Is Preoperative Diagnosis Possible? A Clinical and Radiological Review of Lymphoepithelial Cysts of the Pancreas
Lymphoepithelial cysts of the pancreas are rare lesions found mainly in middle-aged men. They are usually benign and have no clear natural history except one study linking their occurrence with HIV infection. Nevertheless, they often cause enormous psychological stress to patients as they tend to mimic pancreatic neoplasms which are known to carry poor prognosis. The authors have therefore assessed the published literature from PubMed in order to determine whether lymphoepithelial cysts can be diagnosed preoperatively using novel imaging techniques. Based on our findings, it is evident that three-dimensional computed tomography scans, in-phase and out-of phase magnetic resonance imaging studies, and endosonography have enabled better characterization of pancreatic lymphoepithelial cysts than a decade ago. Endoscopic ultrasound-guided fine needle aspiration has also added considerably to the promise of preoperative diagnosis. Thus, the authors can affirm that despite surgical excision of the cyst with pathological examination being the gold standard for diagnosis, it is possible that a combination of the modern imaging techniques and/or minimally-invasive approach can enable patients avoid unnecessary surgery in the future.
Image: Proposed algorithm for diagnosis and management of lymphoepithelial cysts.
Molvar C, Kayhan A, Lakadamyali H, Oto A. Nonneoplastic cystic lesions of pancreas: a practical clinical, histologic, and radiologic approach. Curr Probl Diagn Radiol. 2011;40:141-148. [PMID: 21616276].
Luchtrath H, Schriefers KH. A pancreatic cyst with features of a so-called branchiogenic cyst. Pathologe. 1985;6:217–219. [PMID: 4048076].
Sewkani A, Purohit D, Singh V, Purohit D, Varshney R, Varshney S. Lymphoepithelial cyst of the pancreas: a rare case report and review of literature. Indian J Surg. 2010;72:427–432. [PMID: 22131649].
Truong LD, Rangdaeng S, Jordan PH. Lymphoepithelial cyst of the pancreas. Am J Surg Pathol. 1987;11:899–903. [PMID: 3674287].
Adsay NV, Hasteh F, Cheng JD, Bejarano PA, Lauwers GY, Batts KP. Lymphoepithelial cysts of the pancreas: a report of 12 cases and a review of the literature. Mod Pathol. 2002;15:492–501. [PMID: 12011254].
Raval JS, Zeh HJ, Moser AJ, Lee KK, Sanders MK, Navina S. Pancreatic lymphoepithelial cysts express CEA and can contain mucous cells: potential pitfalls in the preoperative diagnosis. Mod Pathol. 2010;23:1467-1476. [PMID: 20802468].
Kalb B, Sarmiento JM, Kooby DA, Adsay NV, Martin DR. MR Imaging of cystic lesions of the pancreas. Radiographics. 2009;29:1749–1765. [PMID: 19959519].
Kudo D, Hashimoto N, Toyoki Y, Narumi S, Hakamada K. Usefulness of in-phase and out-of-phase magnetic resonance imaging for the detection of pancreatic lymphoepithelial cyst. Hepatogastroenterology. 2011;58:1403-1404. [PMID: 21937416].
Jian B, Kimbrell HZ, Sepulveda A, Yu G. Lymphoepithelial cysts of the pancreas: endosonography-guided fine needle aspiration. Diagn Cytopathol. 2008;36:662-665. [PMID: 18677749].
Garcea G, Ong SL, Rajesh A, Neal CP, Pollard CA, Berry DP, Dennison AR. Cystic lesions of the pancreas. A diagnostic and management dilemma. Pancreatology. 2008;8:236–251. [PMID: 18497542]
Chatelain D, Couvelard A, Hammel P, Terris B, Palazzo L, Verhaeghe P, et al. Lympho-epithelial cyst of the pancreas: a difficult diagnosis. About two cases. Gastroenterol Clin Biol. 2002;26:935-938. [PMID: 12434105].
Barbaros U, Erbil Y, Kapran Y, Bozbora A, Ozarmagan S, Bilgiç B. Lymphoepithelial cyst: a rare cystic tumor of the pancreas which mimics carcinoma. JOP. 2004;5:392-394. [PMID: 15365210].
Toumi Z, Ammori MB, Al-Habba S, Filobbos R, Ammori BJ. Laparoscopic distal pancreatectomy for a large pancreatic lymphoepithelial cyst: a rare entity. Surg Laparosc Endosc Percutan Tech. 2010;20:e211-214. [PMID: 21150405].
Clemente G, Sarno G, De Rose AM, Giordano M, Ricci R, Vecchio FM, Nuzzo G. Lymphoepithelial cyst of the pancreas: case report and review of the literature. Acta Gastroenterol Belg. 2011;74:343-346. [PMID: 21861322].
Truong LD, Stewart MG, Hao H, Yutani C, Jordan PH. A comprehensive characterization of lymphoepithelial cyst associated with the pancreas. Am J Surg. 1995;170:27–32. [PMID: 7793490].
Schwarz RE, Weiss LM. Lymphoepithelial cyst of the pancreas. No evidence for Epstein-Barr virus-related pathogenesis. Int J Pancreatol. 1999;25:223–227. [PMID: 10453424]
Bédat B, Genevay M, Dumonceau JM, Frossard JL, Forget J, Morel P, Berney T. Association between lymphoepithelial cysts of the pancreas and HIV infection. Pancreatology. 2012;12:61-64. [PMID: 22487477].
Iacono C, Cracco N, Zamboni G, Bernardello F, Zicari M, Marino F, et al. Lymphoepithelial cyst of the pancreas. Report of two cases and review of the literature. Int J Pancreatol. 1996;19:71–76. [PMID: 8656030].
Schinke-Nickl DA, Muller MF. Case report: Lymphoepithelial cyst of the pancreas. Br J Radiol. 1996;69:876–878. [PMID: 8983597].
Adsay NV, Hasteh F, Cheng JD, Klimstra DS. Squamous-lined cysts of the pancreas: Lymphoepithelial cysts, dermoid cysts (teratomas), and accessory-splenic epidermoid cysts. Semin Diagn Pathol. 2000;17:56–65. [PMID: 10721807].
Hisaoka M, Haratake J, Horie A, Yasunami Y, Kimura T. Lymphoepithelial cyst of the pancreas in a 65-year-old man. Hum Pathol. 1991;22:924–926. [PMID: 1717361]
Arai T, Kino I, Nakamura S, Ogawa H. Epidermal inclusions in abdominal lymph nodes. Report of two cases studied immunohistochemically. Acta Pathol Jpn. 1992;42:126-129. [PMID: 1561883].
Sako S, Isozaki H, Hara H, Tsutsumi A, Tanigawa N. Cystic lymphoepithelial lesions of the pancreas and peripancreatic region: report of two cases. Surg Today. 1999;29:467–471. [PMID: 10333422].
Capitanich P, Iovaldi ML, Medrano M, Malizia P, Herrera J, Celeste F, et al. Lymphoepithelial cysts of the pancreas: case report and review of the literature. J Gastrointest Surg. 2004;8:342–345. [PMID: 15019932].
Tateyama H, Tada T, Murase T, Fujitake S, Eimoto T. Lymphoepithelial cyst and epidermoid cyst of the accessory spleen in the pancreas. Mod Pathol. 1998;1171-1177. [PMID: 9872647].
Maekawa H, Yoneyama H, Komatsu Y, Orita H, Sakurada M, Sato K, Yamano M. Pancreatic lymphoepithelial cyst with an intracystic papillary projection. Report of a case. JOP. 2009;10:694-696. [PMID: 19890197].
Yamaguchi T, Takahashi H, Kagawar R, Takeda R, Sakata S, Yamamoto M, Nishizaki D. Lymphoepithelial cyst of the pancreas associated with elevated CA 19-9 levels. J Hepatobiliary Pancreat Surg. 2008;15:652-654. [PMID: 18987938].
Nam SJ, Hwang HK, Kim H, Yu JS, Yoon DS, Chung JJ, et al. Lymphoepithelial cysts in the pancreas: MRI of two cases with emphasis of diffusion-weighted imaging characteristics. J Magn Reson Imaging. 2010;32:692-696. [PMID: 20815068].
Micke O, Schafer U, Willich N. Persistent elevation of CA 19–9 levels in a patient with an extended retroperitoneal dermoid. Anticancer Res. 1999;19:2717–2720. [PMID: 10470227].
Matrone A, Russo M, Mollica C, Lombardi D, Lombardi G, Maurea S, Imbriaco M. Lymphoepithelial pancreatic cyst: an atypical benign pancreatic mass presenting with a “cheerios-like” appearance. JOP. 2010;11:170-172. [PMID: 20208329].
Karim Z, Walker B, Lam E. Lymphoepithelial cysts of the pancreas: the use of endoscopic ultrasound-guided fine-needle aspiration in diagnosis. Can J Gastroenterol. 2010;24:348–350. [PMID: 20559575].
Idetsu A, Ojima H, Saito K, Hirayama I, Hosouchi Y, Nishida Y, et al. Lymphoepithelial cyst of the pancreas: report of a case. Surg Today. 2008;38:68-71. [PMID: 18085369].
Madan R, Khan E, Cuka N, Olyaee M, Tawfik O, Fan F. Pancreatic cystic lesions without overt cytologic atypia: proposed diagnostic categories for endoscopic ultrasound-guided fine-needle aspiration cytology with utilization of fluid carcinoembryonic antigen level. Acta Cytol. 2012;56:34-40. [PMID: 22236743].
Kim YH, Auh YH, Kim KW, Lee MG, Kim KS, Park SY. Lymphoepithelial cysts of the pancreas: CT and sonographic findings. Abdom Imaging. 1998;23:185–187. [PMID: 9516512].
Neyman EG, Georgiades CS, Horton KH, Lillemoe KD, Fishman EK. Lymphoepithelial cyst of the pancreas—evaluation with multidetector CT. Clin Imaging. 2005;29:345-347. [PMID: 16153542].
Kim WH, Lee JY, Park HS, Won HJ, Kim YH, Choi JY. Lymphoepithelial cyst of the pancreas: comparison of CT findings with other pancreatic cystic lesions. Abdom Imaging. 2012;[Epub ahead of print]. [PMID: 22610041].
Renou C, Giovannini M, Monges G, Rifflet H, Muller P, Harafa A, et al. Pitfalls of cyst fluid findings obtained by endoscopic ultrasonography fine-needle aspiration on a pancreatic lymphoepithelial cyst. Am J Gastroenterol. 2007;102:213-215. [PMID: 17278274].
Ahlawat SK. Lymphoepithelial cyst of the pancreas: role of endoscopic ultrasound guided fine needle aspiration. JOP. 2008;9:230–234. [PMID: 18326936].
Nasr J, Sanders M, Fasanella K, Khalid A, McGrath K. Lymphoepithelial cysts of the pancreas: an EUS case series. Gastrointest Endosc. 2008; 68:170-173. [PMID: 19513719].
Rafii A, Ferron G, Lacroix-Triki M, Dalenc F, Gladieff L, Querleu D. Abdominal wall metastasis of ovarian carcinoma after low transverse abdominal incision: report of two cases and review of literature. Int J Gynecol Cancer. 2006;16:334–337. [PMID: 16515617].
Hirooka Y, Goto H, Itoh A, Hashimoto S, Niwa K, Ishikawa H. Case of intraductal papillary mucinous tumor in which endosonography- guided fine-needle aspiration biopsy caused dissemination. J Gastroenterol Hepatol. 2003;18:1323–1324. [PMID: 14535994].
Zieliñska-Pajak E, Liszka L, Pajak J, Gołka D, Lampe P, Pilch-Kowalczyk J, Mrowiec S. Two cases of lymphoepithelial cyst of the pancreas: first - a cyst coexisting with a primary gastric lymphoma, second - incidentally found pancreatic cyst. Z Gastroenterol. 2008;46:1188-1193. [PMID: 18937188].
Koga H, Takayasu K, Mukai K, Muramatsu Y, Mizuguchi Y, Furukawa H, et al. CT of lymphoepithelial cysts of the pancreas. J Comput Assist Tomogr. 1995;19:221-224. [PMID: 7890845].
England GM, Allen PW. Selected case from the Arkadi M. Rywlin International Pathology Slide Seminar: benign lymphoepithelial cyst, head of pancreas. Adv Anat Pathol. 2011; 18:98-100. [PMID: 21169743].
Briggs CD, Mann CD, Irving GR, Neal CP, Peterson M, Cameron IC, Berry DP. Systematic review of minimally invasive pancreatic resection. J Gastrointest Surg. 2009;13:1129–1137. [PMID: 19130151].
Copyright (c) 2014 Stephen Osiro, Jonier R Rodriguez, Kevin J Tiwari, Iliana I Rodriguez, Njambi Mathenge, Robert Shane Tubbs, Marios Loukas
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