CASE REPORT

JOP. J Pancreas (Online) 2006; 7(2):200-204.

Duodenal Duplication Cyst with Profound Elevation of Intracystic Carbohydrate Antigen (CA 19-9) and Carcinoembryonic Antigen (CEA): A Rare but Important Differential in the Diagnosis of Cystic Tumours of the Pancreas

Kant J Shah1, Giuseppe Malleo1, Jee Low1, Kassiani Skordilis2, Alistair J Makin3, Ajith K Siriwardena1

1Hepatobiliary Unit, Department of Surgery, 2Department of Histopathology, and 3Department of Gastroenterology, Manchester Royal Infirmary. Manchester, United Kingdom

ABSTRACT

Context Enteric duplication cysts are rare lesions of uncertain incidence and natural history. Pre-operative confirmation of diagnosis can be difficult. This case reports an adult duodenal duplication cyst presenting with grossly elevated intra-lesional levels of tumour markers.

Case report A 57-year-old female was found to have a complex cystic lesion of the head of the pancreas. Intra-lesional fluid analysis revealed a grossly elevated CA 19-9 and CEA. Resection was undertaken under the assumption that this was a cystic tumour. Macroscopic examination after opening the duodenum revealed a villous, circumferential tumour in the proximal duodenum measuring 4 cm in length. A cystic lesion was present in the medial wall of the tumour and did not communicate with the duodenal lumen. Microscopically, the tumour comprised Brunner’s gland hyperplasia with associated mucosal thickening. The wall of the underlying cystic lesion was comprised of muscularis formed by the outer muscle coat of the duodenal wall. The final diagnosis was of a duodenal duplication cyst. There was no evidence of dysplasia or malignancy.

Conclusion This is the first report of a duodenal duplication cyst having elevated intra-cyst fluid levels of amylase, carbohydrate antigen CA 19-9 and carcinoembryonic antigen (CEA). Although rare, this is an important differential diagnosis in the management of cystic tumours of the pancreas.

Full text: HTML format  PDF format

References

  1. Siriwardena AK. Contemporary Management of Pancreatic Pseudocysts. Pancreatology 2005; 5:507-9. [More details]

  2. Brugge WR, Lauwers GY, Sahani D, Fernandez-del Castillo C, Warshaw AL. Cystic neoplasms of the pancreas. N Engl J Med 2004; 351:1218-26. [More details]

  3. Visser BC, Muthusamay VR, Mulvihill SJ, Coakley F. Diagnostic imaging of cystic pancreatic neoplasms. Surg Oncol 2004; 13:27-39. [More details]

  4. Levy MJ, Clain JE. Evaluation and management of cystic pancreatic tumors: emphasis on the role of EUS FNA. Clin Gastroenterol Hepatol 2004; 2:639-53. [More details]

  5. Ryu JK, Woo SM, Hwang JH, Jeong JB, Yoon YB, Park IA, et al. Cyst fluid analysis for the differential diagnosis of pancreatic cysts. Diagn Cytopathol 2004; 31:100-5. [More details]

  6. Martin I, Hammond P, Scott J, Redhead D, Carter DC, Garden OJ. Cystic tumours of the pancreas. Br J Surg 1998; 85:1484-6. [More details]

  7. Frering V, Velecela E, Fouque P, Champetier P, Partensky C. Upper digestive duplications in adults. Ann Chir 1995; 49:928-35. [More details]

  8. Siddiqui AM, Shamberger RC, Filler RM, Perez-Atayde AR, Lillehei CW. Enteric duplications of the pancreatic head: definitive management by local resection. J Pediatr Surg 1998; 33:1117-20. [More details]

  9. Holcomb GW 3rd, Gheissari A, O'Neill JA Jr, Shorter NA, Bishop HC. Surgical management of alimentary tract duplications. Ann Surg 1989; 209:167-74. [More details]

  10. Allen PJ, Jaques DP, D'Angelica M, Bowne WB, Conlon KC, Brennan MF. Cystic lesions of the pancreas: selection criteria for operative and nonoperative management in 209 patients. J Gastrointest Surg 2003; 7:970-7. [More details]

  11. D'Journo XB, Moutardier V, Turrini O, Guiramand J, Lelong B, Pesenti C, et al. Gastric duplication in an adult mimicking mucinous cystadenoma of the pancreas. J Clin Pathol 2004; 57:1215-8. [More details]

  12. Nomura T, Shirai Y, Hatakeyama K, Nikkuni K. An adult ileal duplication cyst containing markedly elevated concentrations of carcinoembryonic antigen and carbohydrate antigen 19-9. Am J Gastroenterol 2002; 97:208-9. [More details]

  13. Ikehata A, Sakuma T. Gastric duplication cyst with markedly elevated concentration of carbohydrate antigen 19-9. Am J Gastroenterol 2000; 95:842-3. [More details]

  14. Legros JR, Duclaut I, Michot F, Teniere P, Scotte M. Cystic mass of the pancreatic region associated with elevated CA 19-9 serum levels revealing a colonic duplication in an adult. Gastroenterol Clin Biol 2004; 28:1291-3. [More details]

  15. Falk GL, Young CJ, Parer J. Adenocarcinoma arising in a duodenal duplication cyst: a case report. Aust N Z J Surg 1991; 61:551-3. [More details]

  16. Shivnani AT, Small W Jr, Benson A IIIrd, Rao S, Talamonti MS. Adenocarcinoma arising in rectal duplication cyst: case report and review of the literature. Am Surg 2004; 70:1007-9. [More details]

Keywords Cyst Fluid; Pancreatic Cyst; Tumor Markers, Biological

Correspondence Ajith K Siriwardena: ajith.siriwardena@cmmc.nhs.uk

JOP Home page