This study compared the outcomes of pleuroamniotic shunting versus intrafetal laser ablation of the feeding vessel in treating bronchopulmonary sequestration (BPS) with massive pleural effusion. The study found that intrafetal laser ablation resulted in a higher rate of complete regression of BPS and higher gestational age at birth, compared to shunting. Laser ablation may be a more effective treatment than shunting for BPS with massive pleural effusion. However, the retrospective design limits conclusions about the preferable treatment approach.

1. UOG Journal Club: October 2014
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.Müller, H. Bachour, A. Heydweiller, U. Gembruch, C. Berg
Volume 44, Issue 4, Date: October 2014, pages 441 - 446
Journal Club slides prepared by Dr Leona Poon
(UOG Editor for Trainees)

2. • Bronchopulmonary sequestration (BPS) and congenital pulmonary
airway malformation (CPAM) are rare lung malformations consisting
of a mass of bronchopulmonary tissue that is separate from the
tracheobronchial tree
Achiron R et al. Ultrasound Obstet Gynecol 2004;24:107-14.
Pryce DM et al. J Pathol Bacteriol 1946;58:457-67.
Sade RM et al. Ann Thorac Surg 1974;18:644-58.
Cavoretto P et al. Ultrasound Obstet Gynecol 2008;32:769-83
Figure 1. Grayscale (a) and color Doppler (b) images of
BPS at 26 weeks’ gestation, showing a feeding vessel (arrows)
arising from the descending aorta.
• The key sonographic
feature for distinguishing
BPS from CPAM is
demonstration of
separate systemic artery,
typically originating from
the descending aorta

3. • A considerable number of echogenic lung lesions show histological
features of both BPS and CPAM.
• BPS usually regresses in intrauterine period and only few cases are
associated rapid growth and/or pleural effusion and warrant
intrauterine treatment.
Achiron R et al. Ultrasound Obstet Gynecol 2004;24:107-14.
Figure 2. BPS at 28 weeks’ gestation, associated with pleural
effusion, polyhydramnios and mediastinal shift
•Intrauterine treatments include
pleuroamniotic shunting,
alcohol injection,
radiofrequency ablation and
interstitial laser coagulation

4. Bronchopulmonary sequestration with massive pleural effusion:
pleuroamniotic shunting vs intrafetal vascular laser ablation
Mallmann et al., UOG 2014
Objective
To assess the incidence of complications among a
relatively large cohort of fetuses with BPS and the
success of two different intrauterine treatment
modalities

5. Bronchopulmonary sequestration with massive pleural effusion:
pleuroamniotic shunting vs intrafetal vascular laser ablation
Mallmann et al., UOG 2014
Patients and Methods
• Retrospective review of all cases with a prenatal diagnosis of BPS detected in a 10-year period
(2002-2011) in 2 tertiary referral centers (Universities of Bonn and Cologne, Germany)
• Laterality, size, presence of mediastinal shift, 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.
• Intervention was performed by 3 dedicated specialists in fetal medicine (C.B., A.G. and U.G.).
The path of access and operative technique were chosen at the discretion of the fetal medicine
specialist performing the intervention.

6. Bronchopulmonary sequestration with massive pleural effusion:
pleuroamniotic shunting vs intrafetal vascular laser ablation
Mallmann et al., UOG 2014
Patients and Methods
• Up to May 2010 severe pleural effusions were treated with pleuroamniotic
shunting.
• After May 2010 ultrasound-guided laser coagulation of the feeding artery using
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)
an Nd:YAG laser through an 18-G needle was performed.
A 700-μm laser fiber was moved forward until 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 5-10s. If color Doppler demonstrated residual
flow, the tip of the laser fiber was repositioned and
coagulation was repeated until complete cessation of
blood flow.

7. Bronchopulmonary sequestration with massive pleural effusion:
pleuroamniotic shunting vs intrafetal vascular laser ablation
Mallmann et al., UOG 2014
Patients and Methods
• Due to 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.

8. Bronchopulmonary sequestration with massive pleural effusion:
pleuroamniotic shunting vs intrafetal vascular laser ablation
Mallmann et al., UOG 2014
Results
• 41 fetuses with BPS were included in the study. Four showed additional
abnormalities (one each with congenital diaphragmatic hernia, tetralogy of
Fallot, hydrocephalus and supraventricular tachycardia)
• In 29 cases treated conservatively. Complete regression, partial regression and
no change were diagnosed in 8 (27.6), 11 (37.9%), and 10 (34.5%) cases,
respectively
• Intrauterine intervention was performed in all 12 (29.3%) fetuses with severe
pleural effusion and mediastinal shift, all with left-sided extralobar BPS
• 7 fetuses were treated with pleuroamniotic shunting at 29.3 (25.3-29.5) weeks

9. Bronchopulmonary sequestration with massive pleural effusion:
pleuroamniotic shunting vs intrafetal vascular laser ablation
Mallmann et al., UOG 2014
Results
• 5 fetuses were treated with laser ablation of feeding vessel at 30.4 (24.3-31.5)
weeks:
• 2 cases required a 2nd intervention within 72 hours because of recurrent
flow in the feeding vessel.
• Complete and partial regression were diagnosed in 4 (80.0%) and 1
(20.0%) case(s), respectively.
• Following intrauterine shunt placement complete regression of the lesion was
significant less frequent (0/7 with shunt placement vs 4/5 with intrafetal laser
treatment) and GA 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.

10. Bronchopulmonary sequestration with massive pleural effusion:
pleuroamniotic shunting vs intrafetal vascular laser ablation
Mallmann et al., UOG 2014
Results
Table 1. Details of 41 fetuses with BPS diagnosed over a period of 10 years.
Characteristics
P<0.05: *no intervention vs shunt; ♯no intervention vs laser; §shunt vs laser.
No intrauterine
intervention
(n=29)
Type of intervention
Pleuroamniotic shunt
(n=7)
Intrafetal laser
(n=5)
GA at diagnosis (w) 23.3 (20.4-27.0)* 29.0 (25.2-29.3)* 24.1 (34.4-31.3)
Type of BPS Intralobar 4 (13.8) 0 (0) 0 (0)
Extralobar 25 (86.2) 7 (100) 5 (100)
Side of BPS Unilateral 23 (79.3) 7 (100) 5 (100)
Bilateral 1 (3.4) 0 (0) 0 (0)
Pleural effusion 0 (0) 7 (100) 5 (100)
Hydrops fetalis 0 (0) 4 (57.1) 1 (20)
Mediastinal shift 13 (44.8) 7 (100) 5 (100)
Polyhydramnios 1 (3.4) 4 (57.1) 5 (100)
Fetal loss 0 (0) 1 (14.3) 0 (0)
Complete regression of BPS 8 (27.6)♯ 0 (0)§ 4 (80)♯§
GA at birth (w) 38.3 (34.0-39.6)* 37.2 (30.3-37.4)*§ 39.1 (38.0-40.0)§

11. Bronchopulmonary sequestration with massive pleural effusion:
pleuroamniotic shunting vs intrafetal vascular laser ablation
Mallmann et al., UOG 2014
Results
Fetuses with BPS (n=44)
Lethal condition (n=3)
No pleural effusion (n=29) Pleural effusion (n=12)
No intervention
(n=29)
Pleuroamniotic
shunting (n=7)
IUFD
(n=1)
Laser coagulation
(n=5)
Live birth
(n=29)
Live birth
(n=6)
Live birth
(n=5)
Sequestrectomy
(n=16)
Sequestrectomy
(n=5)
No intervention
(n=1)
Sequestrectomy
(n=1)
No intervention
(n=4)
No intervention
(n=13)
Figure 4. Flow chart showing management and pregnancy outcome of 41
pregnancies complicated by BPS

12. Bronchopulmonary sequestration with massive pleural effusion:
pleuroamniotic shunting vs intrafetal vascular laser ablation
Mallmann et al., UOG 2014
Discussion
• This study demonstrates that 65.5% of non-hydropic fetuses there
was partial or complete regressions of the lesion during the course of
pregnancy. The small subset of fetuses with hydrops is associated
with high intrauterine and neonatal mortality.
• A substantial number of echogenic lung lesions are hybrid lesions with
concomitance of BPS and CPAM and 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 are not associated with
microcystic CPAM. This distinction is of utmost importance when
prenatal intervention is considered.

13. Bronchopulmonary sequestration with massive pleural effusion:
pleuroamniotic shunting vs intrafetal vascular laser ablation
Mallmann et al., UOG 2014
Discussion
• In the absence of severe pleural effusion and mediastinal shift, BPS
has a high likelihood of spontaneous regression and therefore has a
favorable prognosis, which justifies expectant management
• In cases of BPS with hydrops, which are associated with massive
pleural effusion, the target of intrauterine therapy is either the
abnormal systemic feeding vessel or the pleural effusion
• Established literature for pleuroamniotic shunting, which often results
in resolution of hydrops, recognises the need for repeat shunt
insertions, due to shunt displacement and recurrent amnioreductions
• Ultrasound-guided intrafetal laser ablation of the abnormal systemic
blood supply of BPS might be more effective than shunting as it
targets the echogenic lung lesion rather than its symptoms

14. Bronchopulmonary sequestration with massive pleural effusion:
pleuroamniotic shunting vs intrafetal vascular laser ablation
Mallmann et al., UOG 2014
Limitations
• 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 a higher proportion of hydrops in the shunt group than the
laser group; this difference in morbidity between the treatment groups
adds a further bias to the results and consequently the conclusion
about the preferable form of treatment must be considered carefully

15. Bronchopulmonary sequestration with massive pleural effusion:
pleuroamniotic shunting vs intrafetal vascular laser ablation
Mallmann et al., UOG 2014
Conclusions
• In the absence of pleural effusion, the likelihood of spontaneous BPS
regression is high and the prognosis is favorable
• In cases with massive pleural effusion, ‘vascular’ laser ablation of the
feeding vessel appears to be more effective than pleuroamniotic
shunting, with fewer complications
• Laser treatment might also reduce the need for postnatal surgery
• These results should be confirmed by future studies with larger samples
and a prospective design

16. Bronchopulmonary sequestration with massive pleural effusion:
pleuroamniotic shunting vs intrafetal vascular laser ablation
Mallmann et al., UOG 2014
Discussion points
• Should ‘vascular’ laser ablation of the feeding vessel be the first line management of
BPS with massive pleural effusion, with or without hydrops?
• Over half of BPS cases without pleural effusion required postnatal surgery, whilst 20% of
BPS cases treated with intrauterine laser therapy required postnatal surgery. Is there a
role of laser ablation of the feeding vessel in uncomplicated BPS cases?
• If yes, is it for all cases or for cases that remain unchanged during the course of
pregnancy?
• Can we predict cases that are unlikely to resolve?