YJPSU-59513; No of Pages 5 Journal of Pediatric Surgery xxx (xxxx) xxx
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Thoracoscopic repair of diaphragmatic eventration in children: a comparison of two repair techniques Osama A Bawazir ⁎, Abdulaziz M Banaja 1 Department of Surgery, Umm Al-Qura University King Faisal Specialist Hospital & Research Centre
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Article history: Received 26 August 2019 Received in revised form 27 October 2019 Accepted 14 November 2019 Available online xxxx Key words: Diaphragmatic eventration Thoracoscopy Diaphragmatic plication
a b s t r a c t Background: Thoracoscopic plication has gained popularity in the management of diaphragmatic eventration, and several suturing techniques have been described. However, the superiority of one technique over the other has not been demonstrated. The purpose of this study is to report our experience with diaphragmatic plication and to compare the thoracoscopic interrupted and pleated suture techniques in pediatric patients with diaphragmatic eventration. Methods: This is a retrospective cohort study (level of evidence: 3) performed on 14 patients with diaphragmatic eventration. All patients were symptomatic and had diaphragmatic plication via thoracoscopy. The patients were further divided into two groups according to the repair technique; interrupted repair (n = 9) and pleated repair (n = 5). Preoperative, operative and postoperative data were compared between the two groups. Results: The median age was 9.5 months (25th- 75th percentiles: 6 to 15 months), and 8 (57%) were males. Twelve patients (85.71%) had right side eventration, and nine patients (64.29%) had congenital diaphragmatic eventration. One case was converted to open thoracotomy because of adhesions. There was no difference in the preoperative characteristics between both groups. Median operative time was 117 min (25th- 75th percentiles: 101–129 min) and 77 min (25th- 75th percentiles: 73–83 min) in the interrupted and pleated groups, respectively (p = 0.004). One patient had a postoperative elevation of the diaphragm (incomplete repair) in the pleated group (p = 0.357). No recurrence was reported during the follow-up. Conclusion: Thoracoscopic plication is an effective technique for management of diaphragmatic eventration in children. Pleating technique is easy, fast, and associated with a marked reduction in operative time. Type of the study: Retrospective cohort study. Levels of Evidence: Level of evidence: 3 © 2019 Elsevier Inc. All rights reserved.
Diaphragmatic eventration (DE) is a rare condition characterized by an elevation of the hemidiaphragm without defects of continuity. [1] Diaphragmatic eventration can occur due to a congenital migration defect of myoblasts during embryogenesis or acquired phrenic nerve palsy as a result of birth injury or after surgery. [2] Surgical plication of the diaphragm is indicated for symptomatic patients, and some investigators recommend a conservative approach for focal and asymptomatic eventration. [3] The goal of surgery is to provide a space for lung expansion by restoring the normal position and preventing the paradoxical motion of the diaphragm. Since the original description of diaphragmatic plication, numerous modifications, including the minimally invasive video-assisted thoracoscopic surgery have been described [4,5]. Recently, minimally invasive repair of DE became the standard approach, [6] and the thoracoscopic plication became
more prevalent than laparoscopic plication because it provides ample working space, and gives direct visualization of the phrenic nerve. [7] Thoracoscopic plication of the diaphragm has been performed on children for years and has shown good results. [6] Diaphragmatic plication is usually obtained by suturing the entire dome or by using an external spinal needle and pleating the dome of the diaphragm over it. The advantage of one technique over the other is debatable. The purpose of this study is to report our experience with diaphragmatic plication and to compare the thoracoscopic interrupted and pleated suture repair of diaphragmatic eventration in children.
⁎ Corresponding author at: Department of surgery Faculty of medicine in Umm Al-Qura University at Makkah, P.O.box 715, Makkah 21955, Saudi Arabia. Tel.: +966125270000x4125 (Office). E-mail addresses:
[email protected],
[email protected] (O.A. Bawazir),
[email protected] (A.M. Banaja). 1 Co- Author.
This research is a retrospective cohort study performed on patients who had thoracoscopic plication of the diaphragm between January 2006 and December 2018. All patients had plication for diaphragmatic eventration, and the indications for intervention were difficult weaning from mechanical ventilation (n = 7), failed multiple extubation
1. Materials and methods 1.1. Study design and ethical standards
https://doi.org/10.1016/j.jpedsurg.2019.11.019 0022-3468/© 2019 Elsevier Inc. All rights reserved.
Please cite this article as: O.A. Bawazir and A.M. Banaja, Thoracoscopic repair of diaphragmatic eventration in children: a comparison of two repair techniques, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.11.019
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O.A. Bawazir, A.M. Banaja / Journal of Pediatric Surgery xxx (xxxx) xxx
attempts (n = 3), and recurrent or persistent chest infection (n = 4). The Institutional Review Board of King Faisal Specialist Hospital and Research Center- Jeddah- Saudi Arabia approved the study, and the need for patients' consent was waived (Reference number: RC-J/456/40). The diagnosis of diaphragmatic eventration was suspected on chest X-ray and fluoroscopy. Further investigations using a computed tomography (CT) scan and chest ultrasound were performed to exclude diaphragmatic hernia and show whether the eventration is localized or diffuse. 1.2. Surgical technique Surgical plication of the diaphragm was performed in symptomatic patients. The thoracoscopic repair was performed in all patients under general anesthesia and single-lumen endotracheal intubation. After positioning the patient in the lateral position, CO2 was insufflated using 4 mmHg pressure to deflate the lung. Three ports were inserted, the camera port was placed in the fourth intercostal space in the midaxillary line, and two working ports were placed in the fourth intercostal space on the anterior axillary line and in the fourth or fifth intercostal space on the posterior axillary line. Positions of the ports were adjusted for each patient; occasionally, an extra port was used for retraction. CO2 insufflation collapsed the eventrated diaphragm, which indicated no adhesion to the diaphragm in the abdominal cavity. The eventrated diaphragm was then plicated using two techniques; interrupted sutures (n = 9) and pleated suture (n = 5) using an external long spinal needle as described by Snyder and colleagues. [8] We folded both sides of the
weakened diaphragm to the middle and performed interrupted sutures with nonabsorbable material (Interrupted sutures technique) (Fig. 1). On the other hand, the “pleating” technique was performed like an accordion, over the spinal needle, and with one stitch to put them together in each pass, and a total of 4 to 5 stitches were needed to plicate the whole diaphragm (Fig. 2). At the end of the procedure, the intrathoracic CO2 was removed, and a small chest tube was inserted. The surgical technique was standardized in all patients, and a single consultant surgeon has performed all the operations. The choice of one technique over the other was according to the surgeon preference and not related to patients characteristics. The “pleating” technique presents the evolution of diaphragmatic repair techniques and mobile diaphragm with no adhesion and small space favor its application. 1.3. Data collection Preoperative data including age, gender, weight, side, and etiology of the eventration were collected. Operative time, duration of postoperative mechanical ventilation, and hospital stay were compared between the two groups. 1.4. Statistical analysis Continuous variables were presented as the median, 25th and 75th percentiles and range, and categorical variables as number and percent. Wilcoxon rank-sum test was used to compare continuous variables between the two groups and Fisher exact test to compare the categorical
Fig. 1. A) Preoperative CT scan of right-side diaphragmatic eventration, B and C) Repair of the eventration using the interrupted suture technique. D) Postoperative chest x-ray.
Please cite this article as: O.A. Bawazir and A.M. Banaja, Thoracoscopic repair of diaphragmatic eventration in children: a comparison of two repair techniques, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.11.019
O.A. Bawazir, A.M. Banaja / Journal of Pediatric Surgery xxx (xxxx) xxx
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Fig. 2. A) Preoperative chest x-ray of a right-side diaphragmatic eventration. B and C) Diagramatic demonstration of the pleating technique. D) Postoperative chest X-ray.
variables. A p-value of less than 0.05 was considered statistically significant. All analyses were performed using Stata 14.2 (Stata Corp, College Town, Texas, USA) 2. Results During the study period, 14 patients were diagnosed with diaphragmatic eventration and had thoracoscopic plication. Their median age was 9.5 months (range: 3 to 25 months -25th- 75th percentiles: 6 to 15 months), and 8 (57%) were males. The patients' median body weight was 6.95 Kg (ranged from 3.6 to 12.6 kg- 25th and 75th percentiles: 4.9–9.3 Kg). Twelve patients (85.71%) had right side eventration, and nine patients (64.29%) had congenital diaphragmatic eventration. One case was converted to open thoracotomy due to adhesions. Two techniques for diaphragmatic plication were used; the interrupted suture (n = 9) and the pleated suture repair (n = 5). There was no difference in the preoperative characteristics between both groups. (Table 1).
The median operative time was 103 min (25th and 75th percentiles: 61–151 min). Median operative time was 117 min (25th and 75th percentiles: 101–129 min) and 77 min (25th and 75th percentiles: 73–83 min) in the interrupted and pleated groups, respectively. (Fig. 3) One patient had a postoperative elevation of the diaphragm (incomplete repair) in the pleated group (p = 0.357). No statistically significant difference was found between the groups in the duration Table 1 Preoperative characteristics. (Continuous variables are presented as median (25th- 75th percentile) and categorical variables as number and percent).
Age (months) Weight (Kg) Male Congenital etiology Right side eventration
Interrupted repair (n = 9)
Pleated repair (n = 5)
p-value
9 (7–13) 6.8 (5.7–8.3) 5 (55.56%) 6 (66.67%) 8 (88.9%)
10 (6–19) 7.1 (4.9–11.7) 3(60%) 3 (60%) 4 (80%)
0.739 0.739 0.657 0.622 0.604
Please cite this article as: O.A. Bawazir and A.M. Banaja, Thoracoscopic repair of diaphragmatic eventration in children: a comparison of two repair techniques, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.11.019
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140
160
4
60
80
100
120
p= 0.004
Interrupted reepair
Pleated repair
Fig. 3. A boxplot of the operative time in both groups.
of mechanical ventilation and hospital stay. (Table 2) Total median follow-up was 3.75 years (25th and 75th percentiles: 2.3–7 years), and no recurrence was reported during this period. Median follow-up was 6.5 years (25th and 75th percentiles: 4–7.5 years) and 2.3 years (25th and 75th percentiles: 1.7–2.6 years) in the interrupted and pleated suture repair, respectively. 3. Discussion Diaphragmatic eventration either congenital or acquired impairs normal lung expansion due to the paradoxical motion of the hemidiaphragm. Consequently, eventration can lead to lung collapse and repeated chest infection with CO 2 retention and failure of weaning in mechanically ventilated patients. [9,10] The consequences of diaphragmatic eventration are more pronounced in younger patients, and it was found that children older than two years have better tolerability to DE and can be extubated without plication. [11] Symptomatic patients with diaphragmatic eventration are managed with plication with acceptable results, and asymptomatic patients are followed conservatively. [12] In this research, we reported our experience with thoracoscopic plication of DE in children. Male predominance was observed in this study, and the right hemidiaphragm was affected in 86% of the patients, and these results are consistent with what was reported previously. [3] DE was suspected in patients presenting with a recurrent chest infection or in patients with failure to wean from mechanical ventilation, and the diagnosis was confirmed by chest x-ray and fluoroscopy in all patients. CT scan was performed preoperatively to exclude the diaphragmatic hernia, show the extent of the eventration, and plan the surgery. The objective of diaphragmatic plication is to reduce the paradoxical motion of the diaphragm and provide a space for lung expansion.
Several approaches have been used to perform plication, including the endoscopic repair. Thoracoscopic and laparoscopic repairs of DE have the advantages of a small incision and faster recovery, and both techniques yielded good results in the management of DE. [13] CO2 insufflation during pediatric surgery can lead to hypoxia, hypercapnia, and hemodynamic changes. [14] Children have more permeability to CO2; therefore, they are more susceptible to hypercapnia and acidosis. [15] Monitoring is essential during thoracoscopy in children, especially end-tidal CO2 and O2 saturation. We did not have any complication related to the procedure or CO2 insufflation in our series; generally, we did not use single lung ventilation or high-pressure CO2 insufflation. Eventration is more common on the right side, and thoracoscopic repair of right DE provides a larger working space with a low possibility to sew the bowel with diaphragmatic stitches. A larger operative field is achieved on the left-side with the use of positive pressure CO2 insufflation. Several suturing techniques for thoracoscopic diaphragmatic plication were described; however, comparative studies for those techniques are scarce. [16,17] We used two repair techniques in our series; one with interrupted non-absorbable sutures, placed evenly to distribute the tension. The other technique was performed with pleating sutures, which required an external spinal needle. Both sutures technique can be performed through two operative ports. [18] The results of both techniques were comparable, and no recurrence was reported during longterm follow-up. The pleating technique was associated with shorter operative time; however, no difference was found in the duration of mechanical ventilation and hospital stay. One patient in the pleating repair group had a postoperative elevation of the diaphragm, which was managed conservatively and improved clinically. All patients improved clinically after repair in both groups.
Study limitations Table 2 Postoperative outcomes. Continuous variables are presented as median (25th- 75th percentile).
Operative time (minutes) Mechanical ventilation (hours) Hospital stay (days)
Interrupted repair (n = 9)
Pleated repair (n = 5)
p-value
117 (101–129) 2 (0–10) 7 (6–8)
77 (73–83) 0 (0–5) 9 (6–10)
0.004 0.831 0.460
The study has several limitations, including the small number of patients and its retrospective design. However, this study design is suitable for rare conditions. Small patients number limits the statistical analysis; despite that, the pleating technique showed significantly shorter operative time. Additionally, the treatment was not randomly assigned in the two groups, and the choice of one technique over the other depends mainly on the surgeon preference; therefore, several confounders may have affected the outcome rather than the treatment
Please cite this article as: O.A. Bawazir and A.M. Banaja, Thoracoscopic repair of diaphragmatic eventration in children: a comparison of two repair techniques, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.11.019
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itself. Moreover, the study is a single surgeon experience and the results may reflect an improvement of the primary surgeon's technique. 4. Conclusion Diaphragmatic eventration is a rare disease which occurs more frequently on the right side. Thoracoscopic plication is an effective treatment in symptomatic patients, and it is associated with improved clinical outcomes. Several sutures techniques can be used with comparable results; however, the pleating suture repair was associated with shorter operative time. The manuscript or any of its contents is not previously published in partial or full in the website or printed journal in another language than English. Funding None Authorship All authors attest that they meet the current ICMJE criteria for Authorship. Conflict of Interest Authors have no conflict of interests and the work was not supported or funded by any drug company and the authors declare that they have no competing interests. References [1] Kwak T, Lazzaro R, Pournik H, et al. Robotic thoracoscopic plication for symptomatic diaphragm paralysis. J Robot Surg 2012 Dec;6(4):345–8. [2] Ghribi A, Bouden A, Braiki M, et al. Diaphragmatic eventration in children. Tunis Med 2015 Feb;93(2):76–8.
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[3] Yazici M, Karaca I, Arikan A, et al. Congenital eventration of the diaphragm in children: 25 years' experience in three pediatric surgery centers. Eur J Pediatr Surg Off J Austrian Assoc Pediatr Surg Zeitschrift fur Kinderchirurgie 2003 Oct;13(5): 298–301. [4] Thomas RJ, Kishore R. Kisku S. a helping clamp for thoracoscopic plication of eventration of the diaphragm. J Indian Assoc Pediatr Surg 2011 Jul;16(3):97–8. [5] Morales M, Pimpalwar A. Thoracoscopic plication for diaphragmatic eventration in a 3-month-old infant. Eur J Pediatr Surg Off J Austrian Assoc Pediatr Surg . [et al] = Zeitschrift fur Kinderchirurgie. 2009 Feb;19(1):44–6. [6] Borruto FA, Ferreira CG, Kaselas C, et al. Thoracoscopic treatment of congenital diaphragmatic eventration in children: lessons learned after 15 years of experience. Eur J Pediatr Surg Off J Austrian Assoc Pediatr Surg 2014 Aug;24(4):328–31 [et al] = Zeitschrift fur Kinderchirurgie. [7] Miyano G, Yamoto M, Kaneshiro M, et al. Diaphragmatic eventration in children: laparoscopy versus thoracoscopic plication. J Laparoendosc Adv Surg Tech A 2015 Apr;25(4):331–4. [8] Snyder CW, Walford NE, Danielson PD, et al. A simple thoracoscopic plication technique for diaphragmatic eventration in neonates and infants: technical details and initial results. Pediatr Surg Int 2014 Oct;30(10):1013–6. [9] Floh AA, Zafurallah I, MacDonald C, et al. The advantage of early plication in children diagnosed with diaphragm paresis. J Thorac Cardiovasc Surg 2017 Nov;154(5): 1715–21 e4. [10] Arafat AA, Elkahwagy MS, Sherif MH. Taha A-HM. a case of right atrial compression by the elevated liver successfully treated by diaphragmatic plication. J Thorac Cardiovasc Surg 2015;150(1). [11] Simansky DA, Paley M, Refaely Y, et al. Diaphragm plication following phrenic nerve injury: a comparison of paediatric and adult patients. Thorax 2002 Jul;57(7):613–6. [12] Al-Ebrahim KE, Elassal AA, Eldib OS, et al. Diaphragmatic palsy after cardiac surgery in adult and pediatric patients. Asian Cardiovasc Thorac Ann 2019 Jul;27(6):481–5. [13] Hu J, Wu Y, Wang J, et al. Thoracoscopic and laparoscopic plication of the hemidiaphragm is effective in the management of diaphragmatic eventration. Pediatr Surg Int 2014 Jan;30(1):19–24. [14] Hill RC, Jones DR, Vance RA, et al. Selective lung ventilation during thoracoscopy: effects of insufflation on hemodynamics. Ann Thorac Surg 1996 Mar;61(3):945–8. [15] McHoney M, Corizia L, Eaton S, et al. Carbon dioxide elimination during laparoscopy in children is age dependent. J Pediatr Surg 2003 Jan;38(1):105–10. [16] Demos DS, Berry MF, Backhus LM, et al. Video-assisted thoracoscopic diaphragm plication using a running suture technique is durable and effective. J Thorac Cardiovasc Surg 2017 May;153(5):1182–8. [17] Matsubara H, Miyauchi Y, Ichihara T, et al. Thoracoscopic diaphragmatic plication for eventration of diaphragm in children using no-knife automatic suturing device. Kyobu Geka 2014 Oct;67(11):976–9. [18] Abraham MK, Menon SS. S BP. Thoracoscopic repair of eventration of diaphragm. Indian Pediatr 2003 Nov;40(11):1088–9.
Please cite this article as: O.A. Bawazir and A.M. Banaja, Thoracoscopic repair of diaphragmatic eventration in children: a comparison of two repair techniques, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.11.019