusion.
that neonatal sequestrectomy could not be avoided in all BPS cases reported in the literature that were treated by FLAFA. Complete disruption of blood flow to the lung mass appears to be critical to the success of this fetal intervention. While cases with no residual blood flow after surgery showed regression of the lung mass during fetal follow-up2 – 4 , those with blood supply remaining did not and postnatal sequestrectomy was required1,5 . Our findings support the use of FLAFA as a therapeutic option for fetuses with BPS complicated by hydrops and/or massive pleural effusion. It remains to be determined whether there is a role for such minimally invasive fetal surgery to avoid postnatal surgery even in uncomplicated cases without pleural effusions and an unchanging lung mass during pregnancy but who may require postnatal sequestrectomy. To address this issue, long-term follow-up studies on cases with BPS treated with FLAFA and without subsequent neonatal sequestrectomy are warranted.
Acknowledgments R. Cruz-Martinez was supported by the Mexican National Council for Science and Technology (CONACyT) and wishes to thank Kristen Foundation and KPI Ultrasonidos Mexico for supporting the fetal surgery project in Queretaro, Mexico. R. Cruz-Martinez*†‡§, A. M´endez†‡§, ˜ ˜ J. Duenas-Ria no§, R. Ordorica-Flores¶,
Copyright © 2015 ISUOG. Published by John Wiley & Sons Ltd.
˜ ´ J. Nieto-Zermeno¶, P. Malagon-Salazar†‡, ´ C. Medina-Noyola†‡ and C. Rebolledo-Fernandez†‡§ †Fetal Medicine and Surgery Research Unit, Children’s and Women’s Specialty Hospital, Quer´etaro, Mexico; ‡Unidad de Investigacion ´ en Neurodesarrollo ´ ‘Dr Augusto Fernandez Guardiola’, Instituto de Neurobiolog´ıa, Universidad Nacional Autonoma de M´exico (UNAM) Campus Juriquilla, ´ Quer´etaro, M´exico; §Fetal Medicine Mexico Research Group, Mexico; ¶Department of Pediatric Surgery, Hospital Infantil de Mexico ‘Dr. Federico Gomez’, ´ Mexico City, Mexico *Correspondence. (e-mail: [email protected]) DOI: 10.1002/uog.14921
References 1. Cavoretto P, Molina F, Poggi S, Davenport M, Nicolaides KH. Prenatal diagnosis and outcome of echogenic fetal lung lesions. Ultrasound Obstet Gynecol 2008; 32: 769–783. 2. Oepkes D, Devlieger R, Lopriore E, Klumper FJ. Successful ultrasound-guided laser treatment of fetal hydrops caused by pulmonary sequestration. Ultrasound Obstet Gynecol 2007; 29: 457–459. 3. Witlox RS, Lopriore E, Walther FJ, Rikkers-Mutsaerts ER, Klumper FJ, Oepkes D. Single-needle laser treatment with drainage of hydrothorax in fetal bronchopulmonary sequestration with hydrops. Ultrasound Obstet Gynecol 2009; 34: 355–357. 4. Mallmann MR, Geipel A, Bludau M, Matil K, Gottschalk I, Hoopmann M, Muller 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. Ruano R, de APEJ, 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.
Ultrasound Obstet Gynecol 2015; 46: 627–632.
Letters to the Editor
Fetal laser surgery prevents fetal death and avoids the need for neonatal sequestrectomy in cases with bronchopulmonary sequestration Bronchopulmonary sequestration (BPS) is a lung mass that has no tracheobronchial connection and receives its blood supply from an aberrant systemic feeding artery, originating from the descending aorta. The perinatal prognosis depends on the presence of hydrops, massive fetal pleural effusion and/or lung hypoplasia1 . However, virtually all fetuses, whether they have complicated or uncomplicated lesions, require postnatal surgery with thoracic sequestrectomy to prevent respiratory morbidity during childhood1 . For fetuses with hydrops and/or massive pleural effusion, percutaneous ultrasound-guided laser ablation of the feeding artery has been attempted as an alternative to improve survival1 – 4 but its impact in decreasing the need for postnatal surgery is uncertain. We describe our experience of eight fetuses with isolated BPS complicated by hydrops and/or massive pleural effusion which underwent fetal surgery with full laser ablation of the feeding artery (FLAFA) as described previously2 . Briefly, FLAFA was performed under maternal local anesthesia and fetal anesthesia, injected under ultrasound guidance into the fetal leg or arm. An 18-G needle was introduced percutaneously, directly into the amniotic cavity through the maternal abdominal wall. Under ultrasound guidance, the needle was directed through the fetal thorax to the lung mass and advanced 2 mm to the main systemic feeding artery. The 600-nm laser fiber was inserted through the needle and, at a power of 25 Watts, contacted the aberrant artery for 5–10 s, achieving coagulation (Figure 1). If power Doppler
demonstrated residual flow to the lung mass on fetal follow-up, a repeat procedure was performed until there was complete cessation of blood flow to the mass. Fetal laser surgery was performed at a median gestational age of 27.1 (range, 20.6–31.7) weeks. No complications such as fetal bleeding, placental abruption, uterine bleeding, uterine contractions requiring active maternal tocolysis, or premature rupture of membranes were observed. Blood supply to the mass was disrupted completely with one intervention in five cases, while in three (42.9%) cases flow in the feeding artery reappeared within 72 h after surgery and therefore a second fetal intervention was required. After fetal surgery, a progressive decrease in the lung mass size, an increase in both pulmonary dimensions and disappearance of fetal effusions were observed in all cases (Figure 2). All cases were delivered liveborn at term, at a median gestational age of 38.9 (range, 37.6–39.7) weeks resulting in a perinatal survival rate of 100%. The pediatric surgeons determined the need for neonatal surgery based on the presence of mediastinal shift, respiratory morbidity or persistence of blood supply to the lung mass on computed tomographic angiography (CTA). In all cases, a remaining small avascular mass without mediastinal shift was observed by fetal ultrasound before delivery and by neonatal CTA. All neonates were asymptomatic; none required mechanical ventilation or oxygen support, and consequently no need for neonatal sequestrectomy. In agreement with cases reported previously1 – 4 , our case series provides evidence that successful FLAFA induces a necrotic process in the solid lesion with progressive lung mass regression and disappearance of hydrops, thus preventing fetal demise and even avoiding the need for neonatal surgery. However, it is of note
Figure 1 Ultrasound images of fetal thorax showing bronchopulmonary sequestration (BPS). (a) Sagittal view showing massive pleural effusion, ascites and a large and hyperechogenic lung mass (BPS) with a systemic feeding artery arising from the descending aorta. (b) Transverse view with color Doppler revealing BPS with feeding artery and showing an 18-G needle containing the laser fiber during fetal surgery. (c) Complete disappearance of the feeding artery is observed at the end of fetal surgery.
Copyright © 2015 ISUOG. Published by John Wiley & Sons Ltd.
LETTERS TO THE EDITOR
Letters to the Editor
628
Figure 2 Transverse ultrasound images of fetal thorax at level of four-chamber view, showing intrathoracic sequence of changes: (a) before fetal intervention; (b) 1 week after fetal intervention; and (c) 4 weeks after fetal intervention. Note regression in size of lung mass ( ), associated with increasing dimensions of both lungs and decrease in fetal pleural effusion.
that neonatal sequestrectomy could not be avoided in all BPS cases reported in the literature that were treated by FLAFA. Complete disruption of blood flow to the lung mass appears to be critical to the success of this fetal intervention. While cases with no residual blood flow after surgery showed regression of the lung mass during fetal follow-up2 – 4 , those with blood supply remaining did not and postnatal sequestrectomy was required1,5 . Our findings support the use of FLAFA as a therapeutic option for fetuses with BPS complicated by hydrops and/or massive pleural effusion. It remains to be determined whether there is a role for such minimally invasive fetal surgery to avoid postnatal surgery even in uncomplicated cases without pleural effusions and an unchanging lung mass during pregnancy but who may require postnatal sequestrectomy. To address this issue, long-term follow-up studies on cases with BPS treated with FLAFA and without subsequent neonatal sequestrectomy are warranted.
Acknowledgments R. Cruz-Martinez was supported by the Mexican National Council for Science and Technology (CONACyT) and wishes to thank Kristen Foundation and KPI Ultrasonidos Mexico for supporting the fetal surgery project in Queretaro, Mexico. R. Cruz-Martinez*†‡§, A. M´endez†‡§, ˜ ˜ J. Duenas-Ria no§, R. Ordorica-Flores¶,
Copyright © 2015 ISUOG. Published by John Wiley & Sons Ltd.
˜ ´ J. Nieto-Zermeno¶, P. Malagon-Salazar†‡, ´ C. Medina-Noyola†‡ and C. Rebolledo-Fernandez†‡§ †Fetal Medicine and Surgery Research Unit, Children’s and Women’s Specialty Hospital, Quer´etaro, Mexico; ‡Unidad de Investigacion ´ en Neurodesarrollo ´ ‘Dr Augusto Fernandez Guardiola’, Instituto de Neurobiolog´ıa, Universidad Nacional Autonoma de M´exico (UNAM) Campus Juriquilla, ´ Quer´etaro, M´exico; §Fetal Medicine Mexico Research Group, Mexico; ¶Department of Pediatric Surgery, Hospital Infantil de Mexico ‘Dr. Federico Gomez’, ´ Mexico City, Mexico *Correspondence. (e-mail: [email protected]) DOI: 10.1002/uog.14921
References 1. Cavoretto P, Molina F, Poggi S, Davenport M, Nicolaides KH. Prenatal diagnosis and outcome of echogenic fetal lung lesions. Ultrasound Obstet Gynecol 2008; 32: 769–783. 2. Oepkes D, Devlieger R, Lopriore E, Klumper FJ. Successful ultrasound-guided laser treatment of fetal hydrops caused by pulmonary sequestration. Ultrasound Obstet Gynecol 2007; 29: 457–459. 3. Witlox RS, Lopriore E, Walther FJ, Rikkers-Mutsaerts ER, Klumper FJ, Oepkes D. Single-needle laser treatment with drainage of hydrothorax in fetal bronchopulmonary sequestration with hydrops. Ultrasound Obstet Gynecol 2009; 34: 355–357. 4. Mallmann MR, Geipel A, Bludau M, Matil K, Gottschalk I, Hoopmann M, Muller 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. Ruano R, de APEJ, 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.
Ultrasound Obstet Gynecol 2015; 46: 627–632.
