Esophageal Atresia and Gastric Ectopic Pancreas: Is There a Real Association?

Abstract

Objective Heterotopic pancreas (HP) is a condition in which there is well-differentiated pancreatic tissue that lacks any anatomic or vascular contact with the pancreatic gland. It normally arises from the stomach but can be found in other locations. Although it is usually asymptomatic, obstructive symptoms, bleeding, or malignant degeneration can occur. The incidence is very low, but it is significantly more common in patients with esophageal atresia (EA). The aim of this study is to evaluate the incidence of HP in patients with and without EA and to compare the results in both groups.

Material and Methods We conducted a 2-year prospective study in pediatric patients who benefited from an upper gastrointestinal endoscopy. Patients were divided into two groups: group “A” comprised patients with EA and group “B” those without EA. The variables analyzed were the clinical presentation, presence of HP, location, associated malformations, genetic disorders, and management.

Results A total of 192 consecutive patients were included in the study: 51 (26.6%) in group A and 141 (73.4%) in group B. Indications for endoscopy in group B were eosinophilic esophagitis in 37 (19.2%) patients, celiac disease in 23 (11.95%) patients, and other disorders in 81 (42.2%) patients. Gastric HP was found in seven patients, all of them in group A. All lesions were hosted in the prepyloric antrum. The prevalence of HP in groups A and B was 13.7 and 0%, respectively (p < 0.05). Female gender was predominant in patients with AE and HP, this result being statistically significant (p = 0.044). No other associated malformation or genetic syndrome studied showed association with HP. Only one patient debuted with upper gastrointestinal (GI) bleeding and required excision, while six patients were asymptomatic. The mean follow-up was 54 months (range: 45–78 months).

Conclusion The incidence of gastric HP is more common in patients with EA, with the female gender being a risk factor for their association. Active search and follow-up is recommended as it may become symptomatic anytime and need resection.

Keywords

ectopic pancreas - heterotopic pancreas - esophageal atresia - gastrointestinal bleeding

Publication History

Received: 17 March 2023

Accepted: 10 July 2023

Accepted Manuscript online:
12 July 2023

Article published online:
29 September 2023

• References

• 1 Sathyanarayana SA, Deutsch GB, Bajaj J. et al. Ectopic pancreas: a diagnostic dilemma. Int J Angiol 2012; 21 (03) 177-180
• 2 Trifan A, Târcoveanu E, Danciu M, Huţanaşu C, Cojocariu C, Stanciu C. Gastric heterotopic pancreas: an unusual case and review of the literature. J Gastrointestin Liver Dis 2012; 21 (02) 209-212
• 3 Park J. Heterotopic pancreas of the esophagus and stomach associated with pure esophageal atresia. J Pediatr Surg 2010; 45 (03) E25-E27
• 4 Makarewicz W, Bobowicz M, Dubowik M, Kosinski A, Jastrzebski T, Jaskiewicz J. Endoscopic submucosal dissection of gastric ectopic pancreas. Wideochir Inne Tech Malo Inwazyjne 2013; 8 (03) 249-252
• 5 Rezvani M, Menias C, Sandrasegaran K, Olpin JD, Elsayes KM, Shaaban AM. Heterotopic pancreas: histopathologic features, imaging findings, and complications. Radiographics 2017; 37 (02) 484-499
• 6 Karakus SC, Sarsu SB, Celtık C, Gokce S, Koku N. Rare causes of gastrointestinal hemorrhage in infancy. Pediatr Int 2015; 57 (02) 304-307
• 7 Henderson L, Nour S, Dagash H. Heterotopic pancreas: a rare cause of gastrointestinal bleeding in children. Dig Dis Sci 2018; 63 (05) 1363-1365
• 8 Vitiello GA, Cavnar MJ, Hajdu C. et al. Minimally invasive management of ectopic pancreas. J Laparoendosc Adv Surg Tech A 2017; 27 (03) 277-282
• 9 Halkic N, Nordback P. Soft-tissue images. Malignant degeneration of heterotopic pancreas. Can J Surg 2001; 44 (06) 407
• 10 Sharma S, Agarwal S, Nagendla MK, Gupta DK. Omental acinar cell carcinoma of pancreatic origin in a child: a clinicopathological rarity. Pediatr Surg Int 2016; 32 (03) 307-311
• 11 Ogata H, Oshio T, Ishibashi H, Takano S, Yagi M. Heterotopic pancreas in children: review of the literature and report of 12 cases. Pediatr Surg Int 2008; 24 (03) 271-275
• 12 Seddon K, Stringer MD. Gastric heterotopic pancreas in children: a prospective endoscopic study. J Pediatr Surg 2020; 55 (10) 2154-2158
• 13 Klob L. Pancreas accesorium. Zeitschriftder Kaiserl. Konigl. Gesellschaft der Aerzte zu Wien 1859; 15: 732
• 14 Yuan Z, Chen J, Zheng Q, Huang XY, Yang Z, Tang J. Heterotopic pancreas in the gastrointestinal tract. World J Gastroenterol 2009; 15 (29) 3701-3703
• 15 Andrassy RJ, Mahour GH. Gastrointestinal anomalies associated with esophageal atresia or tracheoesophageal fistula. Arch Surg 1979; 114 (10) 1125-1128
• 16 Zhang Y, Sun X, Gold JS. et al. Heterotopic pancreas: a clinicopathological study of 184 cases from a single high-volume medical center in China. Hum Pathol 2016; 55: 135-142
• 17 Filip R, Walczak E, Huk J, Radzki RP, Bieńko M. Heterotopic pancreatic tissue in the gastric cardia: a case report and literature review. World J Gastroenterol 2014; 20 (44) 16779-16781
• 18 Ormarsson OT, Gudmundsdottir I, Mårvik R. Diagnosis and treatment of gastric heterotopic pancreas. World J Surg 2006; 30 (09) 1682-1689
• 19 Gaspar Fuentes A, Campos Tarrech JM, Fernández Burgui JL. et al. Pancreatic ectopias. Rev Esp Enferm Apar Dig 1973; 39 (03) 255-268
• 20 Ozcan C, Celik A, Güçlü C, Balik E. A rare cause of gastric outlet obstruction in the newborn: pyloric ectopic pancreas. J Pediatr Surg 2002; 37 (01) 119-120
• 21 Wei R, Wang QB, Chen QH, Liu JS, Zhang B. Upper gastrointestinal tract heterotopic pancreas: findings from CT and endoscopic imaging with histopathologic correlation. Clin Imaging 2011; 35 (05) 353-359
• 22 Gottschalk U, Dietrich CF, Jenssen C. Ectopic pancreas in the upper gastrointestinal tract: is endosonographic diagnosis reliable? Data from the German Endoscopic Ultrasound Registry and review of the literature. Endosc Ultrasound 2018; 7 (04) 270-278
• 23 Rodriguez FJ, Abraham SC, Allen MS, Sebo TJ. Fine-needle aspiration cytology findings from a case of pancreatic heterotopia at the gastroesophageal junction. Diagn Cytopathol 2004; 31 (03) 175-179
• 24 Zhang Y, Huang Q, Zhu LH, Zhou XB, Ye LP, Mao XL. Endoscopic excavation for gastric heterotopic pancreas: an analysis of 42 cases from a tertiary center. Wien Klin Wochenschr 2014; 126 (17–18): 509-514
• 25 Hwang JH, Rulyak SD, Kimmey MB. American Gastroenterological Association Institute. American Gastroenterological Association Institute technical review on the management of gastric subepithelial masses. Gastroenterology 2006; 130 (07) 2217-2228
• 26 Moreau B, Lévesque D, Faure C. Association of gastric heterotopic pancreas and esophageal atresia in children. J Pediatr Gastroenterol Nutr 2010; 50 (04) 394-396
• 27 Gálvez-Valdovinos R, Mendoza-Rodríguez A, Coronado-Perez JH, Santillan EM, Funes-Rodríguez F. Laparoscopic treatment of heterotopic pancreas in the prepyloric region. J Minim Access Surg 2006; 2 (04) 224-226
• 28 Khashab MA, Cummings OW, DeWitt JM. Ligation-assisted endoscopic mucosal resection of gastric heterotopic pancreas. World J Gastroenterol 2009; 15 (22) 2805-2808
• 29 Iwamuro M, Tsuzuki T, Ohya S. et al. Ectopic pancreas in the stomach successfully resected by endoscopic submucosal dissection. Case Rep Med 2015; 147927