Journal of Pediatric Surgery 56 (2021) 995–999

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Journal of Pediatric Surgery

journal homepage: www.elsevier.com/locate/jpedsurg

Is the pleating technique superior to the invaginating technique for

plication of diaphragmatic eventration in infants?
Do Hyung Kim a, Si Chan Sung a, Hyungtae Kim a, Kwang Ho Choi a, Bong Soo Son a,
Jong Myung Park b, Sung Kwang Lee a,⁎
a
Department of Thoracic and Cardiovascular Surgery, Pusan National University Yangsan Hospital, Yangsan, Gyoungnam, Republic of Korea
b
Department of Thoracic and Cardiovascular Surgery, Busan Medical Center, Yeonje-gu, Busan, Republic of Korea

a r t i c l e i n f o a b s t r a c t

Article history: Backgrounds: The pleating technique is widely used in plication but is difficult to perform with thoracoscopy be-
Received 8 May 2020 cause of its complex procedure and the limited surgical space. Thus, the invaginating technique was introduced
Received in revised form 10 July 2020 to facilitate thoracoscopic surgery and is now widely used in video-assisted thoracoscopic surgery (VATS) plica-
Accepted 13 July 2020 tion. However, the usefulness of the invaginating technique in children has not been established because of the
lack of data on long-term outcomes after surgery using the technique.
Key words:
Methods: From March 2007 to December 2017, 21 patients who were surgically treated for congenital diaphrag-
Diaphragmatic eventration
Plication
matic eventration and phrenic nerve palsy after congenital cardiac surgery were divided into 2 groups according
Pleating to the surgical method used (pleating technique: 10 patients, invaginating technique: 11 patients). We evaluated
Invaginating the patients for postoperative outcomes and recurrence of diaphragmatic eventration over 5 years. Postoperative
Thoracoscopy recurrence of diaphragmatic eventration was confirmed by calculating the ratio of the eventration level between
the eventrated and normal diaphragms.
Results: In the 21 patients who underwent diaphragmatic plication, the pleating and invaginating techniques
were used in 10 and 11 patients, respectively. The mean follow-up duration was 63.4 ± 48.4 months (pleating
group [P] vs invaginating group [I]: 89.1 ± 52.4 vs 40.1 ± 30.8 months, p = 0.022). The mean eventration
rates in the 21 patients was 26.7% ± 9.1% (P vs I: 26.6% ± 6.1% vs 26.9% ± 11.3%, p = 0.945) before operation
and −2.1% ± 7.3% (−2.8% ± 7.5% vs −1.5% ± 7.4%, p = 0.695) in the immediate postoperative period. From
the first to the fifth postoperative year, no recurrence of diaphragmatic eventration was found in any of the
groups during the follow-up.
Conclusions: The invaginating technique was easier to perform but showed a similar long-term result as com-
pared with the pleating technique in terms of the growth and development of the chest cavity in the pediatric
patients in this study. Thus, we recommend that the invaginating technique be applied in VATS plication for chil-
dren as an alternative to the pleating technique.
Level of evidence: Level III.
© 2020 Elsevier Inc. All rights reserved.

The invaginating technique has been considered as the core tech-
nique of video-assisted thoracoscopic surgery (VATS)-plication since
it was first introduced as a diaphragmatic suture technique by
Mouroux et al. [1]. It involves pushing the center of the diaphragm
toward the abdomen to secure a sufficient surgical space and to
allow suture of both ends of the diaphragm fold. The invaginating
⁎ Corresponding author at: Department of Thoracic and Cardiovascular Surgery, Pusan
National University Yangsan Hospital, Pusan National University College of Medicine, 20
Geumo-ro, Mulgeum-eup, Yangsan-si, Gyeongsangnam-do 50612, Republic of Korea.
Tel.: +82 55 360 2127.
E-mail address: drlsk@naver.com (S.K. Lee).
technique is a simpler surgical procedure than the pleating tech-
nique, which is the most commonly used diaphragm suture tech-
nique of plication. Currently, the invaginating technique is used as
a standard procedure for thoracoscopic plication of adult diaphrag-
matic eventration, with some variations depending on the operator
[2,3]. However, whether the invaginating technique is as effective
as the conventional pleating technique for preventing recurrence of
diaphragmatic eventration has not been verified definitely, and
follow-up results have not been reported, especially in terms of the
growth and development of the chest cavity in infant patients. The
purpose of this study was to evaluate the usefulness of the invaginat-
ing technique in comparison with that of the pleating technique for
infant diaphragmatic eventration or elevation.

https://doi.org/10.1016/j.jpedsurg.2020.07.015
0022-3468/© 2020 Elsevier Inc. All rights reserved.
996 D.H. Kim et al. / Journal of Pediatric Surgery 56 (2021) 995–999

1. Materials and methods

1.1. Study design and patient selection

We retrospectively reviewed the clinical data of infant patients

diagnosed as having diaphragmatic eventration or paralysis within
12 months after birth and who underwent diaphragmatic plication in
our clinic between March 2007 and December 2017. The time of surgery
was determined according to the characteristics of the disease. If venti-
lator weaning was difficult because of the definite phrenic nerve injury
caused by the congenital cardiac or thoracic surgery, the plication sur-
gery was performed immediately. In the cases of congenital diaphrag-
matic eventration, the patients were followed up for N6 months
before operation was decided.
A total of 21 patients underwent diaphragmatic plication. The pa-
tients were divided into 2 groups according to the surgical method
used, either the pleating or invaginating technique. Of the patients, 10
underwent the pleating technique and 11 underwent the invaginating
technique. Patients who underwent the pleating technique had the
eventration after congenital cardiac surgery, and were managed by con-
genital cardiac surgeon who was unfamiliar about VATS. In other group
where congenital diaphragmatic eventration was found incidentally,
the invaginating technique was performed by a thoracic surgeon who
Fig. 1. Evaluation of the eventration level calculated on a chest radiograph. The eventration
was familiar about VATS. In previous study, the invaginating technique level is calculated as a percentage of the distance between the peaks of the
was considered to be a more feasible surgical method for VATS. We de- hemidiaphragm and apex in the bilateral thoraces. The calculation formula is as follows: 1
termined the effectiveness of the invaginating technique by comparing 00− 100⨯B
A , where A is the distance between the peaks of the normal hemidiaphragm and
the postoperative results, including ventilator care duration (days) after apex, and B is the distance between the peaks of the eventrated hemidiaphragm and apex.
plication, operation time, and postoperative and total hospital stay du-
rations, between the 2 groups, and followed up the patients annually
to investigate the ratio between the distance from the normal dia-
phragm to the apex and the distance from the eventrated diaphragm 1.3.2. Invaginating technique
to the apex. Data were collected from the patients' medical records. The invaginating technique was performed using thoracoscopy in
The institutional review board of Pusan National University Yangsan most cases, but was converted to open thoracotomy in 2 patients
Hospital approved this study and waived the need for informed consent. owing to failure of one-lung ventilation. A three-port thoracoscopic sur-
gery was performed via the sixth ICS for the camera port, and via the
1.2. Evaluation of eventration ninth and tenth ICSs for the endoscopic instrument. Surgery was per-
formed by suturing between the eventrating edges to cover and press
The eventration level was measured on chest radiographs in accor- the eventrated diaphragm (Fig. 2b). As in the pleating technique, 6 to
dance with the method reported by Ozkan et al. [4]. For the patients 7 stitches were applied to each nonabsorbable suture to plicate the dia-
who could and could not stand alone, chest radiography was performed phragm into a flat position.
in the posteroanterior and anteroposterior positions, respectively.
The hemidiaphragm level was measured on chest radiography. The 1.4. Statistical analyses
distance between the peaks of the hemidiaphragm and apex of the ipsi-
lateral thorax was measured using this new method. The left and right Continuous data are expressed as a mean value with its standard de-
distances between the peaks of the hemidiaphragm and apex were viation (SD). A data analysis was performed using the SPSS version 21.0
also measured. The eventration level was calculated as a percentage software (SPSS, Chicago, IL, USA). Comparisons between the groups
by using the B/A ratio, where A is the distance between the peaks of were performed using the Student t test; categorical variables were
the normal hemidiaphragm and apex, and B is the distance between analyzed using the Fisher exact or chi-square test. Differences with
the peaks of the eventrated hemidiaphragm and apex. The calculation P values of b 0.05 were considered statistically significant.
formula used was 100 − (100 × B/A). The patients were followedC
up before, after, 1 month after, and 1–5 years after operation, and 2. Results
underwent measurement for their bilateral diaphragm level. The post-
operative period was measured using the same formula and chest 2.1. Operative results
radiography (Fig. 1).
In the 21 patients who underwent diaphragmatic plication, the
1.3. Surgical technique pleating and invaginating techniques were used in 10 and 11 patients,
respectively. Of the patients, 16 were male and 5 were female. The
1.3.1. Pleating technique mean age of the 21 patients was 7.7 ± 6.1 months. Overall, 10 and 11
The pleating technique is the most widely used plication method. patients had right and left diaphragmatic eventrations, respectively.
Posterolateral thoracotomy was performed in all the cases. The thoracic The diaphragmatic eventration occurred after cardiac surgery in 10 pa-
cavity was entered through the seventh or eighth intercostal space tients in the pleating (P) group and 3 patients in the invaginating
(ICS), thereby revealing a diaphragmatic eventration. Six to 7 stitches (I) group, and after thoracic surgery, in 1 patient in the invaginating
were applied to each nonabsorbable suture, with pledgets to plicate group, but it was congenital in 7 patients in the invaginating group. In
the diaphragm into a flat position. The sutures were performed from the pleating group, diaphragmatic plication was performed using pos-
the posteromedial to the anterolateral direction to prevent phrenic terolateral thoracotomy. In the invaginating group, all plications were
nerve damage (Fig. 2a). started with a VATS. However, in 2 patients, VATS was converted to
 D.H. Kim et al. / Journal of Pediatric Surgery 56 (2021) 995–999 997

Fig. 2. Schematic diagram of the surgical procedure. The dotted line is the level of the preoperative diaphragm (open arrows). The red line is the level and shape of the postoperative
diaphragm (wide arrow). The black line represents the stitches of the plication (narrow arrow). a) Pleating technique: 6 to 7 fine stitches are applied to each 4-0 polypropylene
suture, forming an accordion-like fold with Teflon pledgets to the plication. b) Invaginating technique: the eventrating edges are sutured to cover and press the eventrated diaphragm
toward the abdominal cavity.

thoracotomy because of failure of one-lung ventilation and failure to
secure a sufficient operative space.
The mean ventilator care duration was 4.8 ± 10.5 days (P vs I: 5.6 ±
12.4 vs 4.0 ± 8.9 days, p = 0.736) before plication and 2.0 ± 3.7 days
(P vs I: 2.5 ± 3.6 vs 1.6 ± 4.0 days, p = 0.571) after plication. However,
the differences were not statistically significant. The mean operative
time was 118.8 ± 55.4 min (P vs I: 156.0 ± 51.7 vs 85.0 ± 33.2 min,
p = 0.001). The operative time of the invaginating technique was statis-
tically significantly shorter than that of the pleating technique. The
mean total and postoperative hospital stay durations were 34.5 ±
36.8 days (P vs I: 36.2 ± 30.8 vs 32.9 ± 43.1 days, p = 0.844)
and 20.4 ± 24.5 days (P vs I: 20.9 ± 20.3 vs 19.9 ± 28.8 days, p =
0.929; Table 1), respectively. The differences were not statistically signif-
icant. One patient in the pleating group died not from a cause related to
the plication but from a previous severe cardiac problem. In the invaginat-
ing group, if no other accompanying congenital malformations were
found, the patients were discharged within 1 week after surgery.
2.2. Follow-up of eventration rate
The mean follow-up duration was 63.4 ± 48.4 months (P vs I:
89.1 ± 52.4 vs 40.1 ± 30.8 months, p = 0.022). The mean eventration
rate of the 21 patients was 26.7% ± 9.1% (P vs I: 26.6% ± 6.1% vs
26.9% ± 11.3%, p = 0.945) before operation, − 2.1% ± 7.3% (P vs I:
−2.8% ± 7.5% vs −1.5% ± 7.4%, p = 0.695) in the immediate postoper-
ative period, 5.9% ± 8.0% (P vs I: 4.8% ± 9.4% vs 6.8% ± 7.0%, p = 0.622)
in the first postoperative month, and 3.4% ± 11.9% (P vs I: 2.4% ± 5.6%
vs 4.4% ± 16.4%, p = 0.734) in the 12th postoperative month. From the
first to the fifth postoperative year, the degree of diaphragm elevation
was maintained well. No recurrence of diaphragmatic eventration was
found in any of the groups during the follow-up (Fig. 3). Musculoskele-
tal abnormalities were investigated on chest radiography and were
found only in 2 patients in the pleating group, with no statistically sig-
nificant difference between the 2 groups (p = 0.114).
3. Discussion
Most patients with diaphragmatic eventration are asymptomatic or
complain of mild dyspnea during exercise. Worsening of these symp-
toms is caused by heart deviation and tamponade owing to decreased
lung volume and diaphragm elevation. Diaphragmatic movements di-
rected inferiorly and paradoxical movements can cause adverse effects
on respiratory function. Patients with symptomatic manifestations re-
quire surgical correction [5,6]. Children or infants with diaphragmatic
eventration experience severe respiratory distress or are difficult to
wean from ventilator support because of their immature lungs and re-
duced alveolar-to-lung mass ratio. Ventilation/perfusion mismatch in
a nonventilated lung causes chronic hypoxia and pulmonary hyperten-
sion. In severe cases, it causes delayed neurological development [7–9].
Therefore, a more active correction is required in pediatric patients than
in adult patients.
Diaphragmatic eventration can be successfully treated with dia-
phragmatic plication. The increased central part of the diaphragm is
folded into one or more layers and fixed in this position. Diaphragmatic
plication is intended to decrease lung compression and improve lung
volume, and strengthen the action of the intercostal and abdominal
muscles. It allows for a more effective diaphragmatic recruitment,
increased diaphragmatic strength, and the maximum voluntary ventila-
tion possible. While recovering its diaphragmatic function, the plicated
diaphragm maintains its growth proportionally on the other side.

Table 1
Characteristics of patients and perioperative results.

Total (21) Pleating group (10) Invaginating group (11) P value

Male:Female 16:5 8:2 8:3 0.696

Ages (months) 7.7 ± 6.1 5.8 ± 5.7 9.5 ± 6.1 0.175
Laterality (right:left) 10:11 3:7 7:4 0.123
Thoracotomy:thoracoscopy 12:9 10:0 2:9 b0.001
Ventilator care before plication (days) 4.8 ± 10.5 5.6 ± 12.4 4.0 ± 8.9 0.736
Ventilator care after plication (days) 2.0 ± 3.7 2.5 ± 3.6 1.6 ± 4.0 0.571
Operation time (min) 118.8 ± 55.4 156.0 ± 51.7 85.0 ± 33.2 0.001
Hospital stay (days) 34.5 ± 36.8 36.2 ± 30.8 32.9 ± 43.1 0.844
Postoperative stay (days) 20.4 ± 24.5 20.9 ± 20.3 19.9 ± 28.8 0.929
Musculoskeletal abnormality 2 (9.5%) 2 (20%) 0 0.119
Follow up duration (months) 63.4 ± 48.4 89.1 ± 52.4 40.1 ± 30.8 0.022

Values are presented as mean±standard deviation or n (%) unless otherwise indicated.

998 D.H. Kim et al. / Journal of Pediatric Surgery 56 (2021) 995–999

Fig. 3. Graph of the mean ± standard deviation of the percentage of the eventration level in all the groups. An approximately 26% difference can be observed between the level of the
bilateral diaphragms in each group before surgery, but no signficant difference can be found between the diaphragms after the surgery, indicating good results after the surgery, which
were maintained well over 5 years. The vertical axis is the ratio between the eventrated and normal diaphragms. The horizontal axis is the follow-up period (pleating technique vs
invaginating technique). The lower table presents the results of the long-term follow-up, and the values are means and standard deviations. No statistically significant diference can be
found for each period between the groups. POD: postoperative day.

Plication is necessary in pediatric patients to maintain and develop
proper lung function [10].
Traditionally, plication is performed using open surgery such as tho-
racotomy or laparotomy. However, thoracotomy divides the latissimus
dorsi or anterior serratus muscle, which may lead to scoliosis, winged
scapula, and musculoskeletal deformities or anomalies of the chest
wall in the long term. It may result in decreased lung function in the fu-
ture. As the trend of thoracic surgery is changing from open thoracot-
omy to thoracoscopic surgery, the number of reports of VATS plication
in children is increasing. VATS plication can inhibit the deterioration of
respiratory function caused by thoracotomy and respiratory muscle in-
juries. As thoracotomy itself may affect the growth of the thorax, a
thoracoscopic approach may be necessary if surgery is possible. How-
ever, it is not yet widely used as a standard technique. Pediatric VATS
plication is challenging to perform as compared with the conventional
open technique because of the difficulty of one-lung ventilation and se-
curing a sufficient surgical space. In addition, the long-term results of
the VATS technique have not been reported, so the appropriateness of
the application of VATS in pediatric patients must be verified [11].
Recently, the most commonly used suture techniques in plication
are the pleating and invaginating techniques. The pleating technique in-
volves several interrupted sutures on multiple folds in the center of the
diaphragmatic eventration, resulting in an accordion-like shape [12].
This technique is generally chosen for conventional open thoracotomy.
The incidence of recurrence is low because most of the diaphragm is
fixed with the thread. In the invaginating technique, which was first re-
ported by Mourux et al. [1], the eventrating edges are sutured to cover
and press the center of the eventrated diaphragm toward the abdominal
cavity. From the point of view of preventing eventration, pleating is a
more appropriate method of plication than invaginating. However, in
pleating, which involves repeated sutures according to several folds,
VATS is more difficult to implement than in invaginating. Especially in
infants, it is even more difficult because of the narrow surgical space.
Although CO2 insufflation can be used in VATS, it is difficult to apply in
conventional plication techniques (pleating). Therefore, the invaginat-
ing and continuous suture technique presented by Mouroux is the
most suitable for the VATS procedure in pediatric plication. This tech-
nique, however, approximates the two folds, which widen the dia-
phragm area where the diaphragm can stretch again. Therefore, the
possibility of reoperation may be higher if the VATS invaginating tech-
nique is applied in the pediatric group instead of the conventional
open technique. However, in this study, the long-term results of the in-
vaginating technique were not inferior to those of the pleating tech-
nique, which is a conventional open surgery that considers the equal
growth of both lungs. The invaginating technique does not leave many
redundant diaphragm areas and is associated with a low risk of
recurrence.
The long-term results of plication can be determined by diaphragm
reelevation or pulmonary function tests. Simon et al. [13] reported an
improvement in pulmonary function of approximately 25% in 15 pa-
tients who underwent the conventional open surgery and were
followed up for N7 years. However, 3 patients (20%) showed decreased
lung function as compared with that immediately after surgery. Mourux
et al. [14], who first introduced the invaginating technique, reported
that 6 of 12 surgical patients who underwent long-term follow-up for
N 5 years showed significant improvement in lung function. However,
when comparing pulmonary function results at 1 and 5 years, they
found that 2 of the 6 patients showed decreased lung function, which
indicates a decreased lung volume owing to diaphragm elevation in
some parts regardless of open surgery or VATS. In adults, long-term re-
sults can be compared using pulmonary function tests, but in the pedi-
atric group, reports of long-term results are rare because lung function
itself is difficult to assess.
In the pediatric group, pulmonary function continues to develop
owing to the increased lung volume as the lung grows. Long-term out-
comes are difficult to assess on the basis of pulmonary function changes
 D.H. Kim et al. / Journal of Pediatric Surgery 56 (2021) 995–999
because contralateral healthy lungs can compensate for the ipsilateral
lung volume reduction owing to diaphragmatic eventration. Therefore,
to confirm the long-term outcome of plication in the pediatric group,
it is reasonable to determine the size of the lung on one side of the pli-
cation as compared with the growth of the contralateral lung. The
change in diaphragm height on the plication side must be confirmed
on the basis of the diaphragm height on the healthy side [4]. We wanted
to analyze whether the diaphragmatic plication procedure had a posi-
tive effect on the equal growth of both lungs in terms of lung growth.
Therefore, we compared the level of diaphragm elevation of the ipsilat-
eral hemithorax with that of the contralateral hemithorax to determine
the effect of diaphragmatic plication.
None of the patients showed significant elevation after diaphrag-
matic plication as compared with the height of the contralateral dia-
phragm. No significant diaphragmatic elevation was found in the
patients who underwent long-term follow-up of N5 years. This
means that diaphragmatic plication can be effective even during
lung growth in children, and its effects last for a long time. Therefore,
in patients with confirmed diaphragmatic elevation, plication can
help equalize lung growth and is thus strongly recommended. The
results of the invaginating technique were equivalent to those of
the pleating technique. The long-term results were good, possibly
owing to the effects of thoracotomy and VATS, as well as the plication
suture technique.
4. Conclusion
Surgical treatment of diaphragmatic eventration is effective for a
balanced lung volume development in pediatric patients with con-
firmed irreversible diaphragmatic eventration. Contrary to the emerg-
ing concerns, the results of the invaginating technique, which is
mainly used for VATS, were equivalent to those of the pleating tech-
nique. Therefore, VATS with the invaginating technique is a good option
for pediatric patients with diaphragmatic eventration who require sur-
gical correction.
999
Acknowledgment
This work was supported by a 2-Year Research Grant of Pusan Na-
tional University Yangsan Hospital.
Declarations of interest
None.
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