Bronchopulmonary sequestration with massive ... - Wiley Online Library

Ultrasound Obstet Gynecol 2014; 44: 441–446 Published online 2 September 2014 in Wiley Online Library (wileyonlinelibrary.com). DOI: 10.1002/uog.13304

Bronchopulmonary sequestration with massive pleural effusion: pleuroamniotic shunting vs intrafetal vascular laser ablation M. R. MALLMANN*, A. GEIPEL*, M. BLUDAU*, K. MATIL†, I. GOTTSCHALK†, ¨ M. HOOPMANN†‡, A. MULLER§, H. BACHOUR¶, A. HEYDWEILLER¶, U. GEMBRUCH* and C. BERG*† *Division of Fetal Surgery, Department of Obstetrics and Prenatal Medicine, University of Bonn, Bonn, Germany; †Division of Prenatal Medicine and Gynecologic Sonography, Department of Obstetrics and Gynecology, University of Cologne, Cologne, Germany; ‡Department of Obstetrics and Gynecology, University of Tuebingen, Tuebingen, Germany; §Department of Neonatology, University of Bonn, Bonn, Germany; ¶Division of Pediatric Surgery, University of Bonn, Bonn, Germany

K E Y W O R D S: bronchopulmonary sequestration; fetus; hydrothorax; laser; prenatal diagnosis; shunt

ABSTRACT Objective To assess the incidence of complications among a relatively large cohort of fetuses with bronchopulmonary sequestration (BPS) and the success of two different intrauterine treatment modalities. Methods All cases with a prenatal diagnosis of BPS detected in a 10-year period (2002–2011) in two tertiary referral centers were reviewed retrospectively for intrauterine course and outcome. Up to May 2010 severe pleural effusions were treated with pleuroamniotic shunting. Thereafter, they were treated with ultrasound-guided laser coagulation of the feeding artery. Results A total of 41 fetuses with BPS were included in the study. In 29 (70.7%) there was no pleural effusion or hydrops and they were treated conservatively. In 19/29 (65.5%) there was partial or complete regression of the lesion during the course of pregnancy. All were born alive (median age at delivery, 38.3 (interquartile range (IQR), 34.0–39.6) weeks) and 16 (55.2%) required sequestrectomy. Intrauterine intervention was performed in all 12 (29.3%) fetuses with pleural effusion. Seven fetuses were treated with pleuroamniotic shunting. One fetus with severe hydrops died in utero. There was no complete regression in any case of BPS in this group. Six infants were born alive (median age, 37.2 (IQR, 30.3–37.4) weeks), of which five (83.3%) required sequestrectomy. Five fetuses were treated with laser ablation of the feeding vessel. In all cases of BPS there was regression after laser ablation. All infants were delivered at term (median age, 39.1 (IQR, 38.0–40.0) weeks).

One (20.0%) neonate required sequestrectomy after birth. Following intrauterine shunt placement complete regression of the lesion was significantly less frequent (0/7 (0%) with shunt placement vs 4/5 (80%) with intrafetal laser treatment) and gestational age at birth was significantly lower, compared to treatment with intrafetal laser. Complete regression of the lesion was also significantly more frequent in the laser group compared to cases without intervention. Conclusion In the absence of pleural effusion, the likelihood of spontaneous regression of BPS is high and the prognosis is therefore favorable. In cases with massive pleural effusion, treatment by laser ablation of the feeding vessel seems to be more effective than is pleuroamniotic shunting, with fewer complications. It might also reduce the need for postnatal surgery. Copyright © 2014 ISUOG. Published by John Wiley & Sons Ltd.

INTRODUCTION Bronchopulmonary sequestration (BPS) is a rare malformation of the respiratory tract consisting of a mass of bronchopulmonary tissue that is separate from the tracheobronchial tree and fed by a separate systemic artery1–3 . This arterial supply typically originates from the descending aorta and occasionally from the intercostal, celiac or splenic arteries, while venous drainage is via the azygos veins or the inferior vena cava. In a rare subset that most likely represents a combination of BPS and congenital pulmonary airway malformation (CPAM), the BPS is fed by pulmonary arteries and is drained by

¨ Pranatale ¨ ¨ ¨ ¨ Correspondence to: Prof. C. Berg, Bereich fur Medizin und Gynakologische Sonographie, Universitatsfrauenklinik Koln, ¨ Kerpenerstr. 34, 50931 Koln, Germany (e-mail: [email protected]) Accepted: 31 December 2013

Copyright © 2014 ISUOG. Published by John Wiley & Sons Ltd.

ORIGINAL PAPER

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The aim of our study was to assess the incidence of complications among a relatively large cohort of cases and to assess the success of two different intrauterine treatment modalities.

METHODS

Figure 1 Grayscale (a) and color Doppler (b) images of bronchopulmonary sequestration (BPS) at 26 weeks of gestation, showing pathognomonic feeding vessel (arrows) arising from the descending aorta.

Figure 2 Bronchopulmonary sequestration (BPS) at 28 weeks of gestation, associated with pleural effusion, polyhydramnios and mediastinal shift.

the pulmonary veins. The key sonographic feature for diagnosing BPS during the fetal period is demonstration of a hyperechoic mass in the thorax that receives an atypical arterial blood supply from the descending aorta4 (Figure 1). The latter enables a differentiation from CPAM of the microcystic type, although a considerable number of lesions show histological features of both diseases as well as those of other foregut malformations5 . Therefore, recently proposed classification systems refer to these ultrasound findings as abnormal lung with abnormal vascular supply (Type III lesion according to the classification proposed by Achiron et al.1 ). BPS usually regresses in the intrauterine period and only few cases are associated with rapid growth and/or pleural effusion (Figure 2) and warrant intrauterine treatment. Intrauterine treatment modalities include pleuroamniotic shunting, alcohol injection, radiofrequency ablation and interstitial laser coagulation. However, because of the rarity of the disease, the true incidence of intrauterine complications is unknown and optimal treatment has yet to be defined.


All cases with a prenatal diagnosis of BPS in a 10-year period (2002–2011) in two neighboring tertiary referral centers (Universities of Bonn and Cologne, Germany) were retrospectively reviewed for intrauterine course and outcome. A prenatal diagnosis of BPS was made in the presence of a hyperechoic lesion in the thorax or abdomen that received its blood supply through an aberrant vessel originating from the descending aorta. Laterality, size, presence of mediastinal shift (cardiac position completely left or right of the midline), relation to the diaphragm, origin of feeding vessel, associated malformations, presence of pleural effusion/hydrops, intrauterine evolution and neonatal outcome were recorded in all cases. During the study period all patients underwent a complete fetal anatomic survey that included fetal echocardiography and Doppler sonography. For all ultrasound examinations, 5-MHz, 7.5-MHz or 9-MHz curved array probes were used (ATL HDI 5000 and IU22 Philips, Hamburg, Germany; Voluson 730 Expert, Voluson E8, GE Healthcare, Solingen, Germany). Intervention was performed by three dedicated specialists in fetal medicine (C.B., A.G. and U.G.). The path of access, as well as the operative technique, was chosen at the discretion of the fetal medicine specialist performing the intervention and was consequently not identical in each case. Up to May 2010 severe pleural effusions were treated by pleuroamniotic shunting with a double pigtail catheter (Harrison Fetal Bladder Stent Set, Cook Medical, Bloomington, IN, USA) using a technique previously described by Rodeck et al.6 . Briefly, under ultrasound guidance, a pleuroamniotic shunt was placed percutaneously into the pleural effusion using a 16-G needle. Fetal anesthesia was not given prior to shunting. Reintervention was undertaken in cases with dislocation of the pigtail catheter and/or recurrence of pleural effusion. After May 2010 ultrasound-guided laser coagulation of the feeding artery using an Nd:YAG (neodymium: yttrium aluminium garnet) laser through an 18-G needle was performed as previously described by Oepkes et al.7 (Figure 3). In all cases fetal anesthesia was performed by an intramuscular injection into the fetal thigh of fentanyl (15 μg/kg) and pancuronium (2 mg/kg), using a 22-G needle with ultrasound guidance. An 18-G needle was then placed into the fetal thorax with its tip directly adjacent to the feeding vessel. A 700-μm laser fiber was moved forward through the needle with the tip of the laser fiber 2–3 mm adjacent to the feeding vessel. The feeding vessel was coagulated using an output of 50 Watts for 5–10 s. If color Doppler demonstrated residual flow, the tip of the laser fiber was repositioned and coagulation was repeated until complete cessation of blood flow.

Ultrasound Obstet Gynecol 2014; 44: 441–446.

Shunt vs laser for bronchopulmonary sequestration

Figure 3 Ultrasound-guided laser coagulation of the feeding artery using an Nd:YAG 700-μm laser fiber ( ) moved through an 18-G needle (arrows).

Because of the extended study period and the retrospective study design, the mean duration of procedures and postoperative complications, such as separation of membranes, were not evaluated. In cases of stable disease or regression of the lesion, delivery and postnatal management were carried out at the discretion of the referring institution. Statistical analysis was performed using the Mann– Whitney U-test. All values are given as median (interquartile range) unless indicated otherwise. A P-value of < 0.05 was considered significant.

RESULTS During the study period 47 diagnoses of BPS were made in the two centers. In three cases the diagnosis was not confirmed after birth. In two of these cases subdiaphragmatic BPS had been diagnosed prenatally. Postnatally, one infant had a jejunal perforation and the other had an adrenal hematoma. In a third case with prenatal diagnosis of a small left-sided extralobar sequestration, an esophageal duplication cyst was the only postnatal finding. Lethal conditions were present in three fetuses (bilateral renal agenesis in two and generalized lymph drainage disorder in one); these cases were therefore excluded from further analysis. Of the 41 remaining fetuses included in the study, four showed additional abnormalities (one each with congenital diaphragmatic hernia, tetralogy of Fallot, hydrocephalus and supraventricular tachycardia). Details of the cohort are given in Table 1. In 29 fetuses (70.7%) with BPS there was no pleural effusion or hydrops and they were treated conservatively with expectant management (Figure 4), including all 5/41 (12.2%) cases with subdiaphragmatic BPS. During the course of pregnancy, complete regression, partial regression and no change were diagnosed in eight (27.6%), 11 (37.9 %) and 10 (34.5%) cases, respectively. There was no case of progressive disease. All infants were born alive with a median age of delivery of 38.3


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(34.0–39.6) weeks. One of the neonates required extracorporeal membrane oxygenation (ECMO) treatment. Sixteen (55.2%) neonates required sequestrectomy and 13 (44.8%) required no intervention. Intrauterine intervention was performed in all 12 (29.3%) fetuses with severe pleural effusion and mediastinal shift, all with left-sided extralobar BPS fed from an aberrant vessel originating from the descending aorta. Seven fetuses were treated exclusively with pleuroamniotic shunting at a median gestational age of 29.3 (25.3–29.5) weeks). A single trocar insertion was performed during each intervention in all cases. Hydrops and polyhydramnios were present in four/seven (57.1%) fetuses. One severely hydropic fetus died despite bilateral shunt placement because of low output cardiac failure. During the course of pregnancy, partial regression, no change and progression in the size of BPS were diagnosed in two (28.6%), four (57.1%) and one (14.3%) case(s), respectively. There was no case of complete regression in any of the cases of BPS in this group. In five/six (83.3%) surviving children pleural effusion resolved after a single shunt placement. In the remaining case three interventions were necessary because of shunt displacement with recurrent pleural effusion. Preterm prelabor rupture of membranes (PPROM) within days after shunt placement occurred in two cases at 33 + 2 and 28 + 3 weeks, respectively. Six infants were born alive with a median gestational age of 37.2 (30.3–37.4) weeks, of which five (83.3%) required sequestrectomy, whereas one did not require any intervention postpartum. Five fetuses were treated with laser ablation of the feeding vessel. In all there was severe pleural effusion, mediastinal shift and polyhydramnios and in one (1/5 (20.0%)) hydrops. Laser treatment was performed at a median gestational age of 30.4 (24.3–31.5) weeks. In all cases, two punctures were performed during each intervention: one for fetal anesthesia and one for the procedure itself. Perfusion of the BPS was successfully disrupted with one intervention in three cases, while in two cases a second intervention within 72 hours was required because of recurrent flow in the feeding vessel. There was no case of intrauterine death and no case of PPROM in this group. In all cases of BPS there was regression after laser ablation (one (20.0%) partial and four (80%) complete). Polyhydramnios, pleural effusion and hydrops resolved in all cases. All infants were delivered at term (39.1 (38.0–40.0) weeks. None required ventilation or ECMO treatment after birth. One neonate required sequestrectomy after birth, whereas four did not require any intervention postpartum. Following intrauterine shunt placement, complete regression of the lesion was significantly less frequent and gestational age at birth was significantly lower compared to those treated with intrafetal laser. Complete regression of the lesion was also significantly more frequent in the laser group compared to cases without intervention. The only cases of PPROM and intrauterine death occurred following shunt placement, although this difference did not reach statistical significance.


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Characteristic Gestational age at diagnosis (weeks) Type of BPS Intralobar Extralobar Side of BPS Right-sided Left-sided Bilateral Subdiaphragmatic Origin of feeding vessel Throracic descending aorta Abdominal descending aorta Pulmonary arteries Pleural effusion Hydrops fetalis Mediastinal shift Polyhydramnios Gestational age at intervention (weeks) PPROM Fetal loss Intrauterine course of BPS Partial regression Complete regression Constant size Increase in size Gestational age at birth (weeks) Birth weight (g)

Type of intervention

No intrauterine intervention (n = 29)

Pleuroamniotic shunt (n = 7)

Intrafetal laser (n = 5)

23.3 (20.4–27.0)*

29.0 (25.2–29.3)*

24.1 (23.3–31.3)

4 (13.8) 25 (86.2)

0 (0) 7 (100)

0 (0) 5 (100)

4 (13.8) 19 (65.5) 1 (3.4) 5 (17.2)

2 (28.6) 5 (71.4) 0 (0) 0 (0)

1 (20) 4 (80) 0 (0) 0 (0)

16 (55.2) 9 (31.0) 4 (13.8) 0 (0) 0 (0) 13 (44.8) 1 (3.4) N/A 0 (0) 0 (0)

7 (100) 0 (0) 0 (0) 7 (100) 4 (57.1) 7 (100) 4 (57.1) 29.3 (25.3–29.5) 2 (28.6) 1 (14.3)

5 (100) 0 (0) 0 (0) 5 (100) 1 (20) 5 (100) 5 (100) 30.4 (24.3–31.5) 0 (0) 0 (0)

11 (37.9) 8 (27.6)† 10 (34.5) 0 (0) 38.3 (34.0–39.6)* 3200 (2070–3650)

2 (28.6) 0 (0)‡ 4 (57.1) 1 (14.3) 37.2 (30.3–37.4)*‡ 2870 (1230–3193)

1 (20) 4 (80)†‡ 0 (0) 0 (0) 39.1 (38.0–40.0)‡ 3240 (2880–3290)

Data are given as median (interquartile range) or n (%). P < 0.05: *no intervention vs shunt; †no intervention vs laser; ‡shunt vs laser. N/A, not applicable; PPROM, preterm prelabor rupture of membranes.

DISCUSSION Fetal lung lesions can be detected reliably by grayscale ultrasound, and Doppler studies have enhanced our ability to identify BPS; however, defining the exact nature of the anomaly may still prove difficult. BPS may have a sonographic appearance similar to that of CPAM, particularly the microcystic type, whereas cystic lesions with the clinical appearance of CPAM may have a systemic blood supply. No clear histological distinction exists between these two entities and mixed histopathological findings of typical CPAM can be seen in cases of BPS and even in bronchial and laryngeal atresia1 . Moreover, two types of BPS can be differentiated histologically: the extralobar type exists outside the pleural investment of the lung, has its own pleural sheath and has an atypical systemic blood supply. In 10% of cases it is located in the pericardial sac or the abdomen and in 90% of cases it is located on the left side. The intralobar type is located within the substance of a lung lobe, is fed by the pulmonary arteries and, therefore, occurs exclusively inside the chest. Most cases diagnosed in the prenatal and neonatal periods are of the extralobar type; therefore, some authors have considered the intralobar type an acquired disease8 . However, with recent advances in imaging techniques, the number of reported neonatal diagnoses of the intralobar type is constantly growing, suggesting a predominantly congenital origin for both intraand extralobar sequestration9 . On prenatal ultrasound


examination, a reliable differentiation between the intralobar type of BPS and the microcystic type of CPAM is not possible. In our current study, four left-sided hyperechoic lesions without pleural effusion were fed by the pulmonary arteries and drained by the pulmonary veins. As there was complete regression until term in all of these cases and lesions were not detectable at postnatal imaging studies, there is no certainty about their true nature. Although a substantial number of echogenic lung lesions are hybrid lesions with concomitance of BPS and CPAM4 and although, in some cases, differentiation between these entities might not be possible at prenatal ultrasound examination, a subgroup of BPS can be distinguished with high reliability: extralobar sequestration with an atypical systemic feeding vessel and associated pleural effusion, features not associated with microcystic CPAM. This distinction is of utmost importance when prenatal intervention is considered. In the absence of severe pleural effusion and mediastinal shift, BPS has a high likelihood of spontaneous regression and therefore has a favorable prognosis7,10 – 12 , which justifies expectant management4 . In our cohort, in 65.5% of the non-hydropic fetuses there was partial or complete regression of the lesion during the course of pregnancy. However, the small subset of fetuses with hydrops is associated with high intrauterine and neonatal mortality and warrants intervention13 . In the case of CPAM, development of hydrops is related to


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445

Fetuses with bronchopulmonary sequestration (n = 44) Lethal condition (n = 3)

No pleural effusion (n = 29)

No intervention (n = 29)

Live birth (n = 29)

Pleural effusion (n = 12)

Pleuroamniotic shunting (n = 7)

IUFD (n = 1)

Live birth (n = 6)

Laser coagulation (n = 5)

Live birth (n = 5)

Sequestrectomy (n = 16)






Figure 4 Flow chart showing management and pregnancy outcome of 41 pregnancies complicated by bronchopulmonary sequestration diagnosed between 2002 and 2011.

the size of the lesion and is usually not associated with pleural effusion14 . For the microcystic type of lesion, intrauterine therapeutic options are limited, including open fetal surgery, laser ablation of the feeding pulmonary vascular supply or steroids4,15,16 . The outcome of fetal surgery and laser ablation in treating CPAM have so far been disappointing, with survival rates below 50%4,15 ; therefore, some authors advocate transplacental steroid treatment for which survival rates as high as 83% have been reported16 . In cases of BPS with hydrops, which are almost invariably associated with massive pleural effusion, the target of intrauterine therapy is either the abnormal systemic feeding vessel or the pleural effusion, and the reported outcome is considerably better. Therapeutic options include inotropic therapy, alcohol ablation, thoracocentesis, pleuroamniotic shunting and percutaneous laser ablation4,7,15,17 – 24 . A larger body of literature exists for fetal pleuroamniotic shunting which was introduced in 1994 and is now used by several centers in treating fetuses with pleural effusion and hydrops20 – 22 . This procedure often results in resolution of hydrops, but might require repeat shunt insertions, due to shunt displacement, and recurrent amnioreductions21 as also corroborated in our cohort. More recently, preliminary results on ultrasoundguided intrafetal laser ablation of the abnormal systemic blood supply of BPS have been reported4,7,15,23,24 . This curative technique might be more effective than drainage of pleural effusion as it targets the tumor rather than its symptoms. Nevertheless, depending on the gestational week and the size of the feeding vessel, more than one


intervention might be necessary to stop the perfusion. In our cohort laser treatment was highly effective and in all cases of BPS there was partial or complete regression once cessation of blood flow was reached. Moreover, it was associated with a significantly lower risk of preterm birth compared to pleuroamniotic shunting. Laser therapy might also reduce the need for postnatal surgery; in those treated with drainage of the effusions postnatal surgery was necessary to remove the tumor in five of six liveborn cases, whereas, in those treated with antenatal occlusion of the feeding vessel, postnatal surgery was necessary in only one of five cases. Our study is limited by a number of factors, namely the retrospective design, differences in morbidity and the fact that postnatal imaging studies and treatment were made at the discretion of the referring institutions. There was hydrops in 57% of fetuses in the shunt group and in 20.0% in the laser group; this difference in morbidity between treatment groups adds a further bias to the results and consequently our conclusion about the preferable form of treatment must be considered carefully. We conclude that, in the absence of pleural effusion, the likelihood of spontaneous BPS regression is high and the prognosis is therefore favorable. In cases with massive pleural effusion, ‘vascular’ laser ablation of the feeding vessel seems to be more effective than pleuroamniotic shunting, with fewer complications. However, as research is ongoing in this field our conclusions should be confirmed by future studies with larger samples and a prospective design. Particularly, the value of laser therapy in reducing the need for postnatal surgery merits further investigation.


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