Thoracoscopic Treatment of Late-Presenting Congenital Diaphragmatic Hernia in Infants and Children

Introduction

Thoracoscopic surgery has been widely performed in children in recent decades with the increasing acceptance of its benefits.^1–3 Since Becmeur et al. first reported the use of thoracoscopy for the repair of congenital diaphragmatic hernia (CDH) in 2001, ⁴ a series of practical studies have demonstrated the theoretical advantages of the technique such as reduced tissue trauma, pain, and cosmetic deformity. ⁵ The technique also has been gradually and widely applied to treat late-presenting CDH. ⁶

The incidence of late-presenting CDH among all CDH cases varies from 5% to 25%.^7–9 Surgery is the main method used to cure this disease. Becmeur and Zhu et al. reported that thoracoscopic repair of late-presenting CDH has several advantages with good results compared with other operative approaches.^6,10 However, many studies also indicated that the procedures involved in thoracoscopic repair for late-presenting CDH were difficult, ¹¹ and that the recurrence rate was high.^12,13 In addition, recurrent late-presenting CDH is associated with a greater risk of incarceration because of the smaller hernia ring, which can cause significant damage. ¹⁴ Therefore, how to facilitate operative procedures and decrease recurrence becomes the focus.

The purpose of this article is to review clinical data from late-presenting CDH cases and summarize our experiences with thoracoscopic repair of late-presenting CDH. We aim to facilitate the operative procedures, increase the curative rate, and decrease the recurrence rate.

Materials and Methods

Surgical approach

The telescope port was placed into the thorax from the fourth intercostal space at the midaxillary line. After the insertion of the port for the optic device, which was done in an open way, a CO₂ pneumothorax was established. The other two working trocars were positioned in the middle to the anterior and posterior axillary lines in the fifth interspace. After exploring the thorax using thoracoscopy, the diaphragmatic hole was exposed, and the herniated contents were pushed back into the abdominal cavity. The stomach, colon, and small intestine were first reduced, followed by the solid visceral organs such as the spleen or the liver. The diaphragmatic defect was closed with nonabsorbable interrupted sutures (Ethibond 2/0 or 3/0, Ethicon France, Neuilly/Seine, France). A knot pusher was applied to facilitate the tie in the thorax. The periphery of the defect was cauterized by monopolar electrocautery, which was likely to promote periphery adhesion and healing and decrease the recurrence rate. For the triangular defect close to the chest wall that was difficult to address using a conventional method, a homemade hooked needle was used. ¹⁵ The needle that was threaded with a nonabsorbable suture was introduced into the thoracic cavity via direct puncture of the lateral thoracic wall at the level of the diaphragm. In the same way, another suture was introduced into the contralateral diaphragm. Then, the needle was withdrawn, leaving the thread inside the thorax. Using the needle and with the aid of the grasper, the triangular hole was closed with percutaneous circuit suturing without any gap (Fig. 1). No hernia sac was resected. At the end of the procedure, CO₂ was aspirated from the thorax by expanding the lung. A 12-18F chest tube was left in place after the procedure.

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FIG. 1.

Surgical approaches (a). The diaphragmatic defect was closed with interrupted nonabsorbable sutures, and a knot pusher was assistant to tie in thorax (b). The remaining triangular defect close to the chest wall (c). The hooked needle threaded with suture was introduced into the thoracic cavity at the level of the diaphragm (d). The needle was introduced into one side of the defect (e). The needle finished the circuit suture (f). Percutaneous extracorporeal closure for the triangular hole.

Results

Of the 59 patients, 31 were boys and 28 were girls. Their ages ranged from 2 months to 8 years (mean: 18 months). In addition, 25 patients were hospitalized for shortness of breath and dyspnea. Moreover, 34 patients had no symptoms and were found occasionally through chest radiography. There were 46 left-sided hernias and 13 right-sided hernias. Hernial sac was found in 13 cases. All cases involved Bochdalek's hernia. Extralobar pulmonary sequestrations that involved 6 cases were resected by thoracoscopic surgery simultaneously. In addition, 8 patients underwent appendectomy simultaneously for appendix herniating into the thorax. The operating time ranged from 30 to 100 minutes (mean: 55 minutes) and the amount of blood was 3–5 mL (mean: 3.8 mL). The defect size varied from 2 × 2 cm to 5 × 8 cm. No patients used the mesh. Herniated small intestines were found in all cases, herniated colon was found in 47 cases, and herniated stomach was found in 31 cases. Herniated spleen into the thorax was found in 33 cases, and herniated liver into the thorax was found in 8 cases. In the early stage, chest tubes were not inserted in the previous 10 cases, and postoperative tension pneumothorax occurred in 3 cases. In the remaining cases, chest tubes were placed at the end of the procedure, and no cases involved postoperative tension pneumothorax. Chest tubes were taken out within 24 hours after surgery. Abdominal distension was the most common complication, which was encountered in 27 cases and remitted after 3 days of fasting. There were no other complications such as bleeding, infection, small bowel obstruction, and gastroesophageal reflux. The average length of postoperative hospital stay was 5.2 ± 0.4 days (range, 4–6 days). The follow-up period ranged from 3 months to 3 years (mean: 18 months). The chest X-ray revealed no recurrence. All subjects survived and had satisfactory cosmetic outcomes (Fig. 2).

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FIG. 2.

(a). Postoperative 24-hour chest radiography of a patient with left-sided late-presenting CDH showing integrated diaphragm and pulmonary re-expansion (b). Three months after operation showing a normal X-ray (c). Showing a favorable cosmesis. CDH, congenital diaphragmatic hernia.

Discussion

Operative approaches to the repair of late-presenting CDH are diverse, and postoperative recurrence is a troublesome problem encountered with all methods. McHoney and Chan et al. reported that the recurrence rate with minimally invasive surgery (MIS) in association with CDH was 5%–23.1%.^16,17 In particular, the recurrence rate associated with thoracoscopic surgery was higher than that of other operative approaches. Recurrent late-presenting CDH not only required reoperation but also was more likely to incarcerate because of the smaller hernia ring that might cause significant damage. ¹⁴ To analyze the reasons for recurrence, thoracoscopic technology first needed a longer learning curve, as surgical procedures are more complex than transabdominal or transthoracic open techniques as well as laparoscopic MIS. ¹¹ Second, the other reasons for recurrence were that the posterior margin of the diaphragm did not dissociate sufficiently and considerable tension existed. ¹¹ Moreover, it was difficult to suture the diaphragmatic defect near the chest wall because a leak was likely to form, especially a large diaphragmatic defect, and result in recurrence. ¹⁸ Currently, with the widespread use of thoracoscopic surgery in children, ² a longer learning curve was not regarded as a difficulty. The manipulative details of thoracoscopic repair became the most important factor in the prognosis of late-presenting CDH. Researchers also reported that intraoperative carbon dioxide (CO₂) pneumothorax and a patch could be used to enlarge the pleural space and repair a large defect, respectively.^6,19 However, questions associated with the dissociation, suture, and tie related to the diaphragmatic defect were not resolved, and the recurrence rate was still high. Given the aforementioned observations, we modified the operative details of the thoracoscopic surgery to treat late-presenting CDH, and the results showed that the questions could be answered satisfactorily by these details.

Intraoperative CO₂ pneumothorax was established in accordance with previous reports. ⁶ The thoracoscope had an amplifying effect and provided a perfect view of the internal thoracic anatomy. We could clearly identify the facial edge of the diaphragmatic defect and observe the thorax by adjusting the position of the optic lens. Suturing and tying in thorax were considered to be the most difficult manipulations in the endoscopic procedure. We emphasized the use of a knot pusher and homemade hooked needle to facilitate the operative procedure. A knot pusher was assistant to the tie in the thorax and resolved the difficulties such as a small pleural space in children and knotted tension. Aiming at a triangular defect close to the chest wall, a homemade hooked needle referred to as a hernial repair needle was used. Using the needle and with the aid of the grasper, the triangular hole was closed with percutaneous circuit suturing without any gap. Knot tying was performed extracorporeally, which ensured a firm tie. The method of suture for a triangular defect was similar to the percutaneous closure for an inguinal hernia. ¹⁵ The periphery of the defect was cauterized via monopolar electrocautery that made periphery adhesion and healing easier and decreased the possibility of recurrence. The manipulation should be visualized and avoid injuring the viscera below the diaphragm. At follow-up, the chest X-ray revealed no recurrence. The results showed that when the focus was on the operative details, thoracoscopic surgery became easier.

There are controversial reports on the use of chest tubes. Some studies reported good results with chest tube and prophylactic placement,^20,21 whereas others suggested that small residual CO₂ pneumothorax could disappear spontaneously and that chest tubes cause intrathoracic negative pressure that increases the possibility of recurrence.^6,12,18 In this study, chest tubes were not inserted in the previous 10 cases in the early period and postoperative tension pneumothorax occurred in 3 cases. For the remaining cases, chest tubes were placed, and none of the patients encountered postoperative tension pneumothorax and recurrence. From our perspective, intraoperative pulmonary re-expansion through ventilator might cause alveolar rupture. The chest tube could effectively prevent postoperative tension pneumothorax, which might cause postoperative crisis and have no obvious side effects, including recurrence, pain, and infection. Therefore, we suggest that a chest tube should be placed routinely.

Whether a hernial sac should be resected is debatable. Puri et al. reported that the hernial sac should be excised to avoid leaving a space-occupying lesion in the chest, which might increase the recurrent rate. ²² However, protection of the phrenic nerve in the process of resection is difficult. Therefore, researchers have reported that resection of the hernial sac is not mandatory. ¹² In our group, a hernial sac was found in 13 patients, and not all of them were resected. These patients had no postoperative recurrence or other complications. It was not necessary to resect the hernial sac based on our experience. The intraoperative conditions of these cases showed that the hernial sac was helpful in the reduction of the hernia contents and facilitated in pushing them back into the abdominal cavity when muscle relaxants and CO₂ pneumothorax were given. The diaphragmatic defect could be sutured directly, and damage of the phrenic nerve was effectively avoided.

Thoracoscopic surgery has advantages in the treatment of associated malformations in the thorax, but it is impossible to check for mesenteric abnormalities of the bowel. Malrotation was the most common malformation associated with late-presenting CDH. ²³ Baerg et al. reported that it was likely to misdiagnose diseases such as appendicitis after reducing the bowel with malrotation. ²⁴ However, the excision of the appendix in thorax might increase the possibility of intrathoracic contamination and bleeding. In our study, eight appendixes herniated into the thorax and were resected simultaneously. The procedures were the same as those involved in laparoscopic appendectomy and the time was short. No complications occurred in the following short period.

The present data suggest that thoracoscopic repair of late-presenting CDH cases is safe and feasible and has a low recurrence rate after mastering operative skills and details.