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Published in: Indian Journal of Surgery 6/2010

01-12-2010 | Review Article

Lymphoepithelial Cyst of the Pancreas: A Rare Case Report and Review of Literature

Authors: Ajit Sewkani, Deepak Purohit, Vikrant Singh, Aruna Jain, Rajneesh Varshney, Subodh Varshney

Published in: Indian Journal of Surgery | Issue 6/2010

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Abstract

Lymphoepithelial cysts are rare pancreatic lesions of undetermined pathogenesis. The literature on this entity is limited to case reports or small series. We describe a case of 66 year male, incidentally diagnosed as lymphoepithelial cyst of pancreas that was managed by enucleation. This is the first case report of lymphoepithelial cyst from India. An extensive Medline search was carried out for lymphoepithelial cyst of pancreas. Till date less than 100 cases were identified in available literature. All these cases (including our case) were analyzed. This entity has uniform and distinctive clinicopathological features. About half of the reported cases were asymptomatic with most of the lesions diagnosed incidentally. Majority of patients presents with non-specific symptoms making preoperative diagnosis difficult. Lymphoepithelial cyst of the pancreas is a rare benign lesion, which is difficult to diagnose preoperatively. High index of suspicion and preoperative fine needle aspiration cytology may help in making diagnosis and avoiding surgery in asymptomatic patients.
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Literature
1.
go back to reference Brugge WR, Lauwers GY, Sahani D, Fernandez del Castillo C, Warshaw AL (2004) Cystic neoplasms of the pancreas. N Engl J Med 351:1218–1226PubMedCrossRef Brugge WR, Lauwers GY, Sahani D, Fernandez del Castillo C, Warshaw AL (2004) Cystic neoplasms of the pancreas. N Engl J Med 351:1218–1226PubMedCrossRef
2.
go back to reference Kloppel G, Solcia E, Longnecker DS, Capella C, Sobin LH (1996) World Health Organization international histologic classification of tumors. Histologic typing of tumors of the exocrine pancreas. In: World Health Organization—international histologic classification of tumors. Vol 2. Geneva, Switzerland: Springer Kloppel G, Solcia E, Longnecker DS, Capella C, Sobin LH (1996) World Health Organization international histologic classification of tumors. Histologic typing of tumors of the exocrine pancreas. In: World Health Organization—international histologic classification of tumors. Vol 2. Geneva, Switzerland: Springer
3.
go back to reference Adsay NV, Klimstra DS, Compton CC (2000) Cystic lesions of the pancreas. Semin Diagn Pathol 17:1–6PubMed Adsay NV, Klimstra DS, Compton CC (2000) Cystic lesions of the pancreas. Semin Diagn Pathol 17:1–6PubMed
4.
go back to reference Gracea G, Ong SL, Rajesh A, Neal CP, Pollard CA, Berry DP et al (2008) Cystic lesion of the pancreas. A diagnostic & management dilemma. Pancreatology 8(3):236–251CrossRef Gracea G, Ong SL, Rajesh A, Neal CP, Pollard CA, Berry DP et al (2008) Cystic lesion of the pancreas. A diagnostic & management dilemma. Pancreatology 8(3):236–251CrossRef
5.
go back to reference Compton CC (2000) Serous cystic tumors of the pancreas. Semin Diagn Pathol 17:43–56PubMed Compton CC (2000) Serous cystic tumors of the pancreas. Semin Diagn Pathol 17:43–56PubMed
6.
go back to reference Tollefson MK, Libsch KD, Sarr MG et al (2003) Intraductal papillary mucinous neoplasm: did it exist prior to 1980? Pancreas 26:E55–E58PubMedCrossRef Tollefson MK, Libsch KD, Sarr MG et al (2003) Intraductal papillary mucinous neoplasm: did it exist prior to 1980? Pancreas 26:E55–E58PubMedCrossRef
7.
go back to reference Rivera JA, Catillo CF, Pins M et al (1997) Pancreatic mucinous ductal ectasia and intraductal papillary neoplasms. A single malignant clinicopathologic entity. Ann Surg 225:637–646PubMedCrossRef Rivera JA, Catillo CF, Pins M et al (1997) Pancreatic mucinous ductal ectasia and intraductal papillary neoplasms. A single malignant clinicopathologic entity. Ann Surg 225:637–646PubMedCrossRef
8.
go back to reference Luchtrath H, Schriefers KH (1985) A pancreatic cyst with features of a so-called branchiogenic cyst. Pathologe 6:217–219PubMed Luchtrath H, Schriefers KH (1985) A pancreatic cyst with features of a so-called branchiogenic cyst. Pathologe 6:217–219PubMed
9.
go back to reference Adsay NV, Hasteh F, Cheng JD, Bejarano PA, Lauwers GY, Batts KP et al (2002) Lymphoepithelial cyst of the pancreas: a report of 12 cases and a review of the literature. Mod Pathol 15:492–501PubMedCrossRef Adsay NV, Hasteh F, Cheng JD, Bejarano PA, Lauwers GY, Batts KP et al (2002) Lymphoepithelial cyst of the pancreas: a report of 12 cases and a review of the literature. Mod Pathol 15:492–501PubMedCrossRef
10.
go back to reference Truong LD, Rangdaeng S, Jordan PH Jr (1987) Lymphoepithelial cyst of the pancreas. Am J Surg Pathol 11:899–903PubMedCrossRef Truong LD, Rangdaeng S, Jordan PH Jr (1987) Lymphoepithelial cyst of the pancreas. Am J Surg Pathol 11:899–903PubMedCrossRef
11.
go back to reference Chatelain D, Couvelard A, Hammel P, Terris B, Palazzo L, Verhaeghe P et al (2002) Lympho-epithelial cyst of the pancreas: a difficult diagnosis. About two cases. Gastroenterol Clin Biol 26:935–938PubMed Chatelain D, Couvelard A, Hammel P, Terris B, Palazzo L, Verhaeghe P et al (2002) Lympho-epithelial cyst of the pancreas: a difficult diagnosis. About two cases. Gastroenterol Clin Biol 26:935–938PubMed
12.
go back to reference Imamura H, Tada S, Hirota K, Suko H, Kamio T (2002) Forty-six months follow up of a patient with pancreatic lymphoepithelial cyst. Nippon Shokakibyo Gakkai Zasshi 99:527–532PubMed Imamura H, Tada S, Hirota K, Suko H, Kamio T (2002) Forty-six months follow up of a patient with pancreatic lymphoepithelial cyst. Nippon Shokakibyo Gakkai Zasshi 99:527–532PubMed
13.
go back to reference Zou XP, Li YM, Li ZS, Xu GM (2004) Lymphoepithelial cyst of the pancreas: a case report. Hepatobiliary Pancreat Dis Int 3:155–157PubMed Zou XP, Li YM, Li ZS, Xu GM (2004) Lymphoepithelial cyst of the pancreas: a case report. Hepatobiliary Pancreat Dis Int 3:155–157PubMed
14.
go back to reference Capitanich P, Lovaldi ML, Medrano M, Malizia P, Herrara J, Celeste F et al (2004) Lymphoepithelial cyst of the pancreas: a case report and review of the literature. J Gastrointest Surg 8:342–345PubMedCrossRef Capitanich P, Lovaldi ML, Medrano M, Malizia P, Herrara J, Celeste F et al (2004) Lymphoepithelial cyst of the pancreas: a case report and review of the literature. J Gastrointest Surg 8:342–345PubMedCrossRef
15.
go back to reference Jouini M, Bedioui H, Nouira K, Menif E, Haouet S, Slim R (2004) Lymphoepithelial cyst of the pancreas: a case report. Ann Chir 129:293–296PubMed Jouini M, Bedioui H, Nouira K, Menif E, Haouet S, Slim R (2004) Lymphoepithelial cyst of the pancreas: a case report. Ann Chir 129:293–296PubMed
16.
go back to reference Au-Young IT, Wemyss-Holden S, Llyod DM (2004) Laparoscopic distal pancreatectomy for lymphoepithelial cyst of the pancreas. JSLS 8:389–390 Au-Young IT, Wemyss-Holden S, Llyod DM (2004) Laparoscopic distal pancreatectomy for lymphoepithelial cyst of the pancreas. JSLS 8:389–390
17.
go back to reference Barbaros U, Erbil Y, Kapran Y, Bozbora A, Ozarmagan S, Bilgic B (2004) Lymphoepithelial cyst: a rare cystic tumor of the pancreas which mimics carcinoma. JOP 5:392–394PubMed Barbaros U, Erbil Y, Kapran Y, Bozbora A, Ozarmagan S, Bilgic B (2004) Lymphoepithelial cyst: a rare cystic tumor of the pancreas which mimics carcinoma. JOP 5:392–394PubMed
18.
go back to reference Ciprian Corby S, Weinbreck N, Nicolas M, Laurent V, Vignaud JM, Regent D (2005) Lymphoepithelial cyst of the pancreas: a case report. J Radiol 86:335–336PubMedCrossRef Ciprian Corby S, Weinbreck N, Nicolas M, Laurent V, Vignaud JM, Regent D (2005) Lymphoepithelial cyst of the pancreas: a case report. J Radiol 86:335–336PubMedCrossRef
19.
go back to reference Neyman EG, Georgiades CS, Horton KH, Lillemore KD, Fishman EK (2005) Lymphoepithelial cyst of the pancreas—evaluation with multidetector CT. Clin Imaging 29:345–347PubMedCrossRef Neyman EG, Georgiades CS, Horton KH, Lillemore KD, Fishman EK (2005) Lymphoepithelial cyst of the pancreas—evaluation with multidetector CT. Clin Imaging 29:345–347PubMedCrossRef
20.
go back to reference Kanno A, Suzuki M, Onodera H, Suzuki S, Kayaba Y, Sasaki A et al (2005) Lymphoepithelial cyst of the pancreas: report of two cases. Nippon Shokakibyo Gakkai Zasshi 102:612–618PubMed Kanno A, Suzuki M, Onodera H, Suzuki S, Kayaba Y, Sasaki A et al (2005) Lymphoepithelial cyst of the pancreas: report of two cases. Nippon Shokakibyo Gakkai Zasshi 102:612–618PubMed
21.
go back to reference Castaldo ET, Stumph JR, Merchant N (2006) Lymphoepithelial cyst of the pancreas. Surgery 140:476–478PubMedCrossRef Castaldo ET, Stumph JR, Merchant N (2006) Lymphoepithelial cyst of the pancreas. Surgery 140:476–478PubMedCrossRef
22.
go back to reference Shinmura R, Gabata T, Matsui O (2006) Lymphoepithelial cyst of the pancreas: case report with special reference to imaging—pathologic correlation. Abdom Imaging 31:106–109PubMedCrossRef Shinmura R, Gabata T, Matsui O (2006) Lymphoepithelial cyst of the pancreas: case report with special reference to imaging—pathologic correlation. Abdom Imaging 31:106–109PubMedCrossRef
23.
go back to reference Colovic R, Grubor N, Radak V, Micev M, Colovic N (2006) Lymphoepithelial cyst of the pancreas. Srp Arh Celok Lek 134:64–66PubMedCrossRef Colovic R, Grubor N, Radak V, Micev M, Colovic N (2006) Lymphoepithelial cyst of the pancreas. Srp Arh Celok Lek 134:64–66PubMedCrossRef
24.
go back to reference Ivicic J, Hermanova M, Vomela J (2007) Benign Lymphoepithelial cyst of the pancreas: a case report. Rozhl Chir 86(6):300–302PubMed Ivicic J, Hermanova M, Vomela J (2007) Benign Lymphoepithelial cyst of the pancreas: a case report. Rozhl Chir 86(6):300–302PubMed
25.
go back to reference Younus S, Bleibel W, Bleibel H, Hernady N (2007) Lymphoepithelial cyst of the pancreas. Dig Dis Sci 52(11):3136–3139PubMedCrossRef Younus S, Bleibel W, Bleibel H, Hernady N (2007) Lymphoepithelial cyst of the pancreas. Dig Dis Sci 52(11):3136–3139PubMedCrossRef
26.
go back to reference Idetsu A, Ojima H, Saito K, Hirayama I, Hosouchio Y, Nishida Y et al (2008) Lymphoepithelial cyst of the pancreas: report of a case. Surg Today 348(1):68–71CrossRef Idetsu A, Ojima H, Saito K, Hirayama I, Hosouchio Y, Nishida Y et al (2008) Lymphoepithelial cyst of the pancreas: report of a case. Surg Today 348(1):68–71CrossRef
27.
go back to reference Frezza EE, Wachtel MS (2008) Lymphoepithelial cyst of the pancreas tail: case report and review of literature. JOP 9(1):46–49PubMed Frezza EE, Wachtel MS (2008) Lymphoepithelial cyst of the pancreas tail: case report and review of literature. JOP 9(1):46–49PubMed
28.
go back to reference Roger N, Rousseau G, Bardier A, Vaillant JC, Hannoun L (2008) Lymphoepithelial cyst of the pancreas: a new case report. Gastroentérol Clin Biol 32(67):640–644PubMedCrossRef Roger N, Rousseau G, Bardier A, Vaillant JC, Hannoun L (2008) Lymphoepithelial cyst of the pancreas: a new case report. Gastroentérol Clin Biol 32(67):640–644PubMedCrossRef
29.
go back to reference Matsukawa H, Takakura N, Shiozaki S, Aoki H (2008) A case of Lymphoepithelial cyst of the pancreas. Nippon Shokakibyo Gakkai Zasshi 105(6):854–859PubMed Matsukawa H, Takakura N, Shiozaki S, Aoki H (2008) A case of Lymphoepithelial cyst of the pancreas. Nippon Shokakibyo Gakkai Zasshi 105(6):854–859PubMed
30.
go back to reference Kobayashi T, Shimura T, Araki K, Ogawa A, Mochida Y, Suzuki H et al (2008) Lymphoepithelial cyst of the pancreas: report of a case. Hepatogastroenterology 55(84):1107–1109PubMed Kobayashi T, Shimura T, Araki K, Ogawa A, Mochida Y, Suzuki H et al (2008) Lymphoepithelial cyst of the pancreas: report of a case. Hepatogastroenterology 55(84):1107–1109PubMed
31.
go back to reference Zielinska Pajak E, Liszka L, Pajak J, Golka D, Lampe P, Pilch-Kowalczyk J et al (2008) Two cases of lymphoepithelial cyst of the pancreas: first—a cyst coexisting with a primary gastric lymphoma, second—incidentally found pancreatic cyst. Z Gastroenterol 46(10):1188–1193PubMedCrossRef Zielinska Pajak E, Liszka L, Pajak J, Golka D, Lampe P, Pilch-Kowalczyk J et al (2008) Two cases of lymphoepithelial cyst of the pancreas: first—a cyst coexisting with a primary gastric lymphoma, second—incidentally found pancreatic cyst. Z Gastroenterol 46(10):1188–1193PubMedCrossRef
32.
go back to reference Yamaguchi T, Takahashi H, Kagawa R, Takeda R, Sakata S, Yamamoto M et al (2008) Lymphoepithelial cyst of the pancreas associated with elevated CA 19-9 levels. J Hepatobiliary Pancreat Surg 15(6):652–654PubMedCrossRef Yamaguchi T, Takahashi H, Kagawa R, Takeda R, Sakata S, Yamamoto M et al (2008) Lymphoepithelial cyst of the pancreas associated with elevated CA 19-9 levels. J Hepatobiliary Pancreat Surg 15(6):652–654PubMedCrossRef
33.
go back to reference Alvarez Catro AM, Molina-Perez E, Fernandez-Castrogudin J, Iglesian Garcia J, Bustamante Montalvo M, Dominguez Munoz E (2008) Lymphoepithelial cyst—a very uncommon cause of pancreatic mass. Rev Esp Enferm Dig 100(9):596–598 Alvarez Catro AM, Molina-Perez E, Fernandez-Castrogudin J, Iglesian Garcia J, Bustamante Montalvo M, Dominguez Munoz E (2008) Lymphoepithelial cyst—a very uncommon cause of pancreatic mass. Rev Esp Enferm Dig 100(9):596–598
34.
go back to reference Langan R, Paragi P, Guarino R, Chamberlain R (2009) Lymphoepithelial cyst of the pancreas: a sheep in wolves clothing. Am Surg 75(6):520–522PubMed Langan R, Paragi P, Guarino R, Chamberlain R (2009) Lymphoepithelial cyst of the pancreas: a sheep in wolves clothing. Am Surg 75(6):520–522PubMed
35.
go back to reference Scheiman JM (2008) Management of cystic lesions of the pancreas. J Gastroenterol Surg 12(3):405–407CrossRef Scheiman JM (2008) Management of cystic lesions of the pancreas. J Gastroenterol Surg 12(3):405–407CrossRef
36.
go back to reference Al-Haddad M, Raimondo M, Woodward T, Krishna M, Pungpapong S, Noh K, Wallace MB (2007) Safety and efficacy of cytology brushings versus standard FNA in evaluating cystic lesions of the pancreas: a pilot study. Gastrointest Endosc 65(6):894–898PubMedCrossRef Al-Haddad M, Raimondo M, Woodward T, Krishna M, Pungpapong S, Noh K, Wallace MB (2007) Safety and efficacy of cytology brushings versus standard FNA in evaluating cystic lesions of the pancreas: a pilot study. Gastrointest Endosc 65(6):894–898PubMedCrossRef
37.
go back to reference Cappellari JO (1993) Fine-needle aspiration cytology of a pancreatic lymphoepithelial cyst. Diagn Cytopathol 9:77–81PubMedCrossRef Cappellari JO (1993) Fine-needle aspiration cytology of a pancreatic lymphoepithelial cyst. Diagn Cytopathol 9:77–81PubMedCrossRef
38.
go back to reference Lopicarpio-Nicholas ML, Shami VM, Kahaleh M, Adams RB, Mallery S, Stanley MW et al (2006) Fine needle aspiration cytology of Lymphoepithelial cyst of the pancreas. Cancer 108(6):501–506CrossRef Lopicarpio-Nicholas ML, Shami VM, Kahaleh M, Adams RB, Mallery S, Stanley MW et al (2006) Fine needle aspiration cytology of Lymphoepithelial cyst of the pancreas. Cancer 108(6):501–506CrossRef
39.
go back to reference Centeno BA, Stockwell JW, Lewandrowski KB (1999) Cyst fluid cytology and chemical features in a case of lymphoepithelial cyst of the pancreas: a rare and difficult preoperative diagnosis. Diagn Cytopathol 21:328–330PubMedCrossRef Centeno BA, Stockwell JW, Lewandrowski KB (1999) Cyst fluid cytology and chemical features in a case of lymphoepithelial cyst of the pancreas: a rare and difficult preoperative diagnosis. Diagn Cytopathol 21:328–330PubMedCrossRef
40.
go back to reference Liu J, Shin HJ, Rubenchik I, Lang E, Lahoti S, Staerkel GA (1999) Cytologic features of lymphoepithelial cyst of the pancreas: two preoperatively diagnosed cases based on fine-needle aspiration. Diagn Cytopathol 21:346–350PubMedCrossRef Liu J, Shin HJ, Rubenchik I, Lang E, Lahoti S, Staerkel GA (1999) Cytologic features of lymphoepithelial cyst of the pancreas: two preoperatively diagnosed cases based on fine-needle aspiration. Diagn Cytopathol 21:346–350PubMedCrossRef
41.
go back to reference Mitchell ML (1990) Fine needle aspiration biopsy of peripancreatic lymphoepithelial cysts. Acta Cytol 34:462–463PubMed Mitchell ML (1990) Fine needle aspiration biopsy of peripancreatic lymphoepithelial cysts. Acta Cytol 34:462–463PubMed
42.
go back to reference Jian B, Kimbrell HZ, Sepulveda A, Yu G (2008) Lymphoepithelial cysts of the pancreas: endosonography-guided fine needle aspiration. Diagn Cytopathol 36(9):662–665PubMedCrossRef Jian B, Kimbrell HZ, Sepulveda A, Yu G (2008) Lymphoepithelial cysts of the pancreas: endosonography-guided fine needle aspiration. Diagn Cytopathol 36(9):662–665PubMedCrossRef
43.
go back to reference Frossard JL, Amouyal P, Amouyal G, Palazzo L, Amaris J, Soldan M, Giostra E, Spahr L, Hadengue A, Fabre M (2003) Performance of endosonography-guided fine needle aspiration and biopsy in the diagnosis of pancreatic cystic lesions. Am J Gastroenterol 98(7):1516–1524PubMedCrossRef Frossard JL, Amouyal P, Amouyal G, Palazzo L, Amaris J, Soldan M, Giostra E, Spahr L, Hadengue A, Fabre M (2003) Performance of endosonography-guided fine needle aspiration and biopsy in the diagnosis of pancreatic cystic lesions. Am J Gastroenterol 98(7):1516–1524PubMedCrossRef
44.
go back to reference Nasr J, Sanders M, Fasanella K, Khalid A, McGrath K (2008) Lymphoepithelial cyst of the pancreas: an EUS case series. Gastrointest Endosc 68(1):170–173PubMedCrossRef Nasr J, Sanders M, Fasanella K, Khalid A, McGrath K (2008) Lymphoepithelial cyst of the pancreas: an EUS case series. Gastrointest Endosc 68(1):170–173PubMedCrossRef
45.
go back to reference Attasaranya S, Pais S, LeBlanc J, McHenry L, Sherman S, DeWitt JM (2007) Endoscopic ultrasound-guided fine needle aspiration and cyst fluid analysis for pancreatic cysts. JOP 8(5):553–563PubMed Attasaranya S, Pais S, LeBlanc J, McHenry L, Sherman S, DeWitt JM (2007) Endoscopic ultrasound-guided fine needle aspiration and cyst fluid analysis for pancreatic cysts. JOP 8(5):553–563PubMed
46.
go back to reference Zhu LC, Grieco V (2008) Diagnostic value of unusual gross appearance of aspirated material from endoscopic ultrasound-guided fine needle aspiration of pancreatic and peripancreatic cystic lesions. Acta Cytol 52(5):535–540PubMedCrossRef Zhu LC, Grieco V (2008) Diagnostic value of unusual gross appearance of aspirated material from endoscopic ultrasound-guided fine needle aspiration of pancreatic and peripancreatic cystic lesions. Acta Cytol 52(5):535–540PubMedCrossRef
47.
go back to reference Vazquez-Sequeiros E (2007) Endoscopic ultrasound and fine needle aspiration in inflammatory and cystic pancreatic pathology. Minerva Med 98(4):357–360PubMed Vazquez-Sequeiros E (2007) Endoscopic ultrasound and fine needle aspiration in inflammatory and cystic pancreatic pathology. Minerva Med 98(4):357–360PubMed
48.
go back to reference Ahlawat SK (2008) Lymphoepithelial cyst of the pancreas: role of endoscopic ultrasound guided fine needle aspiration. JOP 9(2):230–234PubMed Ahlawat SK (2008) Lymphoepithelial cyst of the pancreas: role of endoscopic ultrasound guided fine needle aspiration. JOP 9(2):230–234PubMed
Metadata
Title
Lymphoepithelial Cyst of the Pancreas: A Rare Case Report and Review of Literature
Authors
Ajit Sewkani
Deepak Purohit
Vikrant Singh
Aruna Jain
Rajneesh Varshney
Subodh Varshney
Publication date
01-12-2010
Publisher
Springer-Verlag
Published in
Indian Journal of Surgery / Issue 6/2010
Print ISSN: 0972-2068
Electronic ISSN: 0973-9793
DOI
https://doi.org/10.1007/s12262-010-0152-y

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