Autopsy and Case Reports
https://autopsyandcasereports.org/article/doi/10.4322/acr.2021.369
Autopsy and Case Reports
Clinical Case Report and Review

Epidermoid cyst in an intrapancreatic accessory spleen with abnormally high CEA level in cyst fluid: a case report

Chun-hai Lo; Po-man Tsang; Shui-ying Cheng; Cheuk-nam Ling; Cheuk-lam Ho

Downloads: 2
Views: 244

Abstract

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.

Keywords

Carcinoembryonic Antigen, Epidermal Cyst, Pancreas, Spleen

References

1 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.

2 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.

3 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.

4 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.

5 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.

6 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.

7 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.

8 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.

9 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.

10 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.

11 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.

12 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.

13 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.

14 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.

15 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.

16 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.

17 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.

18 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.

19 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.

20 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.

21 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.

22 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.

23 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.

24 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.

25 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.

26 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.

27 Zhang Z, Wang JC. An epithelial splenic cyst in an intrapancreatic accessory spleen. A case report. JOP. 2009;10(6):664-6. PMid:19890189.

28 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.

29 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.

30 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.

31 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.

32 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.

33 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.

34 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.

35 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.

36 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.

37 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.

38 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.

39 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.

40 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.

41 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.

42 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.

43 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.

44 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.

45 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.

46 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.

47 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.

48 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.

49 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.

50 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.

51 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.

52 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.

53 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.

54 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.

55 Ramsden KL, Newman J. Lymphoepithelial cyst of the pancreas. Histopathology. 1991;18(3):267-8.

56 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.

57 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.
 


Submitted date:
12/07/2021

Accepted date:
02/14/2022

Publication date:
04/14/2022

625836cea9539572f510a3e3 autopsy Articles
Links & Downloads

Autops Case Rep

Share this page
Page Sections