European Journal of Cardio-Thoracic Surgery 54 (2018) 246–251 ORIGINAL ARTICLE

doi:10.1093/ejcts/ezy049 Advance Access publication 28 February 2018

Cite this article as: Khen-Dunlop N, Farmakis K, Berteloot L, Gobbo F, Stirnemann J, De Blic J et al. Bronchopulmonary sequestrations in a paediatric centre:
ongoing practices and debated management. Eur J Cardiothorac Surg 2018;54:246–51.

Bronchopulmonary sequestrations in a paediatric centre:

ongoing practices and debated management
Naziha Khen-Dunlopa,b,c,*, Konstantinos Farmakisa, Laureline Bertelootd, Francesca Gobboa,
Julien Stirnemannb,c,e, Jacques De Blicb,f, Francis Brunelleb,d, Christophe Delacourtb,f and Yann Revillona,b
a
Deparmtent of Pediatric Surgery, AP-HP, Hopital Necker-Enfants malades, Paris, France

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b
Université Paris Descartes, Sorbonne Paris Cite, Paris, France
c
EA FETUS, Paris, France
d
Deparmtent of Pediatric Radilogy, AP-HP, Hopital Necker-Enfants malades, Paris, France
e
Department of Obstetrics, AP-HP, Hopital Necker-Enfants malades, Paris, France
f
Department of Pediatric Pulmunology, AP-HP, Hopital Necker-Enfants malades, Paris, France

* Corresponding author. Deparmtent of Pediatric Surgery, Hôpital Necker-Enfants malades, 149 rue de Sevres, 75015 Paris, France. Tel: +33-1-44494198; fax: +33-1-
44494200; e-mail: naziha.khen-dunlop@nck.aphp.fr (N. Khen-Dunlop).

Received 30 August 2017; received in revised form 3 January 2018; accepted 14 January 2018

Abstract
OBJECTIVES: Bronchopulmonary sequestration (BPS) is the second most common congenital lung malformation, with an estimated inci-
dence ranging from 0.15% to 1.8%. Surgical treatment is elective in patients with symptoms, but the management of asymptomatic
patients remains controversial.
METHODS: We retrospectively reviewed the medical records of 99 patients treated for BPS in our institution from January 2000 to
December 2015. BPS was diagnosed prenatally in 86 (87%) cases. Management throughout this 16-year period was based on 3 interven-
tions: resection by open surgery, resection by thoracoscopy and embolization.
RESULTS: Among the 86 patients with a prenatal diagnosis of BPS, 14% had symptoms at birth and 10% had delayed symptoms at a
median delay of 8 months (4.5–42 months). For the other 13 patients, symptoms occurred at a median age of 34 months (range 3–
96 months). Embolization of the feeding vessel was performed in 46 patients with 6 secondary surgical resections (13%). A total of 59
patients were operated on: 23 cases by open surgery and 36 cases by thoracoscopy. The mean hospitalization stay was significantly longer
for open surgery: 4.8 ± 1.3 days vs 4.1 ±1.5 days, respectively (P = 0.03). Differences in hospitalization stay were also found between asymp-
tomatic and symptomatic patients: 3.5 ± 1.2 vs 5.1 ±1.6 days, respectively (P = 0.002). Two of the operated patients died.
CONCLUSIONS: When surgery is chosen, thoracoscopy appears to be a valuable procedure. A better understanding of the natural history
of BPS is still needed to define the optimal management and the respective roles of surgery, embolization or non-interventional follow-up.
Keywords: Child • Congenital lung malformation • Prenatal • Video-assisted thoracic surgery

INTRODUCTION hydrothorax or hydrops are reported in cases of voluminous

Foetal lung malformations occur in about 1/10 000 pregnancies
[1, 2] Advances in antenatal imaging in the 2010s have changed
their diagnosis and management [3]. Bronchopulmonary seques-
tration (BPS) is the second most common congenital lung anom-
aly after cystic pulmonary airway malformations. BPS is
characterized by a non-functional lung area, without communi-
cation with the normal bronchial tree and vascularized by an
aberrant systemic arterial supply that most commonly arises
from the descending thoracic aorta, less frequently from the
upper abdominal aorta (or celiac and splenic branches) and in
rare cases from intercostal, subclavian or coronary arteries [4].
BPS is usually well tolerated by the foetus during pregnancy.
However, exceptional prenatal complications such as
compressive forms [3]. After birth, patients with BPS are known
to be at risk of infection, haemoptysis, haemorrhage and, in rare
cases, heart failure [5–7].
Although surgical resection is elective in the case of a sympto-
matic malformation, management of asymptomatic cases is still
controversial and ‘management based only on patients observa-
tion, without any invasive intervention’ can be proposed [8, 9].
This controversy is even more relevant for BPS than for malfor-
mations with large cysts. Because of the absence of natural con-
nections with the normal airways, the rate of infectious
complications is expected to be much lower for BPS than for
cystic malformations [10]. Also, there is currently no description
of an association between BPS and a lung tumour, in opposition
to cystic malformations. Embolization of the systemic vessel has

C The Author(s) 2018. Published by Oxford University Press on behalf of the European Association for Cardio-Thoracic Surgery. All rights reserved.
V
 N. Khen-Dunlop et al. / European Journal of Cardio-Thoracic Surgery
been proposed as an alternative to surgery because thrombosis
of the systemic vessel leads to the regression of the malformation
[11, 12]. This therapeutic modality has been used for about
15 years in our centre. The aim of our study was to assess the
outcome of a large cohort of children with BPS according to the
therapeutic modalities used: resection by open surgery, resection
by thoracoscopy and embolization.
METHODS
Data collection
The medical records of all (n = 99) consecutive patients treated
for BPS in our paediatric surgery department from 1 January
2000 to 31 December 2015 were reviewed retrospectively. We
defined pulmonary malformations with a systemic artery pedicle,
confirmed by angiography or during surgical exploration, as BPS,
whether cystic components were visualized or not. There were
61 (62%) boys and 38 (38%) girls. Information for the pre- and
post-natal periods and imaging data were retrieved from the
medical records.
In all cases, and before treatment procedures, thoracic com-
puted tomography (CT) scans with contrast injections were per-
formed. For all 65 patients with a prenatal diagnosis who
remained asymptomatic (76%), early thoracic CT scans were per-
formed after birth, according to our survey plan, to characterize
the lung malformation at a median age of 3.7 months (range 2.6–
7.4 months). In asymptomatic cases, CT scans were performed
after pulmonary complications occurred, at a median age of
36 months (range 6–96 months). All the malformations were
located in the inferior lobes and mainly on the left side (74 vs 25
cases). The BPS form was intralobar in 65 (65%) cases and extra-
lobar in 34 (35%) cases.
Procedures
In cases with a non-cystic associated lesion and a solitary arterial
branch, embolization was performed. The right femoral artery
was punctured with the patient under general anaesthesia. A 3 Fr
introducer allowed the use of a 3 Fr catheter to reach the BPS
pedicle. Embolization was performed with acrylic spheres (embo-
lospheres) with a diameter ranging from 300 to 500 microns.
A micro coil (0.18 inch) was placed in the feeding artery to com-
plete the occlusion.
When cystic components were observed or multiple arterial
vessels were found on CT scans, surgical resection was per-
formed. In 2000 and 2001, only thoracotomies were performed.
In 2002, the first thoracoscopic resections were conducted in our
institution. From 2002 to 2009, both procedures were used,
depending on preoperative imaging results and preoperative
conditions. Since 2010, however, all the interventions have been
performed by thoracoscopy, in which case systemic arteries are
controlled from the start of the intervention. Vessels are divided
1 by 1 in the case of multiple pedicles. Since 2012, the section is
R
made after the clip is positioned and secured by an EndoloopV
(Ethicon, Johnson & Johnson, New Brunswick, USA) node at the
aortic side. For parenchymal sections, thermal tissue fusion
(LigaSure; Valleylab, Boulder, CO, USA) is used, and no staples are
used. For thoracoscopic resections, a 5-mm optical port and two
247
or three 5-mm operative ports are used with an insufflation at a
maximum of 4–5 mmHg.
Statistical analysis
Statistical analyses were performed with Statview 5.0 software
(SAS Institute Inc., Cary, NC, USA). Qualitative variables were
expressed as percentages and quantitative variables, as medians
and extremes. Discontinuous data were compared with the v2
test and continuous data with the Mann–Whitney U-test. Tests
were considered to be significant for a P-value below 0.05.
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Ethical statement
THORACIC
This study was conducted in accordance with the amended. The
institutional review board approved the study (CENEM 2016/
NKD). Because it was a retrospective anonymous study based on
medical reports only, no written consent was required.
RESULTS
Diagnosis
BPS was diagnosed prenatally in 86 (87%) cases, at a median
term of 22 weeks of gestation (range 17–32 weeks). Prenatal
imaging follow-up was performed using ultrasound; in the 14
most recent cases, complementary prenatal magnetic resonance
images were also obtained (Fig. 1). In all cases, a hyperechogenic
lesion was described, with a systemic vessel identified in 51 (52%)
cases. Three (3.5%) cases were associated with foetal symptoms
(Table 1). In 5 (5%) cases, the malformation was said to have dis-
appeared on the last prenatal ultrasound, but for all the patients,
BPS was seen on post-natal CT scans.
Associated malformations were found in 5 (6%) cases: scimitar
syndromes in 3 cases, left congenital diaphragmatic hernia in
1 case and VACTERL syndrome in 1 case.
Among the 86 patients with a prenatal diagnosis of BPS,
12 (14%) patients had symptoms at birth: respiratory distress
requiring tracheal ventilation in 3 cases, dyspnoea with oxygen
therapy in 7 cases and pneumothorax treated by exsufflation in 2
cases.
The 3 patients with scimitar syndrome did not present with
respiratory symptoms at birth; nor did the patient with VACTERL
syndrome. The patient with a congenital diaphragmatic hernia
was immediately intubated before any respiratory impairment
could occur, according to the local neonatal procedure for con-
genital diaphragmatic hernia management.
Delayed respiratory symptoms were observed for 9 patients
with a prenatal diagnosis (10%) at a median age of 8.0 months
(range 4.5–42.0 months): bronchopulmonary infections in 5 cases
and recurrent bronchitis in 4 cases.
Among the 13 patients without prenatal detection of BPS, only
1 had respiratory signs at birth, leading to the neonatal diagnosis
of BPS associated with cysts. For the other 12 patients without a
prenatal diagnosis, symptoms occurred at a median age of
34 months (range 3–96 months): bronchopulmonary infections in
7 cases, lung abscesses in 2 cases and recurrent bronchitis in
3 cases. A diagnosis of BPS was made from thoracic CT scans
after pulmonary complications occurred at a median age of
36 months (range 6–96 months).
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Figure 1: Prenatal magnetic resonance images for 2 cases of BPS. The malformation appears as a hypersignal zone in the thoracic area. Asymptomatic case. (A) Axial
view. The heart is right-shifted but not compressed. The malformation is left-sided with homolateral and contralateral visible normal lungs. (B) Coronal view. The mal-
formation occupies two-thirds of the left chest. A systemic pedicle is visualized from the aorta to the BPS in hyposignal (white star). Homolateral normal lung is visible
at the apex. The left diaphragm is well placed. Symptomatic case. (C) Axial view. The heart is right-shifted, pushed against the chest wall and compressed. There is no
visible lung. (D) Coronal view. The malformation occupies the entire left chest with a transmediastinal hernia that compressed the heart. The left diaphragm is flat-
tened. Compression is associated with a large abdominal ascites. BPS: bronchopulmonary sequestration.

Table 1: Prenatal symptoms of bronchopulmonary sequestrations

Age at symptoms Prenatal symptoms Prenatal procedure Birth Post-natal evolution

Case 1 26 WG Mediastinal shift + ascites No 38 WG, 3150 g Acute respiratory distress/tracheal ventilation
Surgery Day 2
Case 2 24 WG Mediastinal shift + hydrothorax Pleuroamniotic shunt 40 WG, 4020 g Transient respiratory distress/oxygen therapy
Surgery Day 12
Case 3 28 WG Mediastinal shift + hydrothorax + Pleuroamniotic shunt 37 WG, 2860 g Asymptomatic
polyhydramnios
n = 83 22 weeks (17–32) No No 39 ± 1.5 WG, n = 10 (12%)
3220 ± 550 g. 8 with respiratory distress
2 with pneumothorax
1 neonatal surgery at Day 9

WG: weeks of gestation.

Treatments Because the timing of the CT-scan follow-up exami-

Embolization. In 46 patients, embolization of the feeding vessel was
proposed at a median age of 21.6 months (range 5.2–63.5 months)
(Fig 2). All patients were asymptomatic before the procedure.
No complications occurred during the endovascular procedure.
Twenty-five (54%) patients had a moderate fever for 2 days following
the embolization. The mean hospitalization stay was 1.7 (±0.6) days.
nations was not defined, control imaging was not proposed
systematically. In 17 cases, the CT scans were obtained at a
mean delay of 13.3 (±2.7) months after the embolization.
In 10 (59%) cases, there was no persistent vascularization
but in all cases a residual mass was still visible without
contrast enhancement. In 7 (41%) cases, the embolization was
incomplete.
 N. Khen-Dunlop et al. / European Journal of Cardio-Thoracic Surgery
Figure 2: Bronchopulmonary sequestration embolization procedure. (A) Axial computed tomography scan with injection. The malformation is a hyperdense area
within the normal lung. The feeding vessel is visible (black arrow). (B) A catheter is in the pedicle (black arrow). The malformation is visualized by the injection before
coil placement.
Figure 3: Flow chart. BPS: bronchopulmonary sequestration.
Among all the patients who were embolized, a surgical resection
was performed in 6 (13%) cases because of the secondary respiratory
infections: 3 by thoracotomy and 3 by thoracoscopy. Preoperative
CT scans showed a residual mass for the 6 patients who had persis-
tent vascularization (3 cases) and complete arterial occlusion (3 cases).
Flow chart gives the repartition of the patients (Fig. 3).
Surgical resection
In 59 patients, surgical resection of the BPS was performed. After
the feeding vessel was visualized and ligated, a wedge resection
was done, following the delimitation of the ischaemic paren-
chyma. Among patients with a post-natal diagnosis, 12/13 (92%)
were operated on whereas 47 (55%) of the 86 patients with a
prenatal diagnosis were operated on.
In 23 cases, open surgery was proposed at a median age of
10 months (range 5 days–12.6 years). In 36 cases, thoracoscopy
was performed at a median age of 17 months (range 8 months–
15.2 years). The mean operative time was 86 ±18.6 min for open
surgery and 103 ±38.8 min for thoracoscopy (P = 0.15).
In 57 cases, there were no perioperative complications (Fig. 4).
Two deaths occurred. The first was a 5-year-old patient.
Because of postoperative exteriorized haemorrhage caused by a
chest tube 2 h after the end of the operation, a revision thoracot-
omy was performed showing an aortic ulceration at the contact
of the clip used to close the systemic vessel. Despite the reinter-
vention, the boy died. Since this case, the systemic vessel is
closed by an Endoloop node at the contact with the aorta. The
second death occurred in the neonatal period in 1 of the
2 patients with symptoms at the prenatal diagnosis. It was
249
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THORACIC
attributed to uncontrolled refractory arterial pulmonary hyper-
tension despite early BPS resection on the 2nd day of life.
Follow-up
The global median follow-up was 26.5 months (range
13.6 months–16.3 years).
At the last review, 12 patients treated by embolization had
symptoms: the 6 cases who were finally operated on because of
secondary infection and 6 other cases who were followed for
asthma (n = 2), scoliosis (n = 2), pectus excavatum (n = 1) and
mental retardation on the TUB b2b gene mutation (n = 1).
Among the operated patients, 5 had symptoms: asthma [4],
including 2 patients who were also followed for scoliosis and dia-
betes [1]. None had complementary surgery.
DISCUSSION
This paediatric experience is one of the largest published cohorts
on BPS. Historical data show a high rate of congenital malforma-
tions associated with BPS. Half of the patients with extralobar
BPS seemed to have such malformations, especially congenital
diaphragmatic hernia but also cardiac abnormalities, pulmonary
hypoplasia or foregut duplication cysts, whereas associated mal-
formations were reported rarely in the intralobar forms [1, 13].
We did not find such differences in our population, which had a
low incidence of associated malformations. We observed 3 asso-
ciated malformations among the extralobar forms (2 scimitar
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Figure 4: Thoracoscopic view of BPS resection. (A) Sequestration appears as an independent dense area, without bronchial branching. The feeding pedicle is easily
visualized (white star). (B) Dissection of the pedicle shows 2 arteries. (C) After the ligation and the sectioning of the arteries, the last connection is an adhesion to the
diaphragm. (D) Mobilization of the sequestration shows associated cysts. BPS: bronchopulmonary sequestration.

syndromes and 1 congenital diaphragmatic hernia) and 2 associ-
ated malformations among the intralobar forms (1 scimitar and
1 VACTERL syndrome). Even though the rate of these malforma-
tions was much lower than the 50% previously reported, the pro-
portion of extralobar forms remained higher. One important
point is that lung malformations appeared to be a lesser cause
for concern for all of our 5 patients with associated anomalies
(scimitar syndrome, congenital diaphragmatic hernia and
VACTERL) because no BPS-related prenatal or post-natal symp-
toms were observed.
Because of documented descriptions of spontaneous regres-
sion of BPS, embolization of the systemic artery has been pro-
posed in the management of BPS and has been used by our
team since 1996 [10]. It was also proposed for adults before sur-
gical correction of the sequestration to limit the risk of periopera-
tive bleeding or as a definitive treatment [14, 15]. Embolization
seems thus to be an attractive procedure, less invasive than sur-
gery and with few complications [16, 17]. However, the cases
have to be carefully selected. Histological analyses of all resected
BPSs showed that in half of the cases the malformation was asso-
ciated with another lung malformation (cystic pulmonary airway
malformation, bronchogenic cyst or gut duplication). This com-
plex condition led to differences in the evaluation of the risks
associated with BPS, mainly the risks of developing infections or
cancer [18–20]. Thus, the possible risk of the tumoural evolution
of cystic malformations led us not to propose embolization for
hybrid forms. Secondly, in cases of multiple arterial vessels, the
control of the obstruction did not seem reliable enough: In
3 cases in the beginning of our experience, redo embolizations
were performed because of large or multiple arteries. Our rate of
embolization failure leading to surgery is low, but the data based
on the population with follow-up CT scans at 1 year after the
procedure showed that persistent vascularization was observed
in about half of our cases. In the literature, the reported failure
rates are 15–80% depending on the evaluation criteria: disap-
pearance or volume regression of the lesion in symptom-free
patients [16, 21]. In the absence of symptoms or cysts, emboliza-
tion appears as a possible option, but long-term results are lack-
ing to assess the exact conditions for the use of this procedure
[16, 21].
When consent is given for surgery for symptomatic BPS, resec-
tion of asymptomatic BPS is still debated, especially because of
the unclear evaluation of the benefit/risk balance. In our cohort,
2 deaths were reported. For the neonatal death, surgery was per-
formed in a life-threatening condition and was not sufficient to
reverse the consequences of secondary refractive pulmonary
hypertension. Cardiac failure is exceptional in BPS but represents
one of the most challenging situations [22]. In the second death,
an uncontrolled haemorrhage was secondary to the BPS resec-
tion itself. Because of the high-pressure flow in the systemic ves-
sels, vascular changes in the main artery have been reported,
leading to vascular wall fragility or in some cases to aneurysmal
dilatation [6, 23]. Such an alteration of the arterial wall occurred
in our case. After this accident, we changed our technical
approach: Systemic vessel ligation was secured by an Endoloop
knot at the point of contact with the aorta. We have not had any
bleeding complications since then.
We did not find significant differences in the operative times
between open surgery and thoracoscopic resections. However,
the postoperative hospital stay was significantly longer after open
surgery. Because ours is a retrospective study involving evolu-
tions in practice, the cause of this difference cannot be
 N. Khen-Dunlop et al. / European Journal of Cardio-Thoracic Surgery
ascertained, even if postoperative pain control may have an
influence. Another point is the difference in the age of surgery
between open and thoracoscopic resections (10 vs 17 months,
respectively). Because we prefer to perform sparing surgery as
frequently as possible, we slowly delay the age of surgery. After
the patient is 1-year old, because the chest has grown, the sup-
plementary space provides much better conditions. But such sur-
gery can be performed earlier, and thoracoscopic resections
appear as a valuable first approach in BPS resection, with no
more side effects than the open procedure [24, 25].
In our population, a quarter of the patients had symptoms
before surgery. No difference was found in the operative time
but length of stay after surgery was significantly longer for symp-
tomatic patients compared to those who remained asympto-
matic. Although the evaluation of secondary infection is variably
reported in BPS [19, 26], surgery for lung malformations after
lung infection is associated with higher perioperative complica-
tions, with longer operative times, increased blood loss or higher
rate of conversion when performed thoracoscopically [27].
Postoperative morbidity was also increased with longer lengths
of stay and more frequent postoperative complications, such as
pulmonary fistulas, secondary haemorrhage and reoperation [28].
CONCLUSION
Even if they are rare, prenatal complications of BPS have to be
considered as a predictor of post-natal respiratory distress and
early neonatal surgery. However, respiratory signs can also be
present at birth without prenatal signs, and prenatal counselling
should urge caution.
Post-natal management of asymptomatic BPS still has to be
discussed, taking into account the presence or absence of cystic
components. Thoracoscopy appears to be a valuable approach,
once surgical resection is decided on. However, the presence of a
systemic feeding artery increases the risk of perioperative hae-
morrhage and has to be taken into account. The exact conditions
of the use of embolization remain to be defined, between sur-
gery and conservative management.
Conflict of interest: none declared.
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