Summary
Esophageal duplication is a rare congenital esophageal disorder. Surgical excision is the standard treatment for symptomatic esophageal duplication cysts. Traditionally, the resection is accomplished via thoracotomy; however, a minimally invasive approach is possible, avoiding the long hospital stay, the discomfort and the long recovery time due to a thoracotomy. The authors describe two cases of esophageal duplication resected via a left thoracoscopic approach.
Introduction
Esophageal duplication is a rare congenital esophageal anomaly, probably the most frequently found in adults. It is usually located in the lower one third of the esophagus.1 Esophageal duplication cysts can be asymptomatic at the time of diagnosis or can cause dysphagia.1 Surgical excision is the usual treatment due to the risk of complications such as aspiration and bleeding.2 While the traditional approach has been through a thoracotomy, today there is a growing experience with the thoracoscopic treatment of mediastinal masses and primary esophageal motility disorders, such as achalasia and diffuse esophageal spasm.3,4
The authors describe two cases of esophageal duplication resected via a left thoracoscopy approach.
Patient # 1
A 30-year-old woman presented with a 5-year history of dysphagia. The symptom worsened during the last 4 months. Her past medical history was unremarkable. Physical examination was normal. An upper endoscopy showed a submucosal mass in the posterior wall of the lower esophagus. A computerized tomography scan showed a smooth oval mass in the esophageal wall. An endoscopic ultrasound was compatible with an esophageal duplication cyst (Fig. 1).
Patient # 1. Computerized tomography (A), endoscopic ultrasound (B), and esophagram (C). ED = esophageal duplication.
A left thoracoscopic approach was used, with 5 ports. A double lumen endotracheal tube was used to deflate the left lung. The cyst was initially isolated from the pleura and subsequently dissected off the esophagus. It was decompressed and completely resected. No mucosal damage was present. The muscle layers of the esophagus where the cyst was present were approximated using 2–0 silk interrupted stitches.
A barium esophagram was performed on second postoperative day and showed no leaks. Pathological examination findings were of a dual muscular layer and no cartilage or bronchial glands. The inner layer was composed of squamous epithelium.
The patient swallowing status has been normal since the time of operation.
Patient # 2
A 35-year-old man with dysphagia to both liquids and solids for 7 months was referred for surgery. The patient had lost 20 pounds over a 7-month period. His past medical history was unremarkable. Physical examination was normal. An upper endoscopy showed a submucosal lesion in the right wall of the lower esophagus. A computerized tomography scan revealed a cystic mass on the right side of the esophagus. An endoscopic ultrasound was compatible with a cyst (Fig. 2) and aspiration of the cyst was attempted.
Patient # 2. Computerized tomography (A) and endoscopic ultrasound (B). ED = esophageal duplication. Ao = aorta.
The cyst was approached using a left thoracoscopic approach with 4 ports. A double lumen endotracheal tube was used to deflate the left lung. The cyst was isolated from the pleura and adjacent structures but due to its voluminous size and adhesion with the esophagus the cyst was decompressed and the dissection proceeded with the aid of an endoscope inside the esophagus. At the level of the upper edge of the cyst it appeared to be fused to the esophageal mucosa in the point of a previous transluminal aspiration at the time of endoscopic ultrasound. The cyst was resected. A 1-cm mucosal perforation was noted and repaired with a running suture of 3–0 polyglactin. The muscle layers over the area where the cyst was present were approximated.
The postoperative course was uneventful. A barium esophagram was performed on second day and showed no leaks. Pathological examination findings were a dual muscular layer and no cartilage or bronchial glands. The inner layer was composed of squamous epithelium.
Four months after the operation, the patient started experiencing heartburn. Ambulatory pH monitoring confirmed the presence of gastroesophageal reflux disease (DeMeester score = 118, normal < 14.8). This was attributed to the extensive dissection of the esophagogastric junction at the time of the first operation. Because of the absence of peristalsis in the distal esophagus, a laparoscopic partial fundoplication (180° anterior Dor fundoplication) was performed. The patient has been asymptomatic since then.
Discussion
The first description of esophageal duplication is credit to Blasius, in 1711.5 Since then, less than 100 cases of esophageal duplication have been reported. It accounts for only 3% of the mediastinal masses6 and it is the second most common duplication of the digestive system5 after jejuno-ileal duplications.
Dysphagia is the chief complain in symptomatic cases.5 Several imaging techniques have been used in the diagnosis of esophageal duplication, as computerized tomography, magnetic resonance, and barium examination.7 Endoscopic ultrasound is considered today the most sensitive method.1,8 A preoperative biopsy of the lesion should not be performed as it can complicate the resection of the cyst1 due to adhesions between the cyst wall and the esophageal mucosa, as it was the case in our second patient. The pathological diagnosis of an esophageal duplication requires the presence of a cyst adjacent to the esophagus, covered by two muscle layers and lined with a squamous, columnar, cuboid, pseudostratified, or ciliated epithelium.1,8
Resection of the duplication cyst is the standard treatment. Even though esophageal duplication cysts are often asymptomatic at the time of diagnosis1 surgical excision is advised because definitive diagnosis is better done on the surgical specimen and most patients with untreated esophageal duplication will experience dysphagia or develop complications such as bleeding or aspiration.1,2 Malignant degeneration of esophageal duplication is a very rare event.5,6,9 Traditionally, the resection is accomplished via a thoracotomy; however, there are obvious advantages associated to the minimally invasive approach, such as a short hospital stay, minimal post operative discomfort, and fast recovery and return to regular activities.1,10,11 Laparoscopic resection of abdominal and thoracoabdominal cysts have been reported with excellent results as well.8,12–14 The surgical technique must emphasize evaluation of the integrity of the esophageal mucosa after resection of the cyst, approximation of the muscle edges over the area where the cyst was present in order to avoid a pseudodiverticulum, and identification of vagi nerves. Transillumination through an esophagoscope and aspiration of the cyst can be sometimes useful. Partial resection of the cyst can be done in difficult cases. In our two patients, the thoracospic approach provided the perfect exposure and the possibility to execute the technical steps mentioned above. We feel that in both patients we could have been able to resect the cyst laparoscopically but it would have been very difficult to re-approximate the muscle edges from that approach.
In summary, our report shows that the thoracoscopic approach is safe and effective for esophageal duplication cysts. It avoids the discomfort associated to a thoracotomy and it allows good visualization for the removal of the cyst.
Acknowledgments
Dr Fernando Herbella was supported by Coordenacao de Aperfeicoamento de Pessoal de Nivel Superior – Brazil.
