University of Central Florida College of Medicine Kissimmee, FL
Yu Y. Nway, MD1, Ernesto Robalino Gonzaga, MD2, Parikh Jignesh, MD3, Vergeli-Rojas Jeannette, MD3; 1University of Central Florida College of Medicine, Kissimmee, FL; 2University of Central Florida College of Medicine, Orlando, FL; 3Orlando VA Medical Center, Orlando, FL
Introduction: Salivary gland heterotopia describes salivary gland tissues found in locations other than the salivary gland. A choristoma is a benign tumor or mass formed by heterotopic tissues. It is commonly seen within the head and neck region. Gastrointestinal salivary gland heterotopia or choristoma are rare but have been noted in a few cases, found in rectal or perianal regions, large bowel, jejunum, esophagus and gastroesophageal junction. Here, we report a rare case of salivary gland choristoma within the gastroesophageal junction followed by literature review.
Methods: A 48-year-old gentleman presented with chronic intermittent rectal bleeding worsening to 4 episodes per week for one month. His past medical history included hypertension, hyperlipidemia, mild intermittent asthma, hemorrhoids and gastroesophageal reflux disease. Endoscopic evaluation revealed esopheageal mucosal changes suggestive of Barrett’s esophagus in the lower third of esophagus and a single 5mm nodule in gastroesophageal junction. Biopsy confirmed Barrett’s esophagus, as well as, pancreatic heterotopia in the gastroesophageal junction. Follow up EGD after one year revealed development of salivary gland choristoma in gastroesophageal junction. Patient recommended to continue PPI therapy and follow up EUS in 4 months. Discussion: Salivary gland heterotopia or choristoma in the gastroesophageal junction is a rare entity with only three published cases. All three cases had a history of gastroesophageal reflux disease or its complications such as reflux esophagitis or Barrett’s esophagus. Only one case presented with choristoma while the other two cases revealed heterotopia. Although our case has history of GERD, the main presenting symptom is rectal bleeding which was absent in other cases. Moreover, our case had pancreatic heterotopia at first endoscopy. The pathogenesis of Barrett’s esophagus, intestinal columnar metaplasia, is due to chronic exposure to acid and noxious agents. Pancreatic and salivary gland heterotopia could be due to metaplasia but these findings are yet to be investigated. It is also important to notice that heterotopia or choristoma is related to gastroesophageal reflux disease and the appearance of choristoma can mimic carcinoma which can lead to unnecessary investigation and treatment.
5mm nodule in gastroesophageal junction showing pancreatic heterotopia on biopsy
Esophageal mucosal changes suggestive for long segment Barrett's esophagus present in the lower third of the esophagus. The maximal longitudinal extent of the mucosal changes was 3cm in length.
High magnification appearance of mucus glands of heterotopic salivary glands.
Disclosures: Yu Nway indicated no relevant financial relationships. Ernesto Robalino Gonzaga indicated no relevant financial relationships. Parikh Jignesh indicated no relevant financial relationships. Vergeli-Rojas Jeannette indicated no relevant financial relationships.