Endoscopic septum division of tubular esophageal duplication in two children and systematic review.

Tubular esophageal duplication is a rare congenital malformation The surgical treatment of this lesion can be challenging. We aimed to present our experience in two cases with tubular esophageal duplication. Both cases were endoscopically treated by a dual knife. The first patient underwent a single, while the second patient required three sessions of endoscopic septum division using an electrosurgical knife. Both showed satisfactory clinical, radiological and endoscopic response to treatment. Moreover, a systematic literature review has been performed. To identify all available studies, a detailed search on tubular esophageal duplication was performed according to the PRISMA (Preferred Reporting Items for Systematic reviews and Meta-Analyses) guidelines. Tubular esophageal duplication was reported in 16 pediatric cases in 14 studies in the literature. Surgery was applied to 10 of these cases and endoscopic treatment was applied to two of them. In the follow-up, nine patients who underwent surgical treatment and two patients who underwent endoscopic treatment were uneventful. Tubular esophageal duplications can be successfully treated endoscopically. Endoscopic septum division is a minimally invasive procedure that allows full anatomical recovery and satisfactory therapeutic response.

Multiple Esophageal Duplication Cysts in a Toddler: A Rare Scenario.

The foregut duplication cysts are rare congenital malformation and are usually "solitary" midline noncommunicating mediastinal structures. We present a 2-year-old asymptomatic boy having multiple noncommunicating foregut cysts in the neck as well as the thorax.

[Complete and inverted esophagastric duplicity]. / Duplicidad esofagogástrica completa e invertida.

CASE REPORT: A 4-month old girl affected by gastric duplication discovered as a palpable abdominal mass displacing the spleen and left kidney. During laparotomy, a yellowish secretion is aspirated and the gastric duplication is excised together with the common muscular Wall of the greater curvature of the stomach. Also excised, a tubular structure starting from the inferior pole of the malformation, and communicating with it, passing superior to the head of the pancreas and the second portion of the duodenum, followed by a posterior course before inserting into the right cruz of the diaphragm. The histology of the tubular formation was compatible with esophagus. CONCLUSION: This type of duplication has not been reported in the literature and reinforces the theory that these malformations could be secondary to genetic transcription errors, in the differentiation of the endoderm of the primitive digestive tract and the notochord.

CASO CLINICO: Niña de 4 meses, afecta de una duplicidad gástrica, descubierta como masa abdominal palpable y que desplazaba el bazo y el riñón izquierdo. Mediante laparotomía, se observa una duplicidad gástrica y, tras aspirar una secreción amarillenta, se realiza su exéresis junto con la pared muscular común con la curvatura mayor del estómago y una estructura tubular que, partiendo del polo inferior de la malformación y comunicada con ella, pasaba por encima de la cabeza del páncreas y de la segunda porción duodenal, siguiendo luego un curso posterior hasta insertarse en la base del pilar diafragmático derecho. La histología de esta formación tubular fue compatible con el esófago. CONCLUSIONES: Este tipo de duplicidad no la hemos visto referida en la literatura y refuerza la teoría de que estas malformaciones pudieran ser secundarias a errores de transcripción genéticas de información en la diferenciación del endodermo del tubo digestivo primitivo y de la notocorda.

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.

A rare cause of wheezing in an infant: Esophageal duplication cyst.

Esophageal duplication cyst (EDC) is classified as a subgroup of foregut duplication cyst. They are very rare and predominantly detected in children. We present an unusual cause of wheezing in a 2-month-old infant. The diagnosis of EDC was suspected by bronchoscopy, provisionally confirmed by magnetic resonance imaging, and followed by successful surgical excision of the cyst. We conclude that foregut duplication cyst of the esophagus is very rare, and must be considered in the differential diagnosis of persistent wheezing in infants who do not respond to conventional treatment.

Simultaneous occurrence of extralobar pulmonary sequestration, esophageal duplication, and bronchogenic cysts in a Chinese child: a rare case report.

The occurrence of simultaneous extralobar pulmonary sequestration, esophageal duplication, and bronchogenic cysts is relatively low. We report the case of a 9-month-old Chinese child who had a right lung cyst, detected in utero and was closely monitored until birth. At age 9 months, contrast-enhanced computed tomography revealed right mediastinal extralobar pulmonary sequestration and two cysts. The patient did not exhibit any abnormalities. However, the parents were concerned about the disease. Following positive psychological counseling to the parents, surgery was the strong desire. Subsequently, successful thoracoscopic surgery was performed, excising the three lesions. No postoperative complications occurred. Postoperative pathology confirmed extralobar pulmonary sequestration syndrome combined with esophageal duplication and bronchogenic cysts. The patient was followed-up at 1 and 12 months postoperatively and recovered well with no abnormal space occupation. In such cases, preoperative imaging examinations should be carefully performed, and intraoperative exploration should correspond to that before surgery to avoid lesion omission.

An Uncommon Cause of Recurrent Dysphagia and Chest Pain in an Adolescent Boy.

An 11-year-old boy was brought to the emergency department with a week-long history of widespread pain in his upper abdomen that worsened with deep breathing and eating, sialorrhea, food impaction sensation, and a recent fever. Ten months prior, he had similar symptoms and was diagnosed with a pharyngeal phlegmon. He was treated with antibiotics and dexamethasone. In the current episode, he presented with mild elevation of inflammatory markers, a slight deviation of the trachea on chest X-ray, and a tubular esophageal duplication was identified on a thoracic CT, with its opening observed during the endoscopic study. The patient was admitted for further treatment with fluids, analgesia, and antibiotics, and showed improvement over the next seven days with no significant incidents. Esophageal duplications are a rare congenital anomaly and their exact cause is unknown. Typically found in the posterior mediastinum and lower esophagus, they can cause symptoms such as pain, dysphagia, regurgitation, and malnutrition. Surgical or endoscopic resection can be a treatment option for these malformations.

Severe dysphagia due to an esophageal duplication cyst in sixth decade, unusual presentation of a rare pathology.

BACKGROUND: Esophageal duplication cysts are rare congenital tumors usually diagnosed and treated during childhood. Most of them are located in the mediastinum and appear as a mass besides the esophagus. Unfortunately, symptoms are non-specific and depend on the size and location of the mass; therefore, they can easily be missed. If symptoms appear, surgical resection is necessary to prevent troublesome complications. CASE PRESENTATION: We present the case of a 60-year-old woman who presented with severe progressive dysphagia and epigastric pain. After further evaluation, a paraesophageal cystic mass was found, and surgery was required. Non-communicating esophageal duplication cyst was the final diagnosis. CONCLUSION: Esophageal duplication cysts are a rare pathology in adults; their symptoms will vary depending on their size and location. Preoperative diagnosis is difficult as symptoms are non-specific and can be missed. If severe dysphagia, pain, or any other complication appears, surgery should not be delayed.

Reconstruction of Adult Tubular Esophageal Duplication with Supraclavicular Artery Island Flap: A Rare Case Presentation.

Adult esophageal duplication (ED) is a rare congenital anomaly that is rarely encountered in clinical practice. There have been only a few reported cases of adult tubular esophageal duplication. A patient presented with symptoms of odynophagia and dysphagia. Upon examination, gastroscopy and X-ray contrast imaging revealed the formation of a fistula in the upper esophagus that connected to a sinus tract running along the esophagus. After managing the initial infection, an open surgery was performed. The esophageal tubular duplication was removed and the defect was reconstructed using a supraclavicular artery island (SAI) flap. The post-operative recovery was uneventful and the patient's odynophagia and dysphagia were relieved. In conclusion, ED can be effectively diagnosed through esophagogram and gastroscopy. Surgical excision is currently the preferred treatment option, and the use of the SAI flap technique shows great promise in reconstructing the esophageal defect after surgery.

A case of double cystic esophageal duplication in VACTERL syndrome: the first case report and a review of the literature.

Background: An esophageal duplication cyst (EDC) is a rare malformation resulting from the embryonic foregut. VACTERL syndrome is a genetic disorder affecting many systems of the human body. We report the first case of VACTERL syndrome associated to asymptomatic double EDC. Case report: A girl with anorectal malformation and rectovestibular fistula, kidney malformation, and various vertebral defects came to our attention at the time of birth. VACTERL disease was diagnosed. She underwent Peña anoplasty at 4 months of life without complications. MRI was conducted at the age of 2. It accidentally showed a double esophageal duplication (12 mm × 35 mm × 10 mm) at the D7-D9 level. We planned a thoracoscopy; previous intraoperative esophagogastroduodenoscopy showed an external compression of the native esophagus. Two duplicated esophageal lesions were removed. The patient made an uneventful recovery and was completely asymptomatic at long-term follow-up. Conclusions: VACTERL syndrome is still a not well-defined disease. Based on the current literature, this is the first case of a double esophageal duplication in a patient affected by VACTERL syndrome. According to us, the thoracoscopic approach of esophageal duplications can be followed by experts. Complete surgical excision is possible even if the cyst shares a common muscular wall with the esophagus. For this reason, we suggest to close the muscular wall by a simple interrupted suture.

Tubular Duplication of the Esophagus in a Newborn, Treated by Thoracoscopy.

We present a case of tubular esophageal duplication in a 3-day-old female newborn (38 weeks, 2,500 g) without concomitant abnormal development. Esophageal duplication was diagnosed based on the clinical picture, direct laryngoscopy, esophagography and computed tomography. The duplicated esophagus was resected by thoracoscopy leaving the orthotopic esophagus in place. Isolation from the pharynx was performed via a separate cervical incision. After a follow-up period of 20 months, the child returned to normal growth and development.

Laparoscopic resection of an intra-abdominal esophageal duplication cyst in the ileum: a case report.

BACKGROUND: Esophageal duplication cyst (EDC) is a type of gastrointestinal duplication cyst that involves congenital malformations of the gastrointestinal tract. EDCs are frequently found in the mediastinum and thoracoabdominal region, but rarely occur in the abdominal cavity. However, intra-abdominal EDCs are frequently found in the upper abdomen near the abdominal esophagus. Here, we report, for the first time, a case of intra-abdominal EDC that occurred in the ileum. CASE PRESENTATION: A 14-year-old female patient presented to our hospital with complaints of epigastric pain and vomiting. Abdominal computed tomography (CT) revealed a cystic tumor in the pelvis, suspected of ovarian origin. She was admitted to our gynecology department and underwent emergency surgery. The laparoscopic examination revealed that both ovaries were intact and that a primary tumor had developed from the ileal mesentery. Since the patient's condition was not urgent at the time of the gynecological surgery, the procedure was completed by only performing exploratory laparotomy; the patient was admitted to our department after the surgery. Pelvic magnetic resonance imaging performed on the next day revealed a cystic mass measuring 90 × 65 mm with a smooth margin and homogeneous signal intensity, arising posterior to the uterus. The mass was suspected as an intestinal duplication cyst. On another day, after the examinations were completed, we resected the portion of the small intestine containing the tumor by laparoscopy. The patient had a successful postoperative course and was discharged on the 5th postoperative day. Histological examination showed that the cyst was lined by stratified squamous epithelium, contained esophageal glands, and had a two-layer muscularis propria. Therefore, a diagnosis of intra-abdominal EDC was performed. CONCLUSIONS: An intra-abdominal EDC cyst is relatively rare; this is the first case reported at the distal ileum.

Thoraco-abdominal duplications in children: Two case reports.

INTRODUCTION: Thoraco-abdominal duplication is rare congenital malformations of the notochord that occurs in only 2% of cases of alimentary tract duplications. We report two rare cases of thoraco-abdominal duplication, emphasizing the value of radiological assessment and discussing the place of diagnostic and therapeutic laparoscopy. PRESENTATION OF CASE: It was a 12-year-old girl and an 8-month-old boy, admitted for epigastralgia and dysphagia with respiratory distress respectively. Imaging was in favor of pancreatic duplication with intra-thoracic extension for the first patient and gastro-esophageal duplication for the second. A mass excision was done laparoscopically for the first and by a thoracotomy for the second. The aftermath of the surgery was simple in both cases. CLINICAL DISCUSSION: Thoraco-abdominal duplications are rare congenital malformations that account for only 2% of cases of gastrointestinal duplications. Their diagnosis is difficult since the revealing symptomatology is not common. The treatment is only surgical is facilitated by the laparoscopy which has a diagnostic and therapeutic interest. CONCLUSION: Our case reports focused on the difficulty of the diagnosis that is done by imaging and is confirmed by surgery with anatomopathological examination of the excised mass. Diagnostic and therapeutic minimally invasive approach should be used whenever possible.

Endoscopic Removal of a Cervical Esophageal Duplication Cyst.

A 6-month-old female presented for 2 months of noisy breathing. Flexible laryngoscopy showed limited bilateral vocal fold abduction. Computed tomography revealed a non-enhancing 3.6 × 2.3 × 3.5 cystic prevertebral mass spanning C2-T. Using an endoscopic approach, the overlying mucosa was incised, and the cyst was freed and fully excised from the surrounding mucosa with blunt microlaryngeal instruments without complication. Three months postoperatively she had no respiratory issues and was eating well. Flexible laryngoscopy revealed bilateral vocal fold mobility. We propose that endoscopic removal of a cervical esophageal duplication cyst in selected cases is an alternative to open excision. Laryngoscope, 130:2053-2055, 2020.

Laparoscopic surgery for an esophageal duplication cyst using a near-infrared indocyanine green fluorescence system: A case report.

We herein describe a case of laparoscopic surgery for an esophageal duplication cyst using a near-infrared indocyanine green fluorescence system. A 64-year-old woman with a cystic tumor adjacent to the esophagogastric junction was referred to our hospital for treatment. Esophagogastroduodenoscopy and abdominal CT revealed a 70-mm submucosal tumor derived from the abdominal esophagus. We performed laparoscopic resection and then evaluated the tissue perfusion of the abdominal esophagus by using a near-infrared indocyanine green fluorescence system. A Dor fundoplication was performed to prevent postoperative gastroesophageal reflux disease and reinforce the mucosal layer defect. The postoperative course was uneventful, and pathological evaluation confirmed that the tumor was an esophageal duplication cyst. The patient did not develop recurrence in the 24 months after surgery. We have demonstrated that laparoscopic resection of an esophageal duplication cyst may be performed effectively with intraoperative assessment of tissue perfusion using a near-infrared indocyanine green fluorescence system.

A congenital cystic pulmonary airway malformation occurring together with both an extralobar pulmonary sequestration and an esophageal duplication cyst.

A foregut duplication cyst occurring together with both a congenital cystic pulmonary airway malformation and extralobar pulmonary sequestration is an unusual combination. Prenatal ultrasound, MRI, and postnatal CT are helpful for operative planning. Surgical resection is the definitive management for all three anomalies.

Esophageal duplication and congenital esophageal stenosis.

Esophageal duplication and congenital esophageal stenosis (CES) may represent diseases with common embryologic etiologies, namely, faulty tracheoesophageal separation and differentiation. Here, we will re-enforce definitions for these diseases as well as review their embryology, diagnosis, and treatment.

Tracheal Atresia with Segmental Esophageal Duplication: An Unusual Anatomic Arrangement.

An unusual anatomic configuration of segmental tracheal agenesis/atresia with esophageal duplication on autopsy in a fetus that demised in utero at 29 weeks is reported. The mother was scanned initially for a cardiac anomaly at 20 weeks and on follow-up scan at 27 weeks had polyhydramnios and underwent amnioreduction. The final autopsy diagnosis was vertebral, ano-rectal, cardiac, tracheoesophageal, renal, and limb malformations (VACTERL). We discuss the autopsy findings along with the embryological mechanisms and compare the configuration with Floyd's classification for tracheal agenesis. The difficulties in prenatal diagnosis are discussed.