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JOURNAL OF LAPAROENDOSCOPIC & ADVANCED SURGICAL TECHNIQUES

Volume 26, Number 11, 2016 2016 IPEG Papers


ª Mary Ann Liebert, Inc.
DOI: 10.1089/lap.2016.0247

Approach to Recurrent Congenital Diaphragmatic Hernia:


Results of an International Survey

Nicholas E. Bruns, MD,1 Ian C. Glenn, MD,1 Neil L. McNinch, MS, RN,2 Kelly Arps, BS,3
Todd A. Ponsky, MD,1 and Avraham Schlager, MD1

Abstract

Purpose: Although recurrence remains one of the most feared complications following congenital diaphrag-
matic hernia (CDH) repair, there are minimal data on the optimal surgical approach to these complex situations.
The purpose of this study was to survey the international pediatric surgery community to ascertain practice
patterns for both minimally invasive (MIS) and open approaches for recurrent CDH.
Materials and Methods: A survey was e-mailed to members of an online community of pediatric surgeons. The
questionnaire elicited surgeons’ clinical experience, the continent in which they practice, and their surgical
approach (laparotomy, thoracotomy, laparoscopy, or thoracoscopy) to five clinical cases, including initial and
recurrent Bochdalek hernias. Fisher’s exact test and chi-square test were used for statistical analysis.
Results: Two-hundred eighty pediatric surgeons responded to the survey. In total, 52.1% of surgeons chose an MIS
approach for an initial repair of left CDH with the younger surgeons more likely to use an MIS approach. For the
recurrence scenarios, 42.5%–55.5% of these surgeons would attempt an MIS repair after a recurrence. Specifically,
thoracoscopy was favored over laparoscopy following both prior laparotomy (30.0% versus 7.5%) and prior right
thoracoscopy (26.4% versus 10.0%), less favored following thoracotomy (9.3% versus 18.9%), and relatively similar
proportions following prior left thoracoscopy (17.5% versus 16.4%). Laparotomy was the preferred open approach
both for initial presentation and all recurrence scenarios. Among surgeons who would treat initial CDH with an open
procedure, between 10.4% and 17.9% would switch to an MIS approach, most commonly after prior failed laparotomy.
Conclusions: Approximately half surgeons who approach initial left CDH in an MIS manner would attempt an
MIS approach for recurrence. The tendency to approach CDH recurrence from the opposite body cavity as the
initial repair clearly impacted the surgical approach. This was particularly pronounced for MIS repairs, whereas
for open approach, laparotomy remained, by far, the most popular in all scenarios.

Keywords: congenital diaphragmatic hernia, congenital diaphragmatic repair; minimally invasive surgery,
thoracoscopy, laparoscopy

Introduction ternational pediatric surgery community to ascertain practice


patterns for MIS and open approaches for recurrent CDH.
R ecurrence remains one of the most feared compli-
cations after congenital diaphragmatic hernia (CDH)
repair. Despite this prevalent concern, there is a paucity of
Materials and Methods
data in the medical literature on the optimal approach to CDH A survey was e-mailed to members of an online commu-
recurrence. Minimally invasive (MIS) approaches to CDH nity of pediatric surgeons affiliated with GlobalCastMD
repair have been gaining popularity over the past 10 years (Shaker Heights, OH). The questionnaire was created using
with both thoracoscopic and laparoscopic approaches de- Google Forms (Mountain View, CA) and consisted of 10
scribed. Nevertheless, data on treatment for recurrences are questions. It began by asking surgeons to identify their
limited due to its low incidence,1 especially within a single clinical experience as a pediatric surgeon by selecting how
institution. The purpose of this study was to survey the in- many years they have been in practice (0–10, 11–20, or >20)

1
Division of Pediatric Surgery, Akron Children’s Hospital, Akron, Ohio.
2
Rebecca D. Considine Research Institute, Akron Children’s Hospital, Akron, Ohio.
3
Emory University School of Medicine, Atlanta, Georgia.

1
2 BRUNS ET AL.

Table 1. Preference for Treating Initial Left were diverse: 39.3% were in North America; 27.5% Europe;
Congenital Diaphragmatic Hernia 16.8% Asia; 7.1% South America; and 9.3% other.
by Clinical Experience (N = 280) Younger surgeons were significantly more likely to select
an MIS approach compared to surgeons who had been in
Years of experience n % MIS
practice for longer (Table 1; P < .01). There was no statisti-
0–10 80 66.3 cally significant association between location of practice and
11–20 83 56.6 preference for MIS (P = .25).
>20 117 39.3
Initial CDH repair
MIS, minimally invasive surgery.
52.1% of all responding surgeons chose an MIS approach
for the initial repair of a left Bochdalek CDH (Table 2).
or if they are not a pediatric surgeon (trainee or none of the Hereto forward, the 52.1% of surgeons that chose an MIS
above). Participants that identified themselves as trainees or approach will be referred to as ‘‘MIS surgeons’’ and the
not pediatric surgeons were excluded from the study. 47.9% surgeons that selected an open approach will be re-
Next, participants identified the continent in which they ferred to as ‘‘open surgeons,’’ reflecting their preferred
practice (Asia, Europe, North America, South America, treatment for routine initial CDH. Of the MIS surgeons,
or other). 88.4% chose thoracoscopy and 11.6% chose laparoscopy
Participants were then presented several clinical cases of (Table 3). Among the open surgeons, 97.8% chose laparo-
a hemodynamically stable infant with a CDH (both initial tomy and 2.2% chose thoracotomy (Table 4).
and recurrent). Respondents were prompted to select their
preferred approach (laparotomy, thoracotomy, laparoscopy, CDH recurrence
or thoracoscopy) for cases of both initial and recurrent
Bochdalek CDH. Refer to Appendix A for the complete MIS surgeons would attempt an MIS repair after a recurrence
survey. 42.5%–55.5% of the time (Table 5). Among this group, thor-
Fisher’s exact test and chi-square test were used for ex- acoscopy was the most common approach to both recurrent left
amination of associations among variables of interest, which CDH after laparotomy (47.8%) and right CDH after thoraco-
are outlined below. All testing were completed utilizing SAS scopy (36.3%), although laparotomy was almost as common for
v 9.4/13.2 software (Cary, NC) and evaluated at the type I right CDH recurrence (34.2%). However, even in this MIS
error rate of alpha = 0.05 level for statistical significance. group, laparotomy was the most selected approach for a re-
current left CDH after thoracoscopy and thoracotomy with
43.2% and 49.3%, respectively. Nevertheless, MIS surgeons
were overall more likely to select an MIS approach for all re-
Results
currence types compared to surgeons who prefer an open repair
The survey received 287 responses. Seven respondents did for initial CDH (P < .01 for all cases).
not identify themselves as pediatric surgeons and were excluded For all CDH recurrence scenarios, laparotomy was the most
from analysis. Of the remaining 280 pediatric surgeons, 28.6% popular choice among open surgeons, ranging between 59.7%
had been in practice for 0–10 years, 29.6% for 11–20 years, and and 79.1%. Open surgeons attempted MIS 10.4%–17.9% of
41.8% for 20 years or more. The locations of the respondents the time in the recurrence scenarios. The two most common

Table 2. All Pediatric Surgeons’ Preference for Treating Left and Right Bochdalek
Congenital Diaphragmatic Hernia (N = 280)
CDH type Laparoscopy, n (%) Thoracoscopy, n (%) Laparotomy, n (%) Thoracotomy, n (%)
Initial left 17 (6.1) 129 (46.1) 131 (46.8) 3 (1.1)
Left recurrence after thoracoscopy 46 (16.4) 49 (17.5) 168 (60.0) 17 (6.1)
Left recurrence after laparotomy 21 (7.5) 84 (30.0) 150 (53.6) 25 (8.9)
Left recurrence after thoracotomy 53 (18.9) 26 (9.3) 178 (63.6) 23 (8.2)
Right recurrence after thoracoscopy 28 (10.0) 74 (26.4) 130 (46.4) 48 (17.1)
CDH, congenital diaphragmatic hernia.

Table 3. MIS Surgeons’ Preference for Treating Left and Right Bochdalek Congenital
Diaphragmatic Hernia (N = 146)
CDH type Laparoscopy, n (%) Thoracoscopy, n (%) Laparotomy, n (%) Thoracotomy, n (%)
Initial left 17 (11.6) 129 (88.4) — —
Left recurrence after thoracoscopy 39 (26.7) 42 (28.8) 63 (43.2) 2 (1.4)
Left recurrence after laparotomy 17 (11.6) 64 (47.8) 56 (38.4) 9 (6.2)
Left recurrence after thoracotomy 40 (27.4) 22 (15.1) 72 (49.3) 12 (8.2)
Right recurrence after thoracoscopy 25 (17.1) 53 (36.3) 50 (34.2) 18 (12.3)
CDH, congenital diaphragmatic hernia.
APPROACH TO RECURRENT DIAPHRAGMATIC HERNIA 3

Table 4. Open Surgeons’ Preference for Treating Recurrent Left and Right Bochdalek
Congenital Diaphragmatic Hernia (N = 134)
CDH type Laparoscopy, n (%) Thoracoscopy, n (%) Laparotomy, n (%) Thoracotomy, n (%)
Initial left — — 131 (97.8) 3 (2.2)
Left recurrence after thoracoscopy 7 (5.2) 7 (5.2) 105 (78.4) 15 (11.2)
Left recurrence after laparotomy 4 (3.0) 20 (14.9) 94 (70.1) 16 (11.9)
Left recurrence after thoracotomy 13 (9.7) 4 (3.0) 106 (79.1) 11 (8.2)
Right recurrence after thoracoscopy 3 (2.2) 21 (15.7) 80 (59.7) 30 (22.4)
CDH, congenital diaphragmatic hernia.

recurrence situations for an open surgeon to select an MIS CDH, thoracoscopy may be preferred due to the liver making
approach were recurrent left CDH after laparotomy (14.9% laparoscopy more difficult.
thoracoscopy, 3.0% laparoscopy) and right recurrence after
thoracoscopy (15.7% thoracoscopy, 2.2% laparoscopy). Open repair for initial CDH
Open surgeons generally prefer open repair over MIS due to
Discussion greater comfort operating through laparotomy or thoracotomy
MIS for initial CDH or due to the lower recurrence profile suggested by a number of
articles.3–8 In this study, 48% of surgeons selected an open
There are a number of factors that influence approach to a procedure for initial CDH. Laparotomy was vastly more
left Bochdalek CDH. The potential benefits of MIS repair of popular with 46% of all surgeons and 98% of open surgeons
CDH include decreased pain, improved cosmetic result, choosing it. When comparing the laparotomy to thoracotomy,
faster recovery times, and, most importantly, avoidance of laparotomy may be preferred due to better overall view and
morbidity of laparotomy (adhesive disease) and thoracotomy avoidance of potential chest wall defects and scoliosis asso-
(scoliosis).2–4 Even within MIS techniques, there are a vari- ciated with thoracotomy.2,9 Similar to MIS, in the case of right
ety of factors that can influence which approach to utilize in CDH, the liver can be the main obstacle of the repair. While
various scenarios. Advantages of thoracoscopy over lapa- this may lead some surgeons to use a thoracotomy, laparotomy
roscopy for initial CDH repair include the greater working can visualize the space superior to the liver as well.
space in a thorax with an underdeveloped lung compared to a
scaphoid abdomen and the relative ease of thoracoscopically MIS for recurrent CDH
visualizing and accessing the posterolateral aspect of the
defect which often extends overlying the retroperitoneum MIS surgeons would frequently use MIS for a CDH re-
compared to the laparoscopic approach. Finally, it is often currence as 42.5%–55.5% of MIS surgeons selected an MIS
easier to reduce the herniated viscera by pushing it through approach in recurrence scenarios. Thoracoscopy was the MIS
the defect with the aid of thoracic insufflation compared to procedure of choice in all cases except for a left-sided re-
pulling it laparoscopically. Laparoscopic repair for initial left currence after a thoracotomy in which laparoscopy was fa-
CDH is much less commonly encountered. For recurrences, vored 18.9% versus 9.3% for all surgeons and 27.4% versus
on the other hand, a laparoscopic approach allows dissection 15.1% for MIS surgeons, respectively. This may illustrate the
in a ‘‘virgin’’ workspace (as most primary repairs are per- concept of a ‘‘virgin plane’’ in which the opposite body
formed thoracoscopically) and may allow easier evaluation cavity as the initial repair is preferred due to surgical adhe-
of the herniated viscera, as well as certainty that abdominal sions. This concept has been described in other types of
viscera have not been incorporated into the repair. For right hernia such as inguinal hernia recurrences. For right-sided
recurrence, the liver most likely played a larger factor in
shifting MIS surgeons toward thoracoscopy.
Table 5. Proportion of MIS and Open Surgeons Among open surgeons, between 10% and 17% selected an
Who Select an MIS Approach for Congenital MIS approach for recurrences: majority of these were thor-
Diaphragmatic Hernia Recurrences
acoscopy. This was highest after laparotomy, lending support
MIS surgeons Open surgeons to the virgin plane theory.
CDH type (n = 146), n (%) (n = 134), n (%)
Open repair for recurrent CDH
Left recurrence after 55.5 10.4
thoracoscopy Among all surgeons polled, laparotomy was most popular
Left recurrence after 55.5 17.9 approach for all cases of recurrent CDH, suggesting that it is
laparotomy the open procedure of choice. Thoracotomy appears to have
Left recurrence after 42.5 12.7 fallen out of favor in general with less than 10% selection
thoracotomy rates for all left-sided CDH cases. Even for a recurrence after
Right recurrence after 53.4 17.9 laparotomy, laparotomy remained more popular than thora-
thoracoscopy
cotomy by a large margin (53.6% versus 30.0%, respective-
CDH, congenital diaphragmatic hernia; MIS, minimally invasive ly), indicating that the desire to operate in a virgin field is
surgery. likely not enough to compel a second open incision.
4 BRUNS ET AL.

For right-sided recurrent CDH, there was more of a pref- 2. Westfelt JN, Nordwall A. Thoracotomy and scoliosis. Spine
erence for thoracotomy, although it was never a majority. 1991;16:1124–1125.
This is illustrated by the fact that thoracotomy was utilized 3. Gourlay DM, Cassidy LD, Sato TT, Lal DR, Arca MJ. Be-
17% for the recurrent right-sided CDH after a thoracoscopic yond feasibility: A comparison of newborns undergoing
attempt versus 6% for a recurrent left-sided CDH after a thoracoscopic and open repair of congenital diaphragmatic
thoracoscopic attempt. Perhaps this is because the liver ob- hernias. J Pediatr Surg 2009;44:1702–1707.
structs an abdominal approach, rendering it more difficult. 4. Lao OB, Crouthamel MR, Goldin AB, Sawin RS, Wald-
hausen JHT, Kim SS. Thoracoscopic repair of congenital
Limitations diaphragmatic hernia in infancy. J Laparoendosc Adv Surg
Tech 2010;20:271–276.
The population of pediatric surgeons came from an e-mail 5. Gander JW, Fisher JC, Gross ER, Reichstein AR, Cowles
list from GlobalCastMD. GlobalCastMD is an online virtual RA, Aspelund G, Stolar CJH, Kuenzler KA. Early recur-
platform for medical education. It is possible that this group rence of congenital diaphragmatic hernia is higher after
of surgeons would be more technologically oriented than the thoracoscopic than open repair: A single institutional study. J
rest of the pediatric surgery population and could potentially Pediatr Surg 2011;46:1303–1308.
be more likely to use MIS. 6. Shah SR, Wishnew J, Barsness K, Gaines BA, Potoka
Surgeon preferences on treating initial right CDH were not DA, Gittes GK, Kane TD. Minimally invasive congen-
ascertained in this survey. Without this information, it is ital diaphragmatic hernia repair: A 7-year review of one
difficult to draw conclusions as to how approach changes institution’s experience. Surg Endosc 2009;23:1265–
when faced with a right recurrence. 1271.
Further study is needed to elicit what the advantages and 7. Kim AC, Bryner BS, Akay B, Geiger JD, Hirschl RB, My-
chaliska GB. Thoracoscopic repair of congenital diaphrag-
disadvantages are of the available technical options for repair
matic hernia in neonates: Lessons learned. J Laparoendosc
of CDH.
Adv Surg Tech 2009;19:575–580.
8. McHoney M, Giacomello L, Nah SA, De Coppi P, Kiely
Conclusion EM, Curry JI, Drake DP, Eaton S, Pierro A. Thoracoscopic
Among MIS approaches, thoracoscopy is preferred to repair of congenital diaphragmatic hernia: Intraoperative
laparoscopy for initial CDH and all recurrences except after ventilation and recurrence. J Pediatr Surg 2010;45:355–
thoracotomy. Surgeons who approach an initial left CDH in 359.
an MIS manner would frequently attempt an MIS repair for a 9. Frola C, Serrano J, Cantoni S, Casiglia M, Turtulici I,
recurrence. There is a tendency to approach a CDH recur- Loria F. CT findings of atrophy of chest wall muscle
rence from the opposite body cavity as the initial repair. after thoracotomy: Relationship between muscles in-
volved and type of surgery. AJR Am J Reoentgen 1995;
Among open approaches, laparotomy is favored over thora-
164:599–601.
cotomy for initial and recurrent CDH. These findings may
inform surgeons when confronted with a CDH recurrence.

Disclosure Statement
Address correspondence to:
Dr. Bruns, Dr. Glenn, Mr. McNinch, Ms. Arps, and Dr. Nicholas E. Bruns, MD
Schlager have no disclosures. Dr. Ponsky is the co-owner and Division of Pediatric Surgery
chief medical officer of GlobalCastMD. Akron Children’s Hospital
One Perkins Square
References Suite 8400
1. Torfs CP, Curry CJ, Bateson TF, Honoré LH. A population- Akron, OH 44308
based study of congenital diaphragmatic hernia. Teratology
1992;46:555–565. E-mail: nickebruns@gmail.com

Appendix A. Congenital Diaphragmatic Hernia Survey

1. How long have you been practicing pediatric surgery?


a. 0–10 years
b. 11–20 years
c. 20+ years
d. I am a trainee (resident or fellow)
e. I am not a pediatric surgeon
2. Where do you practice?
a. Asia
b. Europe
c. North America
d. South America
e. Other
APPROACH TO RECURRENT DIAPHRAGMATIC HERNIA 5

3. Suppose you are evaluating an infant in the neonatal intensive care unit (NICU) with a LEFT-SIDED Bochdalek
CDH. The infant is hemodynamically stable and has been cleared for surgery. Would you attempt a minimally
invasive (MIS) repair?
a. Yes, I would use a laparoscopic approach
b. Yes, I would use a thoracoscopic approach
c. No, I would perform a laparotomy
d. No, I would perform a thoracotomy
4. Would you attempt an MIS approach for a RECURRENT LEFT-SIDED Bochdalek CDH after a THORACO-
SCOPIC repair?
a. Yes, I would use a laparoscopic approach
b. Yes, I would use a thoracoscopic approach
c. No, I would perform a laparotomy
d. No, I would perform a thoracotomy
5. How often do you predict that an MIS repair will be successfully completed without conversion to open in the patient
with a prior THORACOSCOPIC repair?
a. <25% of cases
b. 25%–50% of cases
c. 50%–75% of cases
d. >75% of cases
6. Would you attempt an MIS approach for a RECURRENT LEFT-SIDED Bochdalek CDH after a prior THOR-
ACOTOMY?
a. Yes, I would use a laparoscopic approach
b. Yes, I would use a thoracoscopic approach
c. No, I would perform a laparotomy
d. No, I would perform a thoracotomy
7. Would you attempt an MIS approach for a RECURRENT LEFT-SIDED Bochdalek CDH after a prior LAPAROTOMY?
a. Yes, I would use a laparoscopic approach
b. Yes, I would use a thoracoscopic approach
c. No, I would perform a laparotomy
d. No, I would perform a thoracotomy
8. How often would you predict that an MIS technique will be successfully completed without conversion to open
following a prior OPEN repair?
a. <25% of cases
b. 25%–50% of cases
c. 50%–75% of cases
d. >75% of cases
9. Would you attempt an MIS approach for a RECURRENT RIGHT-SIDED Bochdalek CDH after a THORACO-
SCOPIC repair?
a. Yes, I would use a laparoscopic approach
b. Yes, I would use a thoracoscopic approach
c. No, I would perform a laparotomy
d. No, I would perform a thoracotomy
10. Have you ever witnessed or experienced a patch infection following a CDH repair with a prosthetic patch?
a. Yes
b. No
If so, to what do you attribute the infection?
_______________

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