A 72-year-old woman with a history of breast cancer presented with 3 months of progressive dysphagia that increased to total dysphagia to solids. The patient denied any symptoms of reflux or anorexia but noted a 5.44 kg (12 lb) weight loss. Chest CT and magnetic resonance imaging (MRI) demonstrated diffuse wall thickening of the mid to distal portion of the esophagus, with proximal dilation of the esophagus and contained an air-fluid level. An EGD was performed, and she was found to have a
Discussion
Duplication cysts are rare congenital anomalies of the foregut that were first reported in 1711 by Blassium.5 The majority occur in the ileum (35%) and the esophagus (19%), with a male predominance.1, 6 Esophageal duplication cysts are thought to develop because of a failure of vacuolization of a solid esophagus during embryologic development. Esophageal duplication cysts may occur separate from the esophagus, in continuity with the esophagus, or within the esophageal wall. In the current case,
The management of small-bowel duplication cysts remains somewhat controversial because there may be an increased risk of malignant transformation.99,100 Successful endoscopic management of symptomatic duplication cysts has been reported.101-105 There are no data to support surveillance of these patients.
They can be associated with other congenital anomalies, such as small intestinal duplication, esophageal atresia distal to the duplication, tracheoesophageal fistulas, and spinal abnormalities, including scoliosis, hemi vertebrae, and fusion [11,14]. The endoscopic resection of esophageal duplication cysts has increased in adult surgery in the last years because of its minimal invasiveness [12,13]. There are two cases of duplication cysts reported, which were resected only by endoscopy in adults.
Duplications of the gastrointestinal tract are rare malformations with an incidence of one in 4500 [1]–12500 [2]. Esophageal duplication cysts represent about 10% of all foregut duplications and are classified as cystic or tubular type duplications [3]. The tubular type esophageal duplication cyst is a rare subgroup of the esophageal duplication cysts seen in only 5%–10% of all cases [4]. We report about a 5-year- old boy with such a tubular type esophageal duplication cyst presenting with dysphagia and malnutrition. To our knowledge this is the first report about an endoscopic resection of an intraluminal bridge using an insulated scissors- type knife SB knife (SUMITOMO BAKELITE CO., Tokyo, Japan). The postoperative course was uneventful and the patient could be discharged only one day after surgery. The follow-up endoscopy three month later revealed no evidence of residuals of the duplication or remarkable stricture. We conclude that the endoscopic resection of tubular type esophageal duplication cysts is a save and preferable therapeutic option also in children.
In 2005, Will et al8 performed an endoscopic fenestration (diameter of 1 cm) and, after 6 weeks, a partial resection of the anterior wall of an esophageal duplication cyst, resulting in a permanent 4 cm opening. In 2006, Joyce et al6 described the complete endoscopic resection of an esophageal duplication cyst appearing as a submucosal mass with a diameter of 1 cm and a pedicle. In conclusion, this is the first report of a complete endoscopic management of a tubular esophageal duplication.
Esophageal duplication is a rare congenital lesion. Surgery is the standard treatment. Tubular duplication is extremely rare, with esophageal communication in very few cases.
The aim of this study was to document the feasibility of the endoscopic management of tubular esophageal duplication.
Case report.
A 14-year-old boy presented with acute dysphagia and acute retrosternal pain. Based on his radiographic and endoscopic findings, a cystic esophageal duplication with an upper esophageal stricture was initially suspected. A laparoscopic gastrostomy was performed. A cyst resection through right thoracoscopy assisted by flexible endoscopy was decided upon. When no extraluminal cystic duplication was found, a tubular duplication was considered and the procedure was abandoned. An endoscopic treatment was performed. A standard endoscope was inserted through an upper esophageal stricture. Two lumens were identified 25 cm from the incisors. A pediatric endoscope was passed through the main one, revealing a thick intraluminal bridge. By using a guidewire, the endoscope's passage into the narrow lumen revealed a distal communication with the esophagus. With the guidewire left in place, the endoscope was reintroduced into the main lumen. A lengthwise incision of the bridge was performed by using a needle knife. At the end of the procedure, an esophageal dilation was performed. Histology confirmed the diagnosis of duplication.
The endoscopic incision of the duplication was completed uneventfully. For 11 months, the patient followed a normal diet and experienced no symptoms.
Single case.
To our knowledge, this is the first report of successful endoscopic incision of a total tubular esophageal duplication.
Management of symptomatic cases has typically been surgical, although endoscopic treatment has become a promising approach. Endoscopic excision with complete resolution of symptoms has been reported.9-12 With the development of flexible endoscopic techniques, marsupialization of the cyst cavity is now plausible.13
