Foregut Duplication Research Articles

Foregut Duplication Cyst Mimicking Pulmonary Tuberculosis: A Rare Presentation

Foregut duplication cyst is a rare congenital anomaly in children. Most of the children are asymptomatic and are diagnosed incidentally when evaluated for other problems. Complications include infection, rupture, and malignant transformation. Here, we present a case of complicated foregut duplication cyst in a 10-year-old female which mimicked tubercular spondylitis on imaging.

Two’s Company: Multiple Thoracic Lesions on Prenatal US and MRI

Background: Congenital pulmonary airway malformations (CPAM), bronchopulmonary sequestrations (BPS), and CPAM-BPS hybrid lesions are most commonly solitary; however, >1 lung congenital lung lesion may occur. Objectives: To assess the frequency of multiple congenital thoracic anomalies at a high-volume referral center; determine prenatal ultrasound (US) and magnetic resonance imaging (MRI) features of these multifocal congenital lung lesions that may allow prenatal detection; and determine the most common distribution or site of origin. Methods: Database searches were performed from August 2008 to May 2019 for prenatally evaluated cases that had a final postnatal surgical diagnosis of >1 congenital lung lesion or a lung lesion associated with foregut duplication cyst (FDC). Lesion location, size, echotexture, and signal characteristics were assessed on prenatal imaging and correlated with postnatal computed tomographic angiography and surgical pathology. ­Results: Of 539 neonates that underwent surgery for a thoracic lesion, 35 (6.5%) had >1 thoracic abnormality. Multiple discrete lung lesions were present in 19 cases, and a lung lesion associated with an FDC was present in 16. Multifocal lung lesions were bilateral in 3 cases; unilateral, multilobar in 12; and, unilobar multisegmental in 4. Median total CPAM volume/head circumference ratio for multifocal lung lesions on US was 0.66 (range, 0.16–1.80). Prenatal recognition of multifocal lung lesions occurred in 7/19 cases (36.8%). Lesion combinations were CPAM-CPAM in 10 cases, CPAM-BPS in 5, CPAM-hybrid in 2, hybrid-hybrid in 1, and hybrid-BPS in 1. Of 5 unilateral, multifocal lung lesions, multifocality was prenatally established through identification of a band of normal intervening lung or intrinsic differences in lesion imaging features. Conclusions: Although less common, multiple thoracic abnormalities can be detected prenatally. Of multifocal lung lesions, the most common combination was CPAM-CPAM, with a unilateral, multilobar distribution. Prenatal recognition is important for pregnancy counseling and postnatal surgical management.

Small bowel obstruction caused by primary adenocarcinoma arising from an ileal tubular type foregut duplication cyst

Small bowel obstruction is a surgical emergency commonly caused by adhesions, hernia, intussusception and volvulus. Less often, it can be caused by neoplasms. Small intestine adenocarcinoma is an uncommon malignancy, while its occurrence from foregut duplication cyst is extremely rare. Herein we reported a case of complete small bowel obstruction caused by ileal adenocarcinoma arising from a tubular-type foregut duplication cyst. A 49-year-old man with history of inguinal hernia presented with vomiting, abdominal pain and distention, and constipation for 3 days. Abdominal computed tomography (CT) scan showed high-grade distal small bowel obstruction with a transition point in the terminal ileum near the ileocecal valve. Emergency exploratory laparotomy revealed a 4 × 5 cm circumferential mass at the terminal ileum. Right hemicolectomy was performed. Gross examination demonstrated a 2.5 cm long circumferential ileal stricture. Histologically, the stricture contained moderately to poorly differentiated invasive adenocarcinoma arising from ectopic foregut duplication cysts lined by mixed cuboidal and ciliated respiratory epithelial cells with focal low- and high-grade dysplasia. Peritoneal implants developed four months post surgery despite post-operative chemotherapy. Foregut duplication cyst and its derivative neoplasm should be taken into consideration in encountering adult patients with small bowel obstruction.

Respiratory epithelium in an enteric duplication cyst at the ileocecal valve

Abstract Although a duplication cyst of the alimentary tract is rare, it can occur anywhere in the alimentary tract. The inner epithelial lining of a duplication cyst mostly consists of the gastrointestinal epithelium. However, studies have reported that foregut duplication cysts can have a respiratory epithelium lining. It is rare, however, that respiratory epithelium covers midgut duplication cysts. We herein report an enteral duplication lined with respiratory epithelium, occurring in an 18-month-old girl. Our study highlights the need for more studies about the embryonic development of duplication cysts are needed.

Microforceps in the Diagnosis of Pancreatic Bronchogenic Cyst Under Endoscopic Ultrasound Guidance

ABSTRACTBronchogenic cysts belong in the category of foregut duplication cysts, along with esophageal and neuroenteric duplication cysts. They are often found incidentally on cross-sectional imaging. Although they are benign, patients often undergo surgical resection because of diagnostic uncertainties. We report the first case of bronchogenic cyst diagnosed with the microforceps, after previous nondiagnostic fine-needle aspirations, thus allowing the patient to avoid surgery.

Thoracoscopic Resection of an Esophageal Lobe in a Neonate

Abstract Introduction: Thoracoscopy has been well established for resections of lung lesions, foregut duplication cysts, and tumors, as well as for repair of esophageal atresia anomalies. This appr...

Foregut duplications in the superior mediastinum: beware of a common wall with the tracheo-bronchial tree.

Foregut duplication cysts (FD) with a common muscular wall with the oesophagus are well recognised. Our case series highlights the existence of a common wall between superior mediastinal FD and the tracheo-bronchial tree. Over the last 15 years, we have thoracoscopically resected 41 FD. Five cases were identified to have a common wall with the tracheo-bronchial tree at operation. The clinical history, radiology, findings at operation and pathology were evaluated to highlight learning points. Five superior mediastinal cysts with a common wall were identified, with two antenatally and three postnatally diagnosed. All three postnatal cases and one antenatal case presented with symptoms of respiratory compromise and stridor. Only one neonate was relatively asymptomatic before resection. CT similarities in all cases were: separation of trachea and oesophagus by the cyst, oesophageal deviation to the right or compression and compression of trachea/bronchus. Thoracoscopically, two cysts were resected without injury to the airway, while three had inadvertent tracheo-bronchial injury. Repair of the tracheal injury was possible in one case thoracoscopically, while two cases required conversions, as adequate oxygenation could not be maintained despite on-table endotracheal tube advancement beyond the injury and thoracoscopic manoeuvres. All cases had excellent outcomes at follow-up (median 25months, range 4-39months) with resolution of symptoms and no recurrences. Our report highlights the existence of a common wall between the superior mediastinal FD and the tracheo-bronchial tree. Thoracoscopic resections are feasible including repair of inadvertent airway injury, provided adequate oxygenation can be maintained.

Congenital Esophageal Duplication Cyst-A Rare Case Report and Review of Literature

Alimentary tract duplication is a relatively rare congenital anomaly. Esophageal duplication cyst is an uncommon developmental anomaly among them. Respiratory symptoms are the most common symptoms in foregut duplication, most case present with respiratory distress which may be present from birth, or symptoms may insidious with cough, wheeze, or recurrent respiratory infection. We present a case of newly born baby with respiratory distress, which on radiological investigation showed left mediastinal mass that was removed by excisional biopsy and final histopathology revealed an esophageal cyst.

Perforated neuroenteric cyst masquerading as congenital pulmonary airway malformation

A 3-month-old child was presented with haemoptysis with respiratory distress. Imaging was suggestive of a cavitary lesion in the lung with surrounding consolidation. Diagnosis of a primary lung pathology like...

Surgical Experience and Learning Points in the Management of Foregut Duplication Cysts.

Foregut duplication cysts (FDC) have unpredictable natural course and rarely remain asymptomatic. We present our large cohort of FDC to highlight surgical learning points and outcome. Review of 41 children with FDC (April 1997-April 2015) included demographics, clinical presentation, preoperative investigations, surgical management, post-operative complications, and overall outcome. Forty-one children (26 girls/15 boys; male/female [M/F] ratio 1:1.7) had 43 FDC (two cases >1 cyst) consisting of antenatally diagnosed 16 (39%), late with symptoms 21 (51.2%), and incidental 4(9.8%) cases. FDC locations were cervical (3), mediastinal (35), and subdiaphragmatic (5). During all cervical FDC resection, recurrent laryngeal nerve was required to be dissected off the cyst wall. Thoracoscopic resection was feasible in 27 cases, with 4 conversions related to infections and adhesions or accidental injury to trachea/esophagus. Complete resection was achieved in all cases with no recurrence. There was no mortality and morbidity included, chylothorax (1) esophageal injury (1), phrenic nerve injury (1), and tracheal injury (2). The minimally invasive surgery and surgery on asymptomatic lesions significantly affect the outcome by reducing need for chest drain (p = 0.01) and length of hospital stay (p = 0.04). This retrospective study confirms the efficacy and safety of thoracoscopic surgery; important learning points were association of common wall with trachea in some superior mediastinal cysts and association of recurrent laryngeal nerve in cervical FDC.

Foregut duplication presenting as lingual mass

Abstract Congenital cystic masses of the tongue even though detected prenatally are diagnostic challenge even at birth. Very rarely an intestinal duplication cyst may present in the oral cavity leading to difficulty in feeding, swallowing and breathing. These are often diagnosed by histopathologically demonstrating the presence of either gastrointestinal or respiratory epithelium or sometimes both. We present a female newborn with a swelling of the tongue in the oral cavity from birth, interfering with feeding and inability to close the mouth. The swelling was excised by total transoral resection and reconstruction of the tongue was done and confirmed as foregut duplication cyst on histopathological examination. The tongue swelling gradually subsided and the neonate was discharged.

Foregut duplication cyst with pseudostratified columnar ciliated epithelium: A case report and review of the literature

Foregut duplication cysts are extremely rare congenital malformations. Herein we report a case of 73 year old male with a left upper quadrant abdominal lesion identified on CT scan.

Tubular duplication of the esophagus

Esophageal duplication (ED) is a rare congenital anomaly, representing 10%-15% of all foregut duplications. Neonates may present with respiratory distress, while older children usually present with dysphagia. We report here a rare case of tubular duplication of the esophagus presenting with dysphagia in a 12-year-old Saudi boy.

Intraoral Swelling in a Neonate: Airway Management a Challenge

Foregut duplications occurring in tongue have very few reports in literature. These oral and esophageal lesions may cause feeding and respiratory difficulties. For surgical excision securing airway in these babies is very challenging, as visualization of glottis is hampered, because of lack of space in the mouth. We report a case of twelve hours old neonate referred to our institute for the excision of intraoral swelling under general anaesthesia which was revealed as gastric duplication cyst histopathologically.

Foregut Duplication Cysts in Children.

Background and Objectives:Duplications of the alimentary tract are rare anomalies. We report our experience with foregut duplication cysts including their clinical presentation, diagnostic modalities, and surgical management.Methods:We report a 20-year retrospective review of all foregut duplication cysts managed at our institution.Results:Twelve patients with 13 foregut duplication cysts were identified. The ages of the children at the time of surgery ranged from infancy to adolescence, with a mean age of 7.2 years. Half of the patients presented with abdominal pain and vomiting, and the remaining either had respiratory distress or were asymptomatic. All resections were performed electively. Two of the 11 patients had other congenital anomalies, including a congenital pulmonary airway malformation and coarctation of the aorta. One patient had prenatal diagnosis by ultrasonography. Nine patients underwent complete successful excision with no complications. Three patients whose symptoms resolved during hospitalization remained under observation because of parental preference.Conclusions:Foregut malformation in children may present with a variety of symptoms or can be found incidentally. The decision and timing of surgery is based on the clinical presentation. Surgical intervention in asymptomatic patients should be based on a thorough discussion with the parents.

Endoscopic membranotomy of a tubular type esophageal duplication cyst performed on a 5 year old child

Abstract 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.

Retrocardiac mediastinal foregut duplication cyst

Abstract Foregut duplications occur at various locations throughout the upper gastrointestinal tract, including the thoracic cavity. Thoracic foregut duplications are typically intimately associated with the esophagus and therefore are located in either the posterior mediastinum or in pleural cavity. Here we report a case of foregut duplication of the middle mediastinum, intimately associated with the pericardium and great vessels that contained gastric mucosa, ciliated respiratory epithelium, bronchial-type epithelium, pancreatic tissue and hepatocytes. The literature of mediastinal foregut duplications is also reviewed.

Space occupying lesions in the presence of congenital diaphragmatic hernia

IntroductionPrevious reports describe lung malformations and other chest lesions in association with congenital diaphragmatic hernia (CDH), yet little is known how these lesions affect outcomes. We sought to evaluate the incidence and outcomes of patients diagnosed with chest lesions in association with CDH. MethodsThe charts of all infants treated for CDH in a single tertiary center from January 2004 to January 2015 were reviewed. The outcomes of those with space occupying lesions (SOLs) in association with CDH were compared to those with isolated CDH. Statistical analysis was performed using Student's t-test and Mann–Whitney U test for continuous variables and Fisher's exact for categorical variables. ResultsOf the 214 infants treated, 20 had an associated SOL (4 had>1 lesion). SOLs were confirmed by pathological examination and included: bronchopulmonary sequestration (n=10; 4.7%), ectopic liver (n=9; 4.2%), foregut duplication cyst (n=2; 1%), and other lesions (n=3; 1.4%). No statistical difference was noted in the long-term outcomes of patients with SOL in comparison to those with isolated CDH. ConclusionSOLs are not uncommon in neonates with CDH. Despite theoretical concerns, there is no evidence that SOLs are associated with worse outcomes, a finding which is helpful during prenatal counseling of families.

Lingual foregut duplication cysts in the Neonate: Potential role for marsupialization as initial management

Abstract Foregut duplication cysts (FDCs) are a rare clinical entity encountered by otolaryngologists. Imaging can be suggestive of FDCs, but diagnosis is confirmed by pathological analysis. Traditional management has consisted of definitive surgical excision. We describe a case of a neonate born with a ventral tongue FDC which prevented mouth closure and precluded oral intake. Due to excessive potential for morbidity with surgical excision, the cyst was managed primarily by surgical marsupialization. We review the literature, and suggest that marsupialization may be a desirable temporizing measure for oral cavity cysts, offering an initial alternative to complete excision in the neonatal period.

Foregut Duplication Cyst of the Stomach: A Case Report and Review of the Literature

Duplication cyst of the stomach is a rare congenital malformation, typically diagnosed in the first year of life. In most adult cases the cyst remains asymptomatic, but patients may present with abdominal symptoms including epigastric discomfort or pain. We present a case of a 65-year-old male with an asymptomatic gastric tumor diagnosed incidentally during initial workup of his esophageal adenocarcinoma. Computed tomography revealed a low density soft tissue tumor near the gastroesophageal junction. Endoscopic ultrasonography demonstrated a cystic lesion as a hypoechoic round mass with well-defined borders. Following complete laparoscopic resection, microscopic review revealed a cyst lined with respiratory pseudostratified ciliated columnar epithelium and layers of smooth muscle with an outermost thin fibrous capsule consistent with a foregut duplication cyst.