Abstract
Ovarian Brenner tumor with abnormally increased serum carbohydrate antigen 19–9 (CA19-9) level is extremely rare. A 70-year-old woman with abnormally elevated serum CA199 (1289 U/ml) found in routine physical examination. Pelvic CT and MRI scan revealed a large mass with large patches of calcification in the right adnexal area, and the patient achieved total hysterectomy and bilateral adnexectomy. Grossly, the right ovary had a solid enlargement of about 7.0 cm × 6.0 cm × 5.0 cm with irregular nodules and smooth surface and the cut surface of the mass showed that the tumor is cystic and solid. Microscopically, the tumor showed a background of fibrous tissue hyperplasia with nested and adenoid cell clusters with uniform cell size and clear boundaries. The cells were translucent with eosinophilic cytoplasm and calcification. Immunohistochemical staining showed CK7, CA125, and P63 presented diffusely strongly positive staining, while negativity for CK20, GATA3, AR, P53, and CgA. Ki-67 showed weak positive staining, about 1%. The serum CA199 level decreased significantly on the 5th day after surgery. Postoperative pathology and immunohistochemistry confirmed borderline Brenner tumor. This is the first to report a case of borderline Brenner tumor with an abnormally high serum level of CA199 before surgery. In clinical practice, the possibility of ovarian Brenner tumor should be considered when abnormal elevation of serum CA199 level cannot be reasonably explained.
This is a preview of subscription content, log in via an institution to check access.
Similar content being viewed by others
Data availability
The datasets generated and analyzed during the current study are available from the corresponding author on reasonable request.
References
Tsai S, George B, Wittmann D et al (2018) Importance of normalization of CA19-9 levels following neoadjuvant therapy in patients with localized pancreatic cancer. Ann Surg 271(4):740–747
Koprowski H, Steplewski Z, Mitchell K et al (1979) Colorectal carcinoma antigens detected by hybridoma antibodies. Somatic Cell Genet 5:957–971
Koprowski H, Herlyn M, Steplewski Z et al (1981) Specific antigen in serum of patients with colon carcinoma. Science 3;212(4490):53–55
Scarà S, Bottoni P, Scatena R (2015) CA 19–9: Biochemical and clinical aspects. Adv Exp Med Biol 867:247–260
Ogawa T, Yokoi H, Kawarada Y (1998) A case of inflammatory pseudotumor of the liver causing elevated serum CA19-9 levels. Am J Gastroenterol 93(12):2551–2555
Waanders E, van Keimpema L, Brouwer JT et al (2009) Carbohydrate antigen 19–9 is extremely elevated in polycystic liver disease. Liver Int 29(9):1389–1395
Fahrmann JF, Schmidt CM, Mao X et al (2021) Lead-time trajectory of CA19-9 as an anchor marker for pancreatic cancer early detection. Gastroenterology 160(4):1373-1383.e6
Yang J, Xu R, Wang C et al (2021) Early screening and diagnosis strategies of pancreatic cancer: a comprehensive review. Cancer Commun 41:1257–1274
Zeng P, Li H, Chen Y et al (2019) Serum CA199 levels are significantly increased in patients suffering from liver, lung, and other diseases. Prog Mol Biol Transl Sci 162:253–264
Wannhoff A, Rupp C, Friedrich K et al (2015) Inflammation but not biliary obstruction is associated with carbohydrate antigen 19–9 levels in patients with primary sclerosing cholangitis. Clin Gastroenterol Hepatol 13:2372–2379
Ge L, Liu G, Hu K et al (2020) A new risk index combining d-dimer, fibrinogen, HE4, and CA199 differentiates suspecting endometrial cancer from patients with abnormal vaginal bleeding or discharge. Technol Cancer Res Treat 19:1533033819901117
Zhu C, Zhu J, Qian L et al (2021) Clinical characteristics and prognosis of ovarian clear cell carcinoma: a 10-year retrospective study. BMC Cancer 21(1):322
De Cecio R, Cantile M, Collina F et al (2014) Borderline Brenner tumor of the ovary: a case report with immunohistochemical and molecular study. J Ovarian Res 7:101
Zheng R, Heller DS (2019) Borderline Brenner tumor: a review of the literature. Arch Pathol Lab Med 143:1278–1280
Yoshida S, Shidoh M, Shimoya Y et al (2003) Brenner tumour. Lancet 362(9387):858
Kuhn E, Ayhan A, IeM S et al (2013) Ovarian Brenner tumour: a morphologic and immunohistochemical analysis suggesting an origin from fallopian tube epithelium. Eur J Cancer 49(18):3839–3849
Kuhn E, Ayhan A, IeM S et al (2014) The pathogenesis of atypical proliferative Brenner tumor: an immunohistochemical and molecular genetic analysis. Mod Pathol 27(2):231–237
Montoriol PF, Hordonneau C, Boudinaud C et al (2021) Benign Brenner tumour of the ovary: CT and MRI features. Clin Radiol 76(8):593–598
Moon WJ, Koh BH, Kim SK et al (2000) Brenner tumor of the ovary: CT and MR findings. J Comput Assist Tomogr 24(1):72–76
Salibay CJ, Zanfagnin V, Miller H et al (2021) Borderline Brenner Tumor of the ovary coexisting with an ovarian mucinous cystadenoma with focal atypical epithelial proliferation: a rare case with review of the literature. Int J Surg Pathol 29(7):788–793
Zhong PP, Zhu L, Zhang LH et al (2019) Clinicopathological features of ovarian Brenner tumors. Zhonghua Bing Li Xue Za Zhi 48(8):615–619. Chinese
Soslow RA, Rouse RV, Hendrickson MR et al (1996) Transitional cell neoplasms of the ovary and urinary bladder: a comparative immunohistochemical analysis. Int J Gynecol Pathol 15(3):257–265
Esheba GE, Longacre TA, Atkins KA et al (2009) Expression of the urothelial differentiation markers GATA3 and placental S100 (S100P) in female genital tract transitional cell proliferations. Am J Surg Pathol 33(3):347–353
Wannhoff A, Werner S, Tao S et al (2022) Validation of a genotype-based algorithm that identifies individuals with low, intermediate, and high serum CA19-9 levels in cancer-free individuals and in patients with colorectal cancer. J Gastrointest Oncol 13(4):1711–1721
Acknowledgements
All authors would like to thank Dr. Piao Wang (Department of Radiology, The Second Affiliated Hospital of Anhui Medical University) and Dr. Panpan Yang (Department of Pathology, the Second Affiliated Hospital of Anhui Medical University) for their help in this study.
Author information
Authors and Affiliations
Contributions
QY, QZ: acquisition of data, analysis and interpretation of data, drafting and revising the article, and final approval. YS, KX, and YL: acquisition of data, analysis and interpretation of the data, and final approval. LZ: critical revision of the article for important intellectual content and final approval. HF: conception and design of the study, critical revision of the article for important intellectual content, and final approval. All the authors have read and approved the final manuscript.
Corresponding author
Ethics declarations
Conflict of interest
The authors declare no competing interests.
Additional information
Publisher's note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Rights and permissions
Springer Nature or its licensor (e.g. a society or other partner) holds exclusive rights to this article under a publishing agreement with the author(s) or other rightsholder(s); author self-archiving of the accepted manuscript version of this article is solely governed by the terms of such publishing agreement and applicable law.
About this article
Cite this article
Yu, Q., Zhao, Q., Su, Y. et al. Borderline Brenner tumor with abnormally high serum level of carbohydrate antigen 199: a rare case report and literature review. Ir J Med Sci 192, 2071–2075 (2023). https://doi.org/10.1007/s11845-022-03203-7
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s11845-022-03203-7