JOURNAL OF LAPAROENDOSCOPIC & ADVANCED SURGICAL TECHNIQUES Volume 25, Number 4, 2015 ª Mary Ann Liebert, Inc. DOI: 10.1089/lap.2014.0237

2014 IPEG Papers

Diaphragmatic Eventration in Children: Laparoscopy Versus Thoracoscopic Plication Go Miyano, MD,1 Masaya Yamoto, MD,1 Masakatsu Kaneshiro, MD,1 Hiromu Miyake, MD,1 Keiichi Morita, MD,1 Hiroshi Nouso, MD,1 Mariko Koyama, MD,1 Manabu Okawada, MD,2 Takashi Doi, MD,2 Hiroyuki Koga, MD,2 Koji Fukumoto, MD,1 Geoffrey J. Lane, MD,2 Atsuyuki Yamataka, MD,2 and Naoto Urushihara, MD1

Abstract

Aim: To determine the best way to perform diaphragmatic plication for diaphragmatic eventration (DE) using minimally invasive surgery. Materials and Methods: We conducted a retrospective review of pediatric cases of DE treated between 2007 and 2012. Thoracoscopic plication (TP) is performed using single-lung ventilation with three 5-mm ports; laparoscopic plication (LP) is performed using three or four 5-mm ports. The choice of technique was determined preferentially by the treating surgeon. Results: There were 20 subjects (13 treated by LP and 7 treated by TP). Etiology of DE was phrenic nerve injury (LP, n = 11; TP, n = 1) and muscular deficiency (LP, n = 2; TP, n = 6). Mean age (LP, 18 months; TP, 25 months) and weight (LP, 8.0 kg; TP, 9.7 kg) at surgery were not significantly different. Mean operating time was 155.6 minutes in LP and 167.0 minutes in TP (P = not significant). Mean intraoperative end-tidal CO2 was 41.9 mm Hg (range, 35– 52 mm Hg) in LP and 36.9 mm Hg (range, 33–41 mm Hg) in TP (P = .01). Mean duration of postoperative ventilation was 1.2 days in LP and 1.3 days in TP (P = not significant). Mean time taken to recommence feeding postoperatively was 1.6 days in both groups (P = not significant). Complications were one conversion to thoracotomy in TP, 1 case of atelectasis in each group (P = not significant), and 6 cases of recurrence in LP versus none in TP (P = .04). Conclusions: Both TP and LP are beneficial for treating small children with DE. However, there is a higher incidence of recurrence after LP, and the role of TP in cardiac patients requiring subsequent surgery is debatable. Introduction

D

iaphragmatic eventration (DE) occurs as a consequence of phrenic nerve injury or congenital muscular deficiency of the diaphragm.1,2 It may also be iatrogenic, caused by traction on the phrenic nerve during delivery or as a complication of intramediastinal surgery.3,4 Traditional surgical intervention is complicated, particularly in symptomatic smaller children, but diaphragmatic plication performed using a conventional posterolateral thoracotomy is a straightforward procedure: easy to perform technically with a low complication rate. As in all fields of surgery, advancements in technology have enabled more open procedures to be performed endoscopically, and DE has been treated even in infants using minimally invasive surgery (MIS).5–7 There are reports in the literature about laparoscopic diaphragmatic plication with sufficient long-term follow-up in 1 2

adults, with particular mention of its benefits for treating patients with phrenic nerve injury after intramediastinal surgery, including cardiac surgery.8 However, although there are no reports that compare diaphragmatic plication techniques directly, the results of transthoracic and transabdominal approaches would seem to be comparable. This fact motivated us to conduct a retrospective review of our DE patients to determine the most appropriate MIS technique for performing diaphragmatic plication (i.e., thoracoscopic plication [TP] or laparoscopic plication [LP]). Materials and Methods

We reviewed the medical records of children diagnosed with DE at two pediatric surgical centers, Shizuoka Children’s Hospital (Shizuoka, Japan) and Juntendo University Hospital (Tokyo, Japan), between 2007 and 2012 (Institutional Review Board approval number 2013–37). Patients

Department of Pediatric Surgery, Shizuoka Children’s Hospital, Shizuoka, Japan. Department of Pediatric General & Urogenital Surgery, Juntendo University School of Medicine, Tokyo, Japan.

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with DE who had open thoracotomy or laparotomy were excluded from this study. Data for age, sex, incidence and location of the eventration, presence of associated anomalies, symptoms, surgical technique, intraoperative hemodynamic change, outcome, complications, and rates of recurrence and survival were collated. Diagnosis of DE was made based on physical examination, chest x-ray, and fluoroscopy. DE was diagnosed on chest X-ray if the affected hemidiaphragm was at least two intercostal spaces higher than the other side. Patients with radiologic signs but who were asymptomatic or in no respiratory distress were not indicated for surgical intervention. Recurrence of DE after plication of the diaphragm was defined as the presence of at least a two intercostal space difference between the operated and unoperated sides on fluoroscopy (for both sides), with relapse respiratory symptoms. TP is performed with the patient under general anesthesia using single-lung ventilation with a Fogarty embolectomy catheter. The patient is placed in the decubitus position, and three 5-mm ports are created in the fifth intercostal space at the midaxillary line, the mammary line, and a line extending down from the tip of the scapula. Pneumothorax is established at a pressure of 4 mm Hg, and plication of the diaphragm is performed using interrupted 3-0 or 4-0 nonabsorbable sutures tied extracorporeally until no excess tissue remains. No chest tube is used. LP is performed with the patient in a 30 reverse Trendelenburg position after endotracheal intubation. Three or four 5-mm trocars are inserted in the umbilicus and the right and left lower abdomen, and pneumoperitoneum is established at a pressure of 8 mm Hg or less. In cases of right DE, the hepaticoumbilical ligament is excised and freed from the abdominal wall to improve the operative field and enhance plication. LP is performed with 3-0 or 4-0 nonabsorbable sutures tied extracorporeally, with the initial line of sutures for plication being sutured to the anterior abdominal wall for stability. No drainage tube is placed.

Both TP and LP were performed by pediatric surgical trainees supervised by board-certified pediatric surgeons. Choice of procedure (TP or LP) was determined by the operating surgeon according to personal preference. Data were analyzed using standard statistical methods. Demographic data were compared using Student’s t test. The chi-squared test or Fisher’s exact test was used for analyzing the incidence of complications. For all statistics, .05 was used to determine significance. Results

We treated 20 cases of DE by TP or LP during the study period. Of these, 13 (10 male, 3 female) had LP, and 7 (5 male, 2 female) had TP. Etiology in LP was phrenic nerve injury secondary to cardiac surgery (n = 9) or mediastinal tumor resection (n = 2) and congenital muscular deficiency of the diaphragm (n = 2). Etiology in TP was phrenic nerve injury secondary to cardiac surgery (n = 1) and congenital muscular deficiency of the diaphragm (n = 6). In LP, eventration was left-sided in 9 cases, right-sided in 2 cases, and bilateral in 2 cases. In TP, eventration was left-sided in 4 cases and right-sided in 3 cases. Respiratory distress developed in all cases, and preoperative ventilator support was required in 6 LP cases and 3 TP cases; supplemental oxygen was required in 9 LP cases and 4 TP cases. Characteristics of patients in the two groups are summarized in Table 1. Mean age at the time of surgery was 18.3 months (range, 0–45 months) in LP and 25.1 months (range, 0–75 months) in TP. Mean weight at the time of surgery was 8.0 kg (range, 2.7–15.9 kg) in LP and 9.7 kg (range, 2.2– 27 kg) in TP. Mean operating time was 155.6 minutes (range, 90–290 minutes) in LP and 167.0 minutes (range, 122–303 minutes) in TP (P = not significant). Mean intraoperative end-tidal CO2 was 41.9 mm Hg (range, 35– 52 mm Hg) in LP and 36.9 mm Hg (range, 33–41 mm Hg) in TP, which was statistically significant (P = .01). No LP case

Table 1. Comparison of Laparoscopic and Thoracoscopic Diaphragmatic Plication Parameter Number of patients Mean (range) age (months) at surgery Mean (range) weight (kg) at surgery Male/female ratio Pathology (left/right/bilateral) Etiology (phrenic nerve injury/congenital) Preoperative ventilation Mean (range) operative time (minutes) Mean (range) end-tidal CO2 Conversion to open surgery Permanent postoperative ventilation Mean (range) duration (days) of postoperative ventilationa Mean (range) time (days) taken to resume feeding Complications Recurrence Mean length of follow-up (years) a

LP

TP

Significance

13 18.3 (0–45) 8.0 (2.7–15.9) 10/3 9/2/2 11/2 6 155.6 (90–290) 41.9 (35–52) 0 2 1.2 (0–5) 1.6 (1–4) 1b 6 2.7

7 25.1 (0–75) 9.7 (2.2–27) 5/2 4/3/0 1/6 3 167.0 (122–303) 36.9 (33–41) 1 1 1.3 (0–5) 1.6 (1–4) 1b 0 2.4

NS NS NS NS P = .01 NS NS P = .01 NS NS NS NS NS P = .04 NS

Excluding cases requiring permanent ventilation. Atelectasis. LP, laparoscopic diaphragmatic plication; NS, not significant; TP, thoracoscopic diaphragmatic plication.

b

EVENTRATION, LAPAROSCOPY, AND THORACOSCOPY

required conversion to conventional open laparotomy or thoracotomy; however, 1 TP case required conversion to thoracotomy due to technical difficulty (P = not significant). Postoperative ventilator support was required permanently in 2 LP cases and 1 TP case as a consequence of the original indication for plication, including chromosomal anomaly and chronic respiratory failure (P = not significant). Mean duration of postoperative ventilator support was 1.2 days (range, 0–5 days) in LP and 1.3 days (range, 0–5 days) in TP (P = not significant). Mean time taken to recommence feeding postoperatively was 1.6 days (range, 1–4 days) in LP and 1.6 days (range, 1–4 days) in TP (P = not significant). As complications, there was 1 case of atelectasis in each group (P = not significant) and 6 cases of recurrence in LP versus none in TP, which was statistically significant (P = .04). All six recurrences were treated by re-do LP. Mean duration of follow-up was 2.7 years for LP and 2.4 years for TP (P = not significant). Discussion

Advances in MIS have led to conventional open thoracic and abdominal surgical procedures being performed almost entirely using thoracoscopy or laparoscopy. In 1995, Gharagozloo et al.9 reported using video-assisted thoracic surgery to plicate the diaphragm in a 72-year-old women, and in 1998, Van Smith et al.10 reported the use of this approach to plicate the diaphragm after phrenic nerve injury complicated surgery for congenital heart disease in a neonate. Since then, there have been several case reports and case series describing successful TP in children.5–7,11,12 In contrast, reports about LP in adults are limited,8 and there are no reports in children. Although the conventional surgical treatment for DE is open transthoracic plication, intrathoracic adhesions may hinder progress, especially in patients with phrenic nerve palsy complicating intramediastinal surgery.8 In such cases underlying cardiac anomalies often require staged repair (i.e., palliative surgery prior to definitive surgery); adhesions could be more problematic during TP than during conventional open transthoracic surgery,8 and TP could hinder future planned cardiac surgery by also causing adhesions. Although TP is less invasive than open surgery, single-lung ventilation and artificial pneumothorax are required and could be an issue of concern, particularly in children (i.e., CO2 insufflation could lead to hemodynamic instability).13,14 However, in this study, although the sample size is small, we found intraoperative endtidal CO2 was lower in TP than LP, with no significant differences between TP and LP for postoperative hemodynamic recovery, including the timing of extubation and cessation of oxygen supplementation. Time taken to recommence feeding was also not statistically significant between the two groups, an indication that the surgical impact of both techniques was similar. In fact, we believe our approach is physiologically sound because we were able to reduce intrathoracic insufflation pressure to 4 mm Hg, which is less than the reported safe level in the literature (< 5 mm Hg),7 and we operated successfully on small infants and neonates despite the small intrathoracic working space, with only 1 case requiring conversion to thoracotomy and no postoperative complications after a mean follow-up of 2.4 years.

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Although LP typifies the benefits of MIS, with excellent wound cosmesis because only three or four 5-mm incisions are required in the upper and central abdomen and LP could be scheduled earlier if needed to help speed recovery from cardiac surgery, the fact remains that there was a significantly higher incidence of recurrence with LP in our series, which may be related to differences in etiology of DE (phrenic nerve palsy in LP and congenital muscular deficiency in TP) or limited view of the diaphragm during LP because of obstruction by the liver, stomach, or intestines, which could pose greater problems in smaller patients, even though we managed to treat a case of bilateral DE in a single operation using LP. We found that both TP and LP appear to be safe and beneficial for treating small children with DE. However, because of statistically higher incidence of recurrence of DE after LP, continuing debate about the role of TP in treating DE in patients requiring further cardiac surgery because of surgical intervention in the thorax, and possible risk for hemodynamic instability caused by pneumothorax, we cannot recommend one technique over the other at this stage. A larger series would provide more evidence to establish which technique is the MIS option of choice for the repair of DE in small children. Disclosure Statement

No competing financial interests exist. References

1. Wayne ER, Campbell JB, Burrington JD, et al. Eventration of the diaphragm. J Pediatr Surg 1974;9:643–651. 2. Smith CD, Sade RM, Crawford FA, et al. Diaphragmatic paralysis and eventration in infants. J Thorac Cardiovasc Surg 1986;91:490–497. 3. Mearns AJ. Iatrogenic injury to the phrenic nerve nerve in infants and young children. Br J Surg 1977;64:558– 560. 4. Mok Q, Ross-Russell R, Mulvey D, et al. Phrenic nerve injury in infants and children undergoing cardiac surgery. Br Heart J 1991;65:287–292. 5. Takahashi T, Okazaki T, Ochi T, et al. Thoracoscopic plication for diaphragmatic eventration in a neonate. Ann Thorac Cardiovasc Surg 2013;19:243–246. 6. Becmeur F, Talon I, Schaarschmidt K, et al. Thoracoscopic diaphragmatic eventration repair in children: About 10 cases. J Pediatr Surg 2005;40:1712–1715. 7. Sato M, Hamada Y, Takada K, et al. Thoracoscopic diaphragmatic procedures under artificial pneumothorax. Pediatr Surg Int 2005;21:34–38. 8. Huttl TP, Wichmann MW, Reichart B, et al. Laparoscopic diaphragmatic plication: Long-term results of a novel surgical technique for postoperative phrenic nerve palsy. Surg Endosc 2004;18:547–551. 9. Gharagozloo F, McReynolds SD, Snyder L. Thoracoscopic plication of the diaphragm. Surg Endosc 1995;9:1204–1206. 10. Van Smith C, Jacobs JP, Burke RP. Minimally invasive diaphragm plication in an infant. Ann Thorac Surg 1998; 65:842–844. 11. Abraham MK, Menon SS, S BP. Thoracoscopic repair of eventration of diaphragm. Indian Pediatr 2003;40:1088– 1089.

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12. Hines MH. Video-assisted diaphragm plication in children. Ann Thorac Surg 2003;76:234–236. 13. Hill RC, Jones DR, Vance RA, et al. Selective lung ventilation during thoracoscopy: Effects of insufflation on hemodynamics. Ann Thorac Surg 1996;61:945–948. 14. Jones DR, Graeber GM, Tanguilig GG, et al. Effects of insufflation on hemodynamics during thoracoscopy. Ann Thorac Surg 1993;55:1379–1382.

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Address correspondence to: Go Miyano, MD Department of Pediatric Surgery Shizuoka Children’s Hospital 860 Urushiyama Aoi-ku, Shizuoka 420-8660 Japan E-mail: [email protected]

Diaphragmatic eventration in children: laparoscopy versus thoracoscopic plication.

To determine the best way to perform diaphragmatic plication for diaphragmatic eventration (DE) using minimally invasive surgery...
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