Original Paper
Fetal Diagn Ther
DOI: 10.1159/000485035
Systemic Venous Drainage Is Associated
with an Unfavorable Prenatal Behavior in
Fetal Bronchopulmonary Sequestration
Mei-Fang Lin a Hong-Ning Xie a Xiu-Hua Zhao a Ruan Peng a Ju Zheng a
Zhen-Peng Peng b  
a Department of Ultrasonic Medicine and Fetal Medical Center, The First Affiliated Hospital of Sun Yat-sen
University, Guangzhou, China; b Department of Radiology, The First Affiliated Hospital of Sun Yat-sen University,
Guangzhou, China
Keywords
Bronchopulmonary sequestration · Fetus · Venous
drainage · Doppler ultrasound · Prenatal history
Abstract
Objective: This study aimed to determine the significance of
the venous drainage pattern of bronchopulmonary seques-
tration (BPS) in the prenatal course. Methods: The venous
drainage pattern of fetuses with BPS was determined with
high-definition flow and confirmed by postnatal three-di-
mensional computed tomography angiography scan or au-
topsy. The volume of BPS lesions during gestation was re-
corded by the three-dimensional ultrasonographic Virtual
Organ Computer-Aided Analysis software. The relationship
between venous drainage pattern and prenatal characteris-
tics was determined. Results: Seventy-one fetuses were en-
rolled: 35 with systemic venous drainage (SVD) and 36 with
pulmonary venous drainage (PVD). The volumes of BPS le-
sions significantly increased from the middle second trimes-
ter to the later second trimester in the SVD group. A marked
decrease from the later second trimester to the third trimes-
ter was observed in the PVD group. The incidences of associ-
© 2017 S. Karger AG, Basel
E-Mail karger@karger.com
www.karger.com/fdt
Received: June 1, 2017
Accepted after revision: November 6, 2017
Published online: December 21, 2017
ated anomalies, hydrops, and polyhydramnios in the SVD
group were 14.2, 23.3, and 33.3%, respectively, significantly
higher than those in the PVD group (0, 0, and 5.6%, respec-
tively). Conclusions: Our data indicate that SVD is correlated
with a higher risk of associated anomalies and an unfavor-
able prenatal course in fetal BPS. Identification of the venous
drainage pattern is of clinical significance in predicting the
prenatal behavior of fetal BPS. © 2017 S. Karger AG, Basel
Introduction
Bronchopulmonary sequestration (BPS) is a congeni-
tal malformation of the respiratory tract consisting of
nonfunctional bronchopulmonary tissue that is separate
from the normal tracheobronchial tree and fed by an ab-
errant systemic artery. BPS was first described by Huber
in 1777 as an accessory pulmonary lobe; it was not until
1946 that Pryce introduced the term sequestration, which
comprises 0.15–6.45% of all pulmonary malformations
[1, 2]. The natural prenatal history of BPS is variable: the
lesions can regress, stay the same size, or grow during the
148.61.13.133 - 12/25/2017 6:38:12 AM
Hong-Ning Xie
Grand Valley State University
Department of Ultrasonic Medicine and Fetal Medical Center
The First Affiliated Hospital of Sun Yat-sen University
Guangzhou, Guangdong (China)
Downloaded by:
E-Mail hongning_x @ 126.com
 a b c

Color version available online

Fig. 1. a HDF sonogram showing an AA originating from the DAO supplying a BPS lesion in a fetus at 21 weeks.
b HDF sonogram showing PVD of the BPS lesion. c Doppler spectrum demonstrating the typical notching char-
acteristic of PVD. AA, anomalous artery; BPS, bronchopulmonary sequestration; DAO, descending aorta; HDF,
high-definition flow; PVD, pulmonary venous drainage.

Color version available online

Fig. 2. Pathologic specimen confirming the vein of the BPS lesion in the fetus in Figure 1 returning to the left
pulmonary vein (left panel) and the fetus in Figure 3 draining into the inferior vena cava (right panel). AA, anom-
alous artery; BPS, bronchopulmonary sequestration; DAO, descending aorta; LP, left pulmonary; LPV, left pul-
monary vein; RA, right atrium; RP, right pulmonary; V, vein.

prenatal course. Although the prognosis of the majority
of cases is favorable, there are still some cases that develop
hydrops and pulmonary hypoplasia, which can result in
high intrauterine and neonatal mortality [3–6]. However,
few factors have so far been found to correlate with dif-
ferent prenatal course and outcomes [7–9]. Kitano et al.
[7] were the first to apply assessment of the venous blood
flow in BPS to predict clinical outcomes. They found that
a decrease in blood flow in the drainage vein was associ-
ated with hydrops in extralobar pulmonary sequestration
(EPS), and therefore highlighted the importance of ve-
nous drainage assessment in the evaluation of fetuses
with BPS.
High-definition flow (HDF), with high capacity to detect
slow blood flow and depict smaller vessels compared to tra-
ditional color Doppler ultrasound, is used to visualize the
feeding arterial blood and to analyze the venous drainage of
the lesions [10]. For example, HDF was applied for prelim-
inary detection of the venous drainage of BPS in our previ-
ous study [3]. Two different venous drainage patterns were
identified: systemic venous drainage (SVD) and pulmonary
venous drainage (PVD). To date, no literature has reported
the natural course of BPS with different venous drainage
patterns. It was of our interest to study whether fetal BPSs
with different venous drainage patterns are accompanied
by different prenatal courses and perinatal outcomes.
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2 Fetal Diagn Ther Lin/Xie/Zhao/Peng/Zheng/Peng

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DOI: 10.1159/000485035
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Fig. 3. a HDF sonogram showing SVD into
the inferior vena cava. b Doppler spectrum
demonstrating a continuous phasic char-
acteristic of SVD. AADS, anomalous ar-
tery Doppler spectrum; HDF, high-defini-
tion flow; SVD, systemic venous drainage;
SVDS, systemic venous Doppler spectrum.
In this study, the venous drainage patterns of fetal BPS
were determined by HDF Doppler ultrasound. The rela-
tionships between the venous drainage patterns and as-
sociated anomalies, prenatal complications, the growth of
the lesions, and outcomes were examined.
Patients and Methods
Patients
This study was conducted on an unselected obstetric popula-
tion at a tertiary referral center from January 2008 to July 2015. All
suspected cases found during routine ultrasound scan or referred
from their local hospitals were referred to the observer (H.-N. Xie),
an expert in prenatal ultrasound diagnosis. The observer made the
final diagnosis and ascertained the venous drainage pattern after
re-examining the fetus. All patients underwent follow-up for out-
come data; the diagnosis and the venous drainage pattern were
confirmed by postnatal three-dimensional computed tomography
angiography (3DCTA) scan for the live-born cases or by autopsy
when intrauterine death and termination of pregnancy occurred.
Diagnostic Criteria and Identification of Venous Drainage
A Voluson 730 Expert and a Voluson E6 ultrasound machine
equipped with a 4- to 8-MHz annular array transducer (GE Health-
care, Kretztechnik, Zipf, Austria) were used to detected BPS le-
sions. The BPS diagnosis was made based on the presence of an
echogenic mass in the fetal thorax or the abdomen that received an
aberrant systemic arterial supply identified by HDF Doppler imag-
ing (Fig. 1a). The HDF was also applied to document the venous
flow of the BPS lesions. When the aberrant artery was identified as
it appeared from the aorta to the BPS, the aberrant vein was then
identified next to the aberrant artery with a reverse direction of
flow. The vein was then traced to the drainage point as possible as
it can delineate the whole course. Meanwhile, flow velocity wave-
forms of the vein were obtained. PVD was identified when the
typical notching characteristic of the pulmonary venous flow pat-
Venous Drainage Pattern Predicts Clinical
Behavior of Fetal BPS
a b
Color version available online
tern (a peak of forward flow during ventricular systole followed by
continuous flow until a second peak in early diastole) was present
(Fig. 1, 2). SVD was defined if the spectrum showed a continuous
phasic flow (Fig. 2, 3) [11]. To identify low-velocity vein and avoid
movement artifacts, a medium wall filter and a 50% gain were set,
and the pulse repetition frequency was adjusted to 0.6 KHz. To
record the spectrum, the sample volume size was adapted to the
vessel diameter, and the insonation angle was limited to 30°.
Measurement of BPS Volume
Prenatal 3D ultrasound was performed in all cases. The 3D ul-
trasonographic Virtual Organ Computer-Aided Analysis (VO-
CAL) software was used to assess the growth of the lung masses
over gestational age, according to the method used in our previous
study [3] (Fig. 4). The observer (H.-N. Xie) performed the volume
acquisitions and measurements to eliminate the interobserver
variability.
Determination of Prenatal Course and Perinatal Outcomes
All patients were recommended to undergo serial follow-up
ultrasound examinations in three gestational stages. Gestation of
19–24 weeks was defined as middle second trimester, 24+1–30
weeks as later second trimester, and 30+1–39 weeks as third trimes-
ter. In each case, the laterality and size of the BPS lesion, the origin
of the feeding vessel, the associated malformations, the presence of
nonimmune hydrops and polyhydramnios, intrauterine evolu-
tion, and neonatal outcome were recorded. Nonimmune hydrops
was defined as the presence of one or more of the following sono-
graphic features: ascites, pleural effusion, pericardial effusion, skin
or scalp edema, and placentomegaly [12].
Statistical Analysis
The relationship of coexistent anomalies, complications, and
outcomes between the two groups was determined by χ2 or Fisher
exact test. Variations in volumes between gestational stages were
estimated with the Wilcoxon rank test. Statistical analysis was per-
formed with SPSS version 16.0 (SPSS Inc., Chicago, IL, USA). The
level of statistical significance was set at p < 0.05.
148.61.13.133 - 12/25/2017 6:38:12 AM
Fetal Diagn Ther 3
Grand Valley State University
DOI: 10.1159/000485035
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Fig. 4. Three-dimensional image illustrating the volume measurement of bronchopulmonary sequestration le-
sions of a fetus at 21 weeks using the Virtual Organ Computer-Aided Analysis (VOCAL) software.
Results
General Characteristics
Seventy-nine fetuses with BPS identified by antenatal
ultrasound examinations were recruited in this study.
Eight cases were excluded because of indetermination of
the pattern of venous drainage due to small lesion size
(<15 mm). The venous drainage patterns were success-
fully determined in 71 fetuses with BPS, including 35 cas-
es with SVD and 36 cases with PVD. The BPS diagnosis
and venous pattern of 54 cases were confirmed by post-
natal 3DCTA scan, 11 cases were confirmed by autopsy,
and 6 cases were confirmed with postnatal 3DCTA to re-
gress in the uterus, the venous drainage patterns were de-
termined according to the results of ultrasound scan. The
median age of the gravidas of these 71 cases was 30 years
(range 21–38). The median gestational age at the diagno-
sis of fetal BPS was 24 weeks (range 18–32). A male pre-
dominance was observed in both groups, with an approx-
imately 2:1 overall male-to-female ratio. Most BPS lesions
in the SVD group were located within the thorax, except
for 5 cases lying in the abdomen, whereas all BPS lesions
in the PVD group were located within the thorax. There
4 Fetal Diagn Ther
DOI: 10.1159/000485035
Color version available online
was no difference in the congenital cystic adenomatoid
malformation volume ratio (CVR) at diagnosis between
the PVD (0.68 ± 0.5, range 0.03–2.57) and the SVD group
(0.56 ± 0.5, range 0.05–1.69) (p > 0.05). All lesions in the
PVD group were draining into the pulmonary vein,
whereas the venous drainage approach in the SVD group
included the inferior vena cava (n = 26), the azygos vein
(n = 5), the intercostal vein (n = 1), the hemiazygos vein
(n = 1), the splenic vein (n = 1), and the subdiaphragmat-
ic vein (n = 1) (Table 1). Unlike the recognitions of the
venous drainage patterns, the definite drainage approach-
es were not all correctly identified by ultrasound, includ-
ing 3 cases draining into the inferior vena cava located in
the abdomen where we fail to name the definite drainage
veins by ultrasound, whereas 2 cases with azygos vein
drainage and 1 case with intercostal vein drainage were
misdiagnosed to drain into the inferior vena cava by ul-
trasound.
The Volume of Fetal BPS with SVD Increases during
Pregnancy Course
Seventeen cases in the SVD group received two or
three ultrasound examinations during the three gesta-
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Lin/Xie/Zhao/Peng/Zheng/Peng
Grand Valley State University
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Table 1. Clinical characteristics of fetuses with BPS in the PVD and Table 2. Comparison of the outcomes of fetal BPSs in the PVD and
SVD groups SVD groups

Variable PVD group SVD group Outcome PVD group SVD group p value
(n = 36) (n = 35) (n = 36) (n = 35)

Sex Coexistent anomalies 0 (0%) 5 (14.2%) 0.025

Male 24 (67.0%) 22 (62.0%) Hydrops 0 (0%) 7 (23.0%)a 0.003
Female 12 (33.0%) 13 (38.0%) Polyhydramnios 2 (5.6%) 10 (33.3%)a 0.004
Location Live-born 33 (91.6%) 27 (90%)a 0.319
Left thorax 24 (67.0%) 29 (82.9%) Cesarean delivery rate 11 (33.3%)b 9 (33.3%)b 0.21
Right thorax 12 (33.0%) 1 (2.9%) Respiratory disorders 4 (12.1%)b 7 (25.9%)b 0.20
Subdiaphragmatic 0 (0%) 5 (14.2%) Postnatal surgery 6 (18.1%)b 4 (14.8%)b 0.47
Origin of the feeding vessel
Thoracic and abdominal DAO 34 (94.4%) 33 (94.2%) Data are shown as n (%). BPS, bronchopulmonary sequestra-
Splenic artery 0 (0%) 1 (2.9%) tion; PVD, pulmonary venous drainage; SVD, systemic venous
Intercostal artery 1 (2.8%) 0 (0%) drainage. a Thirty isolated cases were included (5 cases with associ-
Subdiaphragmatic artery 1 (2.8%) 1 (2.9%) ated structural anomalies were excluded). b  Only live-born cases
Venous drainage were included.
Pulmonary vein 36 (100%) 0 (0%)
Inferior vena cava 0 (0%) 26 (74.3%)
Azygos vein 0 (0%) 5 (14.1%)
Intercostal vein 0 (0%) 1 (2.9%)
Hemiazygos vein 0 (0%) 1 (2.9%) terminated without additional follow-up and therefore
Splenic vein 0 (0%) 1 (2.9%) excluded from further analyses. Among the remaining 30
Subdiaphragmatic vein 0 (0%) 1 (2.9%) fetuses in the SVD group, 7 cases developed hydrops:
Mean CVR 0.68±0.5 0.56±0.5 pleural effusion (n = 5), pericardial effusion (n = 1), and
Data are shown as n (%) or mean ± standard deviation. BPS, skin edema and pleural effusion (n = 1), whereas the cor-
bronchopulmonary sequestration; CVR, congenital cystic adeno- responding number in the PVD group was 0 (23.3 vs. 0%,
matoid malformation volume ratio; DAO, descending aorta; PVD, p = 0.003). Furthermore, 33.3% (10/30) of cases in the
pulmonary venous drainage; SVD, systemic venous drainage. SVD group presented associated polyhydramnios, which
was significantly higher than in the PVD group (33.3 vs.
5.6%, p = 0.004). No significant difference was found in
the live birth rate between the two groups (10.0 vs. 8.3%,
tional stages. Statistically, the volumes of fetal BPSs in the p > 0.05).
SVD group significantly increased from the middle sec- The median gestational age at birth was 38.9 weeks
ond trimester to the later second trimester (p < 0.01), but (range 38.0–40.0) in the SVD group and 39.0 weeks (range
remained unchanged to the third trimester (p = 0.158) 34.0–40.0) in the PVD group (p = 0.86). Cesarean delivery
(Fig. 5a). In the PVD group, 23 cases received two or three was executed in 33.3% of cases in both the SVD group
ultrasound examinations during the three gestational (9/27) and the PVD group (11/33) (p = 0.21). The mean
stages. The volumes of the BPSs remained constant from postnatal follow-up was 6 years (range 1–9). Postnatal
the middle second trimester to the later second trimester surgery was given to 6 cases in the SVD group and 4 cas-
(p = 0.441), followed by a marked decrease from the later es in the PVD group (p > 0.05). The rate of postnatal re-
second trimester to the third trimester (p < 0.01) (Fig. 5b). spiratory disorders, manifested as cough, fever, or neona-
tal respiratory distress, did not significantly differ be-
Fetal BPS with SVD Associated with Other Congenital tween the two groups (Table 2).
Anomalies and Complications
As indicated in Table 2, 14.2% (5/35) of cases in the
SVD group coexisted with anomalies, including dia- Discussion
phragmatic hernia (n = 2), pulmonary artery valve hypo-
plasia (n = 1), Ebstein anomaly (n = 1), and skeletal dys- BPS is a congenital pulmonary disease with variable
plasia (n = 1); meanwhile, no cases in the PVD group were prenatal course and prognosis. Ultrasound is the primary
associated with anomalies (14.2 vs. 0%, p = 0.025). The 5 diagnostic modality for fetal BPS. To date, the relation-
fetuses with associated anomalies in the SVD group were ship between the pattern of venous drainage and the clin-
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Venous Drainage Pattern Predicts Clinical Fetal Diagn Ther 5

Grand Valley State University

Behavior of Fetal BPS DOI: 10.1159/000485035

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 Color version available online
50 ns
20
p < 0.01
40

Volume of BPS, cm3

Volume of BPS, cm3

15
30

20 10

10 5

0
20 22 24 26 28 30 32 34 36 38 0
MS LS T
a Gestational age, weeks

40 ns
20 p < 0.01
30
Volume of BPS, cm3

Volume of BPS, cm3

15
20
Fig. 5. a Volume changes in the BPS lesions 10
of 17 cases with systemic venous drainage. 10
b Volume changes in the BPS lesions of 5
23 cases with pulmonary venous drainage.
0
Data are mean ± SEM. BPS, bronchopul- 19 21 23 25 27 29 31 33 35 37
monary sequestration; ns, not significant; 0
b Gestational age, weeks MS LS T
MS, middle second trimester; LS, later sec-
ond trimester; T, third trimester.

ical outcomes of fetal BPS has not been determined. Our
data showed that fetal BPS with SVD was more likely to
be associated with coexistent anomalies, hydrops, and
polyhydramnios compared to fetal BPS with PVD. The
volumes of fetal BPSs with SVD increased gradually dur-
ing the pregnancy course. To the best of our knowledge,
this study is the first one to have identified SVD as a
promising factor for prenatal behavior of fetal BPS.
Our results demonstrated that the incidence of coex-
isting anomalies in BPS fetuses with SVD was much high-
er than that in BPS fetuses with PVD. This is a reminis-
cence of the difference between EPS and intralobar pul-
monary sequestration (IPS) [13–16]. The spectrum of
coexisting anomalies detected in BPS with SVD also re-
sembled EPS, including congenital diaphragmatic hernia,
congenital heart defects, and vertebral anomalies [13, 14].
Based on these results and the fact that more than 95% of
IPSs drain directly into the pulmonary veins [16–18], we
speculated that our observations were mainly attributed
to the difference between EPS and IPS. However, only 10
cases received surgery to confirm the subtype of BPS le-
sions, thus more investigations are required to draw a
conclusion.
BPS diagnosis in the uterus has a variable natural pre-
natal history that may result in poor outcomes. Expectant
management should be given to BPSs without severe
pleural effusion, whereas fetal intervention is warranted
for BPSs with massive pleural effusion. Therapeutic op-
tions include alcohol ablation, thoracocentesis, pleu-
roamniotic shunting, and percutaneous laser ablation.
Percutaneous laser ablation of the feeding vessel is re-
garded to be most effective with few complications [4].
The prognosis of the BPSs with hydrops, if untreated, is
poor and associated with a high rate of perinatal mortal-
ity and severe respiratory insufficiency in the newborn [4,
6, 8, 19]. In our study, 7 cases with hydrops were diag-
nosed. Among them, 3 cases were terminated and 4 were
term births accompanied by eventful hospital stay.
To date, the underlying mechanism of hydrops re-
mains unclear [19]. Zhang et al. [9] found that BPS fe-
tuses with a CVR >1.6 were prone to have fetal hydrops.
Kitano et al. [7, 8] found that obstruction of the drainage
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6 Fetal Diagn Ther Lin/Xie/Zhao/Peng/Zheng/Peng

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DOI: 10.1159/000485035
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vein, manifested as a decrease in blood flow assessed by
Doppler ultrasound, was associated with massive pleural
effusion, though a large sample size is required to confirm
their findings. Our study, which recruited a larger FBS
cohort, showed that BPSs with SVD were more frequent-
ly complicated by hydrops and polyhydramnios than
those with PVD, although the CVRs of the two groups did
not differ from each other. The obstruction of the vena
cava compressed by the BPS lesions, which was regarded
as an important underlying mechanism of hydrops, will
obstruct the SVD but not the PVD of lesions. Also, the
fetal circulation is different from the neonatal circulation.
This could be partly due to the low systemic pressure in
the systemic veins, leading to an increased flow volume in
the lesions. Nevertheless, the identification of the drain-
age patterns is of clinical implication in predicting hy-
drops.
In the present study, the volumes of BPS lesions in the
SVD group significantly increased from the middle sec-
ond trimester to the later second trimester and remained
to the third trimester. A marked decrease from the later
second trimester to the third trimester was observed in
the PVD group. The notion that lung lesions regress as
they outgrow their vascular supply and partially involute
is postulated as the mechanism of regression [20]. Yet, the
mechanism by which the different growth patterns exist
between different venous drainages remains unclear. The
shapes of the BPSs are irregular and differ among various
types, i.e., the IPS shapes are spherical, whereas the EPS
shapes are triangular [13, 15]. Therefore, it is inappropri-
ate to use the same spherical formula to assess the BPS
sizes of all types. In our present study, we measured BPS
volumes by using 3D ultrasound with VOCAL, as our
previous study [3], which avoids assumptions about le-
sion shapes and assures the accuracy of volume measure-
ments [21]. Collectively, our study provides a new strat-
egy to predict the growth of BPS and the associated hy-
drops in order to offer suggestions regarding the clinical
management.
Our study is limited by a number of factors. First, the
high rate of pregnancy termination due to an insufficient
understanding of the natural history of BPS as well the
one-child family planning policy might have influenced
the comparison of perinatal outcomes between the two
groups. Second, some of the fetuses experienced scans
less than three times during gestation, which might have
affected the assessment of the volumes growth patterns.
Third, studies with a larger sample size are needed to val-
idate our data in the future.
In conclusion, two different venous drainage patterns
of BPS can be identified prenatally. BPS with SVD is cor-
related with a higher risk of associated anomalies, en-
largement of the lesions, and complicating hydrops and
polyhydramnios. Our study, therefore, suggests that
identification of the venous drainage pattern is of clinical
significance in predicting the prenatal course of fetal BPS.
Statement of Ethics
The study protocol was approved by the Institutional Review
Board of the First Affiliated Hospital of Sun Yat-sen University.
Disclosure Statement
The authors declare no conflicts of interest.
Funding Sources
This study was supported by the National Scientific Foundation
Committee of China (81571687, 81501491), the Guangdong Pro-
vincial Department of Science and Technology (2017A020214013),
and the Guangdong Provincial Medical Research Foundation
(A2016006).

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Lin/Xie/Zhao/Peng/Zheng/Peng
Grand Valley State University
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