Esophageal duplication cysts and closure of the muscle layer.

BACKGROUND: Foregut duplication cysts are rare congenital anomalies that require surgical intervention with approximately 10%-15% of all gastrointestinal duplication cysts originating from the esophagus. Consensus is lacking among surgeons regarding closure of the esophageal muscle layer after resection of an esophageal duplication cyst and long-term outcomes are poorly documented. Therefore, we conducted the first study comparing complication rates in patients undergoing closure versus nonclosure of the esophageal muscle layer after esophageal duplication cyst resection. MATERIALS AND METHODS: A retrospective cohort study at Boston Children's Hospital, Massachusetts General Hospital, Brigham and Women's Hospital, and the Floating Hospital for Children at Tufts Medical Center was conducted. Patients undergoing resection of esophageal duplication cysts between 1990 and 2012 were classified according to whether the esophageal muscle layer was closed or left open. Demographic data, surgical technique, preoperative symptoms, and both short-term (<30 d) and long-term (≥30 d) complication rates were abstracted from patient medical records. RESULTS: Twenty-five patients were identified with a median age of 15-y old (range, 2 mo to 68-y old) and an average follow-up of 1 y. Eleven patients had the esophageal muscle layer closed after surgical resection (44%). Of those 11 patients, one developed a short-term complication, dysphagia (9%, 95% CI: 2%, 38%). Only one patient returned to the operating room, after 30 d, for an upper endoscopy after developing symptoms of gastroesophageal reflux disease. Of the 14 patients who had their muscle layer left open, three patients (21%, 95% CI: 8%, 48%) developed short-term complications, two of whom required surgical intervention within 30 d. Furthermore, two additional patients required surgical intervention after 30 d for a long-term complication (diverticulum and cyst recurrence). CONCLUSIONS: Surgical complications occurred more frequently in patients who had the muscle layer left open after resection of an esophageal duplication cyst. In addition, most patients requiring reoperation for both short-term and long-term complications occurred in this group. Though small, this study is the first to evaluate the complications after resecting esophageal duplication cysts. Our results suggest that closing the esophageal muscle layer after removal of an esophageal duplication cyst may be indicated to prevent both complications and the need for reoperations.

Oesophageal duplication cysts, a rare cause of neck lump treated by ultrasound guided drainage: case report and review of the literature.

Oesophageal duplication cysts are a rare congenital anomaly of the foregut which usually present in infancy with respiratory symptoms, recurrent pneumonia and feeding difficulty. Other presenting symptoms depend on the location of the cyst and can include dysphagia, chest pain, arrhythmias and features of mediastinal compression. Treatment is usually surgical resection, recommended for complete resolution of symptoms, histological diagnosis and exclusion of malignancy. Here, we present a case of infected oesophageal duplication cyst which presents as a neck lump in a 43-year-old female with a background of Goltz syndrome, azygos fissure and congenital aortic stenosis. Surgical resection was decided against owing to the patient's high risk secondary to co-morbidities and instead ultrasound guided drainage was carried out successfully. The patient was symptom free and well at 1-year follow-up. Oesophageal duplication is an unusual presentation of a neck lump in an adult and whilst the usual treatment is surgical resection, we present here a case treated in an entirely different manner.

Successful robotic-assisted resection of an esophageal duplication cyst in the upper thoracic esophagus: A case report with surgical video.

Esophageal duplication cysts are rare congenital noncancerous growths. Symptoms of this disease are reported to be asymptomatic in approximately 70% but include respiratory symptoms such as coughing and difficulty breathing. Minimally invasive removal of these cysts without esophagectomy is typically recommended. However, when the cyst is situated in the upper mediastinum, surgical excision becomes technically challenging. Here, we report a case of an obese female patient with esophageal duplication cyst in the upper mediastinum who underwent successfully robotic-assisted complete removal of the cyst. A 50-year-old woman presented to a local clinic with a persistent cough and hoarseness lasting 4 months. A computed tomography scan revealed a large cystic tumor in the upper mediastinum, causing displacement of the trachea. The resection of the cystic tumor was safely performed with robotic assistance. The use of robotic system for the removal of esophageal duplication cyst is technically safe and feasible.

[Congenital esophageal duplication cysts: progressive dysphagia in adulthood]. / Kongenitale zystische Duplikatur des Ösophagus: Progrediente Dysphagie im Erwachsenenalter.

Esophageal duplications are congenital abnormalities of the foregut. We present the case of a 33-year-old woman suffering from progressive dysphagia who had surgery for esophageal duplication. The following three criteria define the cystic lesion: an intimate attachment to the esophageal wall, the presence of a smooth muscle coat and a mucosal lining consisting of squamous and/or ciliated respiratory epithelium. Diverticula, bronchogenic cysts and cystic neoplasms have to be considered in the differential diagnosis. Congenital cystic esophageal duplication is a rare cause of dysphagia in adulthood.

Oesophageal duplication cyst presenting as haemoptysis.

Duplications of the alimentary tract include a variety of cysts, diverticula, and tubular malformations, all believed to have embryological origin. The cysts are most commonly found in children, and the diagnosis is made in infancy in the majority of patients. We report a case of a two-and-a-half year old child, presenting with the history of repeated episodes of haematemesis. Upper GI endoscopy was unremarkable and the chest x-ray showed no pathology. Computed tomography (CT) angiogram revealed soft tissue density lesion in the right chest at the level of T6. Right thoracotomy suggested a cystic mass close to the oesophagus which was shown on histopathology to be lined with gastric mucosa consistent with oesophageal duplication cyst. To the best of our knowledge, this is the first case of its kind reported from Pakistan.

Acute onset of esophageal duplication cyst in adult. Case report.

INTRODUCTION: Esophageal duplication (ED) cyst is unusual congenital disorder of the foregut, accounting for 10% to 15% of duplications of all foregut cysts. We report a case of esophageal duplication with acute clinical presentation, treated successfully with surgical resection. CASE REPORT: 46-year-old man with acute dysphagia and gastroesophageal reflux of 2 weeks duration, showed submucosal bulging mass in the posterior wall of the middle third of oesophagus, fluid-filled cystic structure, dyshomogeneous, low-attenuation mass with smooth borders compatible with an hemorrhagic esophageal duplication cyst. With the suspect of acute presentation of a complicated esophageal cyst, the patient underwent surgery by right posterolateral thoracotomy. A gastrografin esophagogram was performed on third postoperative day and showed no leaks. DISCUSSION: Up to 80% of the esophageal duplication cysts are diagnosed in childhood and the majority of young patients develop symptoms, while symptomatic cyst into adult life is very rare (<7%). Acute onset is generally due to complications, i.e. intracystic hemorrhage, perforation, and infection, especially of the cyst with esophageal communication. Complete surgical excision by thoracotomy or thoracoscopy is the therapy of choice even if the patient is asymptomatic because of the risk of complications such as aspiration and bleeding. CONCLUSIONS: The diagnosis and treatment of esophageal cysts is still evolving. The pathological diagnosis of oesophageal duplication requires the presence of the Ladd and Gross criteria. The infrequent acute onset doesn't modify the management and the surgical resection is the procedure of choice. The future of the treatment of esophageal cysts lies in the advancement of minimally invasive operative techniques.

Intra-abdominal esophageal duplication cysts: a review.

Esophageal duplication cysts (EDCs) are well described within the literature, normally occurring within the mediastinum. Intra-abdominal EDCs are rare and typically occur near the intra-abdominal esophagus. Herein, we describe two cases of intra-abdominal EDCs: a 60-year-old man who was incidentally found to have a retro-duodenal cystic mass and a 50-year-old woman with a cystic lesion near the body and tail of her pancreas causing left flank pain. Both patients underwent enucleation of their respective masses. Pathology revealed ciliated pseudostratified columnar epithelium with scattered mucus-secreting cells and two smooth muscle layers in the cyst wall of both patients, consistent with EDCs. Although intra-abdominal EDCs have been reported in the literature, our two cases and a review of the literature indicate that these lesions are not always adherent to or even near the intra-abdominal esophagus.

Mediastinal foregut duplication cyst presenting as a rare cause of breathing difficulties in an adult.

Foregut duplication cysts are rare congenital anomalies of enteric origin. The diagnosis is usually made in infancy. We report the unusual case of a 71-year-old female presenting to the ENT department with shortness of breath and stridor due to an oesophageal reduplication cyst. The presentation, diagnosis and management of this potential pitfall for the unwary are outlined.

Esophageal duplication cyst with hemivertebrae: A case report and literature review.

BACKGROUND: Esophageal duplication cysts (EDCs) are rare congenital anomalies that can be associated with symptomatic spinal abnormalities, but presentations due to EDC symptoms are rarely found in the presence of spinal abnormalities. CASE SUMMARY: A 6-month-old infant weighing approximately 5.0 kg presented with a 2-month pulmonary infection and more recent difficulty swallowing and nutritional intolerance that did not improve with medical treatment. Contrast-enhanced chest computed tomography showed a well-defined, mediastinal, homogeneous, low-density cystic mass of 11.9 × 5.5 × 5.1 cm, compressing the liver and bending the trachea forward. Hemivertebrae were present (T4 and T3). Diagnostic laparoscopy was performed, but was converted to open surgery. After ensuring that the cyst was not within the abdominal cavity, thoracotomy was performed, and the cyst was completely resected. Pathophysiological examination revealed an EDC. The patient recovered well, without symptoms 6 months later. CONCLUSIONS: Overall, noninvasive imaging and diagnostic procedures may not be sufficient to define the exact location of an EDC. Although hemivertebrae were present, they were asymptomatic and did not require treatment; only the EDC induced nonspecific symptoms that disappeared after surgery.

Two cases of thoracoscopic resection of esophageal duplication in children.

We report two cases of thoracoscopic resection of esophageal duplication cysts. Both patients underwent successful thoracoscopic excision. They were discharged on postoperative day 2 and 4, respectively. They made uneventful recoveries and were completely asymptomatic at 1-month followup. One child was lost to long-term follow-up. In the other child, barium swallow study 10 months after surgery demonstrated a pseudodiverticulum at the site of cyst excision. Thoracoscopic resection of esophageal duplications is safe. Complete excision is possible even if the cyst shares a common muscular wall with the esophagus. Pseudodiverticulum may develop at the site of excision: follow- up is necessary and consideration should be given to closure of the muscular defect at the time of excision. To help avoid esophageal injury and, should it occur, recognize esophageal perforation, we recommend performing the dissection under intraesophageal endoscopic supervision.

Distal Esophageal Duplication Cyst with Gastro-Esophageal Reflux Disease: A Rare Association and a Management Challenge.

Esophageal duplication cysts are rare congenital abnormalities of the foregut and may be associated with other conditions. Association of esophageal duplication with Gastro-Esophageal Reflux Disease (GERD) has not been reported in children. We are reporting a case of a 16 months baby who had antenatal diagnosis of diaphragmatic hernia. Postnatal CTchest, however, suggested a distal esophageal duplication cyst and a contrast esophagogram showed grade-IV GER. A thoracoscopy in another hospital excluded esophageal duplication at that time. Later, he presented with hematemesis in our department and was re-evaluated. Repeat CTconfirmed a persistent 2.5 x 1.3 cm cyst in distal esophagus. Upper GI endoscopy suggested grade-II esophagitis with a wide patent gastro-esophageal junction. The child was treated with left thoracotomy, excision of the duplication cyst and thoracic fundoplication. He had an uneventful post-operative recovery and is doing well at 6 months follow-up.

Oesophageal pseudodiverticulum after foregut duplication cyst excision: Case report and literature review.

Oesophageal pseudodiverticula rarely occur after excision of benign oesophageal neoplasms. While management and outcomes have been reported in the adult leiomyoma literature, sparse data exist on the occurrence and management of pseudodiverticula after foregut duplication cyst excision. We discuss our experience with a paediatric patient and review relevant literature regarding operative techniques and surgical outcomes.

Esophageal duplication cyst: an unusual cause of respiratory distress in infants.

Esophageal duplication cysts are rare congenital anomalies. These should be recognized as a cause of respiratory distress in infants and are usually associated with feeding difficulties. We report 2 such cases of esophageal duplication cyst in infants, causing acute respiratory distress. Excision was curative.

Esophagectomy for a rapidly progressing esophageal duplication cyst.

Esophageal duplication cysts are rare and frequently asymptomatic anomalies of the adult gastrointestinal tract that are often misdiagnosed in clinical practice. Identifying the unique features of esophageal duplication cysts is therefore important. We report a unique case of esophageal duplication cyst in a 52-year-old woman with rapidly progressing chest pain and dysphagia. The cyst was found to share, in part, a remarkably inflammatory and edematous inner lining with the esophagus. Enucleation was not feasible, and therefore, esophagectomy was performed. The only long-term side effect that occurred after 44 months of follow-up examinations was slight acid reflux esophagitis.

Congenital esophageal cysts in adults.

Sixteen adult patients with congenital esophageal cysts were operated on between 1957 and 1979. Preoperatively, 7 patients (44%) were asymptomatic and the cyst was found incidentally on a routine chest roentgenogram. Esophageal symptoms were present in only 3 patients (19%), whereas most symptomatic patients had precordial sensations or arrhythmias. A correct preoperative diagnosis was made in only 1 patient. After enucleation of the cyst, preoperative symptoms were alleviated in all patients and short-term results were excellent. However, long-term follow-up 13.2 +/- 5.6 (+/- standard deviation) years later revealed moderate reflux in 9 (64%) of the surviving 14 patients. During esophagoscopy, macroscopic esophagitis was found in 12 (92%) of 13 patients. On histological examination of specimens obtained by forceps biopsy, esophagitis was present in 10 (77%) of 13 patients and included Barrett esophagus in 2. We conclude that, despite excellent early results, long-term follow-up of patients who undergo operation for congenital esophageal cysts is indicated because of the increased incidence of reflux esophagitis.

Congenital cystic adenomatoid malformation complicated by esophageal duplication cyst in a 6-month-old girl.

We report a case of congenital cystic adenomatoid malformation (CCAM) complicated by an esophageal duplication cyst in a 6-month-old girl. The patient presented with recurrent pneumonia. Magnetic resonance imaging revealed two cystic lesions in the upper and lower lobes of the right lung. After cystectomy, histopathological investigation revealed that the lower cyst was a CCAM Type I, and the upper cyst was an esophageal duplication cyst. The coexistence of these complex anomalies supports the concept that the esophageal duplication cyst is one entity of a broad spectrum of developmental abnormalities caused by abnormal budding of the primitive foregut.