Epidermoid cyst in an intrapancreatic accessory spleen with abnormally high CEA level in cyst fluid: a case report
DOI:
https://doi.org/10.4322/acr.2021.369Keywords:
Carcinoembryonic Antigen, Epidermal Cyst, Pancreas, SpleenAbstract
Epidermoid cyst in an intrapancreatic accessory spleen is a rare benign lesion that is difficult to diagnose preoperatively. Cyst fluid analysis for biochemistry markers has been widely used to aid the diagnosis of pancreatic cysts. A high cyst fluid carcinoembryonic antigen (CEA) level (>800 ng/mL) is said to be useful in distinguishing intraductal papillary mucinous neoplasm (IPMN) and mucinous cystic neoplasm (MCN) from other non-mucinous cysts. We herein report a case of epidermoid cyst in an intrapancreatic accessory spleen with abnormally high CEA level (3582 ng/mL) in the cyst fluid, suggesting a potential pitfall in using cyst fluid CEA level as an indicator of mucinous neoplasms.
Downloads
References
Vikse J, Sanna B, Henry BM, et al. The prevalence and morphometry of an accessory spleen: a meta-analysis and systematic review of 22,487 patients. Int J Surg. 2017;45:18-28. http://dx.doi.org/10.1016/j.ijsu.2017.07.045. PMid:28716661.
Halpert B, Alden ZA. Accessory spleens in or at the tail of the pancreas. A survey of 2,700 additional necropsies. Arch Pathol. 1964;77:652-4. PMid:14130052.
Rana AP, Kaur M, Singh P, Malhotra S, Kuka AS. Splenic epidermoid cyst - a rare entity. J Clin Diagn Res. 2014;8(2):175-6. PMid:24701525.
Davidson ED, Campbell WG, Hersh T. Epidermoid splenic cyst occurring in an intrapancreatic accessory spleen. Dig Dis Sci. 1980;25(12):964-7. http://dx.doi.org/10.1007/BF01308048. PMid:7449592.
Hanada M, Kimura M, Kitada M, Nakajima T, Yamada K, Yoshii M. Epidermoid cyst of accessory spleen. Acta Pathol Jpn. 1981;31(5):863-72. PMid:7304174.
Nakae Y, Hayakawa T, Kondo T, et al. Epidermoid cyst occurring in a pancreatic accessory spleen. J Clin Gastroenterol. 1991;13(3):362-4. http://dx.doi.org/10.1097/00004836-199106000-00024. PMid:2066557.
Morohoshi T, Hamamoto T, Kunimura T, et al. Epidermoid cyst derived from an accessory spleen in the pancreas. A case report with literature survey. Acta Pathol Jpn. 1991;41(12):916-21. PMid:1785350.
Tang X, Tanaka Y, Tsutsumi Y. Epithelial inclusion cysts in an intrapancreatic accessory spleen. Pathol Int. 1994;44(8):652-4. http://dx.doi.org/10.1111/j.1440-1827.1994.tb01726.x. PMid:7952152.
Higaki K, Jimi A, Watanabe J, Kusaba A, Kojiro M. Epidermoid cyst of the spleen with CA19-9 or carcinoembryonic antigen productions: report of three cases. Am J Surg Pathol. 1998;22(6):704-8. http://dx.doi.org/10.1097/00000478-199806000-00007. PMid:9630177.
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;11(12):1171-7. PMid:9872647.
Furukawa H, Kosuge T, Kanai Y, Mukai K. Epidermoid cyst in an intrapancreatic accessory spleen: CT and pathologic findings. AJR Am J Roentgenol. 1998;171(1):271. http://dx.doi.org/10.2214/ajr.171.1.9648813. PMid:9648813.
Sasou S, Nakamura S, Inomata M. Epithelial splenic cysts in an intrapancreatic accessory spleen and spleen. Pathol Int. 1999;49(12):1078-83. http://dx.doi.org/10.1046/j.1440-1827.1999.00983.x. PMid:10632928.
Tsutsumi S, Kojima T, Fukai Y, et al. Epidermoid cyst of an intrapancreatic accessory spleen--a case report. Hepatogastroenterology. 2000;47(35):1462-4. PMid:11100377.
Choi SK, Ahn SI, Hong KC, et al. A case of epidermoid cyst of the intrapancreatic accessory spleen. J Korean Med Sci. 2000;15(5):589-92. http://dx.doi.org/10.3346/jkms.2000.15.5.589. PMid:11068999.
Horibe Y, Murakami M, Yamao K, Imaeda Y, Tashiro K, Kasahara M. Epithelial inclusion cyst (epidermoid cyst) formation with epithelioid cell granuloma in an intrapancreatic accessory spleen. Pathol Int. 2001;51(1):50-4. http://dx.doi.org/10.1046/j.1440-1827.2001.01155.x. PMid:11148465.
Fink AM, Kulkarni S, Crowley P, Crameri JA. Epidermoid cyst in a pancreatic accessory spleen mimicking an infected abdominal cyst in a child. AJR Am J Roentgenol. 2002;179(1):206-8. http://dx.doi.org/10.2214/ajr.179.1.1790206. PMid:12076937.
Yokomizo H, Hifumi M, Yamane T, et al. Epidermoid cyst of an accessory spleen at the pancreatic tail: diagnostic value of MRI. Abdom Imaging. 2002;27(5):557-9. http://dx.doi.org/10.1007/s00261-001-0055-2. PMid:12172997.
Sonomura T, Kataoka S, Chikugo T, et al. Epidermoid cyst originating from an intrapancreatic accessory spleen. Abdom Imaging. 2002;27(5):560-2. http://dx.doi.org/10.1007/s00261-001-0145-1. PMid:12172998.
Watanabe H, Yamaguchi Y, Ohtsubo K, et al. Epidermoid cyst of the intrapancreatic accessory spleen producing CA19-9. Dig Endosc. 2004;16(3):244-8. http://dx.doi.org/10.1111/j.1443-1661.2004.00347.x.
Kanazawa H, Kamiya J, Nagino M, et al. Epidermoid cyst in an intrapancreatic accessory spleen: a case report. J Hepatobiliary Pancreat Surg. 2004;11(1):61-3. http://dx.doi.org/10.1007/s00534-003-0844-9. PMid:15754048.
Won JK, Lee YJ, Kang GH. Epithelial cyst in the intrapancreatic accessory spleen that clinically mimic pancreatic cystic tumor. Korean J Pathol. 2005;39:437-41.
Ru K, Kalra A, Ucci A. Epidermoid cyst of intrapancreatic accessory spleen. Dig Dis Sci. 2007;52(5):1229-32. http://dx.doi.org/10.1007/s10620-006-9376-x. PMid:17385039.
Itano O, Shiraga N, Kouta E, et al. Epidermoid cyst originating from an intrapancreatic accessory spleen. J Hepatobiliary Pancreat Surg. 2008;15(4):436-9. http://dx.doi.org/10.1007/s00534-007-1243-4. PMid:18670847.
Servais EL, Sarkaria IS, Solomon GJ, Gumpeni P, Lieberman MD. Giant epidermoid cyst within an intrapancreatic accessory spleen mimicking a cystic neoplasm of the pancreas: case report and review of the literature. Pancreas. 2008;36(1):98-100. http://dx.doi.org/10.1097/MPA.0b013e3181359e36. PMid:18192891.
Gleeson FC, Kendrick ML, Chari ST, Zhang L, Levy MJ. Epidermoid accessory splenic cyst masquerading as a pancreatic mucinous cystic neoplasm. Endoscopy. 2008;40(Suppl 2):E141-2. http://dx.doi.org/10.1055/s-2007-995735. PMid:18633876.
Reiss G, Sickel JZ, See-Tho K, Ramrakhiani S. Intrapancreatic splenic cyst mimicking pancreatic cystic neoplasm diagnosed by EUS-FNA. Gastrointest Endosc. 2009;70(3):557-8, discussion 558. http://dx.doi.org/10.1016/j.gie.2009.04.050. PMid:19608182.
Zhang Z, Wang JC. An epithelial splenic cyst in an intrapancreatic accessory spleen. A case report. JOP. 2009;10(6):664-6. PMid:19890189.
Itano O, Chiba N, Wada T, et al. Laparoscopic resection of an epidermoid cyst originating from an intrapancreatic accessory spleen: report of a case. Surg Today. 2010;40(1):72-5. http://dx.doi.org/10.1007/s00595-009-4006-9. PMid:20037845.
Motosugi U, Yamaguchi H, Ichikawa T, et al. Epidermoid cyst in intrapancreatic accessory spleen: radiological findings including superparamagnetic iron oxide-enhanced magnetic resonance imaging. J Comput Assist Tomogr. 2010;34(2):217-22. http://dx.doi.org/10.1097/RCT.0b013e3181c1b2bd. PMid:20351508.
Kadota K, Kushida Y, Miyai Y, et al. Epidermoid cyst in an intrapancreatic accessory spleen: three case reports and review of the literatures. Pathol Oncol Res. 2010;16(3):435-42. http://dx.doi.org/10.1007/s12253-009-9229-y. PMid:19949910.
Iwasaki Y, Tagaya N, Nakagawa A, et al. Laparoscopic resection of epidermoid cyst arising from an intrapancreatic accessory spleen: a case report with a review of the literature. Surg Laparosc Endosc Percutan Tech. 2011;21(5):e275-9. http://dx.doi.org/10.1097/SLE.0b013e31822dd14a. PMid:22002295.
Horn AJ, Lele SM. Epidermoid cyst occurring within an intrapancreatic accessory spleen. A case report and review of the literature. JOP. 2011;12(3):279-82. PMid:21546709.
Urakami A, Yoshida K, Hirabayashi Y, et al. Laparoscopy-assisted spleen-preserving pancreatic resection for epidermoid cyst in an intrapancreatic accessory spleen. Asian J Endosc Surg. 2011;4(4):185-8. http://dx.doi.org/10.1111/j.1758-5910.2011.00102.x. PMid:22776306.
Yamanishi H, Kumagi T, Yokota T, et al. Epithelial cyst arising in an intrapancreatic accessory spleen: a diagnostic dilemma. Intern Med. 2011;50(18):1947-52. http://dx.doi.org/10.2169/internalmedicine.50.5340. PMid:21921374.
Khashab MA, Canto MI, Singh VK, Hruban RH, Makary MA, Giday S. Endosonographic and elastographic features of a rare epidermoid cyst of an intrapancreatic accessory spleen. Endoscopy. 2011;43(Suppl 2):E193-4. http://dx.doi.org/10.1055/s-0030-1256272. PMid:21590599.
Hu S, Zhu L, Song Q, Chen K. Epidermoid cyst in intrapancreatic accessory spleen: computed tomography findings and clinical manifestation. Abdom Imaging. 2012;37(5):828-33. http://dx.doi.org/10.1007/s00261-012-9851-0. PMid:22327420.
Harris AC, Chaudry MA, Menzies D, Conn PC. Laparoscopic resection of an epidermoid cyst within an intrapancreatic accessory spleen: a case report and review article. Surg Laparosc Endosc Percutan Tech. 2012;22(4):e246-9. http://dx.doi.org/10.1097/SLE.0b013e31825b3761. PMid:22874714.
Hong R, Choi N, Sun K, Lim S, Han Y. Epidermoid cyst arising from an intrapancreatic accessory spleen: A case report and review of the literature. Oncol Lett. 2013;5(2):469-72. http://dx.doi.org/10.3892/ol.2012.1061. PMid:23420784.
Zavras N, Machairas N, Foukas P, Lazaris A, Patapis P, Machairas A. Epidermoid cyst of an intrapancreatic accessory spleen: a case report and literature review. World J Surg Oncol. 2014;12(1):92. http://dx.doi.org/10.1186/1477-7819-12-92. PMid:24721745.
Kato S, Mori H, Zakimi M, et al. Epidermoid cyst in an intrapancreatic accessory spleen: case report and literature review of the preoperative imaging findings. Intern Med. 2016;55(23):3445-52. http://dx.doi.org/10.2169/internalmedicine.55.7140. PMid:27904107.
Kwak MK, Lee NK, Kim S, et al. A case of epidermoid cyst in an intrapancreatic accessory spleen mimicking pancreas neoplasms: MRI with DWI. Clin Imaging. 2016;40(1):164-6. http://dx.doi.org/10.1016/j.clinimag.2015.09.004. PMid:26422768.
Ko HJ, Shim JR, Lee TB, et al. Epidermoid cyst in an intrapancreatic accessory spleen in the pancreas head: a case report. BMC Gastroenterol. 2020;20(1):392. http://dx.doi.org/10.1186/s12876-020-01540-4. PMid:33218300.
Takagi C, Hoshi N, Kikuchi Y, et al. Epidermoid cyst within an intrapancreatic accessory spleen exhibiting abrupt changes in serum carbohydrate antigen 19-9 level: a case report. Surg Case Rep. 2020;6(1):133. http://dx.doi.org/10.1186/s40792-020-00892-z. PMid:32533275.
Park MS, Kim KW, Lim JS, et al. Unusual cystic neoplasms in the pancreas: radiologic-pathologic correlation. J Comput Assist Tomogr. 2005;29(5):610-6. http://dx.doi.org/10.1097/01.rct.0000172561.62810.6c. PMid:16163029.
Oh HC, Kang H, Brugge WR. Cyst fluid amylase and CEA levels in the differential diagnosis of pancreatic cysts: a single-center experience with histologically proven cysts. Dig Dis Sci. 2014;59(12):3111-6. http://dx.doi.org/10.1007/s10620-014-3254-8. PMid:24965184.
van der Waaij LA, van Dullemen HM, Porte RJ. Cyst fluid analysis in the differential diagnosis of pancreatic cystic lesions: a pooled analysis. Gastrointest Endosc. 2005;62(3):383-9. http://dx.doi.org/10.1016/S0016-5107(05)01581-6. PMid:16111956.
Bürrig KF. Epithelial (true) splenic cysts. Pathogenesis of the mesothelial and so-called epidermoid cyst of the spleen. Am J Surg Pathol. 1988;12(4):275-81. PMid:3354753.
Cianci P, Tartaglia N, Altamura A, et al. A recurrent epidermoid cyst of the spleen: report of a case and literature review. World J Surg Oncol. 2016;14(1):98. http://dx.doi.org/10.1186/s12957-016-0857-x. PMid:27036391.
Vo QD, Monnard E, Hoogewoud HM. Epidermoid cyst of the spleen. BMJ Case Rep. 2013;2013:bcr2013009707. http://dx.doi.org/10.1136/bcr-2013-009707. PMid:23667225.
Brugge WR, Lewandrowski K, Lee-Lewandrowski E, et al. Diagnosis of pancreatic cystic neoplasms: a report of the cooperative pancreatic cyst study. Gastroenterology. 2004;126(5):1330-6. http://dx.doi.org/10.1053/j.gastro.2004.02.013. PMid:15131794.
Cizginer S, Turner B, Bilge AR, Karaca C, Pitman MB, Brugge WR. Cyst fluid carcinoembryonic antigen is an accurate diagnostic marker of pancreatic mucinous cysts. Pancreas. 2011;40(7):1024-8. http://dx.doi.org/10.1097/MPA.0b013e31821bd62f. PMid:21775920.
Lewandrowski KB, Southern JF, Pins MR, Compton CC, Warshaw AL. Cyst fluid analysis in the differential diagnosis of pancreatic cysts. A comparison of pseudocysts, serous cystadenomas, mucinous cystic neoplasms, and mucinous cystadenocarcinoma. Ann Surg. 1993;217(1):41-7. http://dx.doi.org/10.1097/00000658-199301000-00008. PMID: 8424699.
Sand JA, Hyoty MK, Mattila J, Dagorn JC, Nordback IH. Clinical assessment compared with cyst fluid analysis in the differential diagnosis of cystic lesions in the pancreas. Surgery. 1996;119(3):275-80. http://dx.doi.org/10.1016/S0039-6060(96)80113-9. PMid:8619182.
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(1):56-65. PMid:10721807.
Ramsden KL, Newman J. Lymphoepithelial cyst of the pancreas. Histopathology. 1991;18(3):267-8.
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(1):27-32. http://dx.doi.org/10.1016/S0002-9610(99)80247-5. PMid:7793490.
Othman M, Basturk O, Groisman G, Krasinskas A, Adsay NV. Squamoid cyst of pancreatic ducts: a distinct type of cystic lesion in the pancreas. Am J Surg Pathol. 2007;31(2):291-7. http://dx.doi.org/10.1097/01.pas.0000213349.42143.ec. PMid:17255775.
Downloads
Published
Issue
Section
License
Copyright (c) 2022 Autopsy and Case Reports
This work is licensed under a Creative Commons Attribution 4.0 International License.
Copyright
Authors of articles published by Autopsy and Case Report retain the copyright of their work without restrictions, licensing it under the Creative Commons Attribution License - CC-BY, which allows articles to be re-used and re-distributed without restriction, as long as the original work is correctly cited.
