A case of synovial sarcoma following etanercept use in a patient with rheumatoid arthritis

Dear Editor, New targeted therapies have been noted after illumination of the pathogenesis of rheumatic diseases particularly in rheumatoid arthritis (RA), recently. These are usually cytokine-based therapies, tumor necrosis factor alpha (TNF-a) blocker is one of them. Etanercept which is a soluble TNF-a surface receptor has been reported to be associated with various solid organ tumors development. Here in, we wanted to report a case of synovial sarcoma after 3-months of etanercept therapy in a patient with RA that has never been reported in literature, before. A 73-year-old male patient with active RA referred to our rheumatology clinic. He was diagnosed as RA five years ago. Firstly, there was involvement just only in hand joints, bilateral hip pain started to become for one year. FABERE and FADIR was positive on the right side where FABERE was found to be slightly positive on the left side in physical examination. There was no other joints of arthritis. Laboratory findings were determined as follows: erythrocyte sedimentation rate: 32 mm/h, CRP: 83 mg/L (0-6), RF: 128 IU/ml, anti-CCP(+). Leflunomide 20 mg/day, methotrexate 15 mg/week, sulfasalazine 4000 mg/day, prednisolone 5 mg/day and indomethacin 75 mg/day was commenced firstly. In order to to rule out avascular necrosis diagnosis hip MRI was taken. There was no pathological finding except minimal fluid in the joint range. Since DAS 28 score was 5.17, subcutaneous etanercept 25 mg twice weekly was commenced. Pretreatment PPD was determined as 0 mm two times. After chest diseases consultation isoniazid treatment was planned for nine months for tuberculosis prophylaxis. In the third month of treatment a solid mass of 35x30 mm in size 10-12 cm below the knee was determined. Newly diagnosed that immobile mass was painless and fixed under muscle tissue. MR imaging of the right cruris region demonstrated that solid mass had showed malignant character and invaded surrounding tissues (Figure-1). Excisional biopsy of the mass lesion revealed the diagnosis of synovial sarcoma grade III. The most important side effect of TNF-a blockers is development of malignancy. Up to date solid and hematological cancers have been reported in many patients given anti-TNF therapy (1-3). However it had not been understood thoroughly as if it developed due to treatment or due to course of the primary disease or it was just a coincidence. Recently there are some studies reporting that anti-TNF therapy didn t increase the risk of cancer. Lombardy Rheumatology Network (LORHEN) registry is one of them (4). In general, hematologic malignancies such as lymphoma is much more seen than solid organ tumors in anti-TNF therapy receiving patients. In our case the mass in the right cruris appeared in a short period of time such as three months after anti-TNF theraphy, etanercept is thought to be as responsible factor. This is the first case report of synovial sarcoma development due to etanercept use in literature

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  • 1. Shahab N, Doll DC. Testicular Lymphoma. Semin Oncol 1999;26(3):259-269
  • 2. Moller MB, d’Amore F, Christensen BE. Testicular lymphoma: a population-based study of incidence, clinicopathological correlations and prognosis. The Danish Lymphoma Study Group, LYFO. Eur J Cancer 1994;30A(12):1760-1764
  • 3. Eble JN, Sauter G, Epstein JI, Sesterhenn IA. Tumours of the Testis and Paratesticular Tissue . In: World Health Organization Classification of Tumours. Pathology and Genetics of Tumours of the Urinary System and Male Genital Organs Lyon, IARC Press, 2004; 217-279.
  • 4. Ferry JA, Harris NL, Young RH, Coen J, Zietman A, Scully RE. Malignant lymphoma of the testis, epididymis, and spermatic cord. A clinicopathologic study of 69 cases with immunophenotypic analysis. Am J Surg Pathol 1994;18(4):376-390
  • 5. Vural F, Cagirgan S, Saydam G, Hekimgil M, Soyer NA, Tombuloglu M. Primary testicular lymphoma. J Natl Med Assoc 2007;99(11):1277-1282
  • 6. Fonseca R, Habermann TM, Colgan JP, O'Neill BP, White WL, Witzig TE, et al. Testicular lymphoma is associated with a high incidence of extranodal recurrence. Cancer 2000;88(1):154-161
  • 7. Sampat MB, Sirsat MV, Kamat MR. Malignant lymphoma of the testis in Indians. Br J Urol 1974;46(5):569-575
  • 8. Zicherman JM, Weissman D, Gribbin C, Epstein R. Best cases from the AFIP: primary diffuse large B-cell lymphoma of the epididymis and testis. Radiographics 2005;25(1):243-248
  • 9. Mazloom A, Fowler N, Medeiros LJ, Iyengar P, Horace P, Dabaja BS. Outcome of patients with diffuse large B-cell lymphoma of the testis by era of treatment: the M. D. Anderson Cancer Center experience. Leuk Lymphoma 2010;51(7):1217-1224
  • 10. Mlika M, Chelly I, Benrhouma M, Haouet S, Horchani A, Zitouna MM, et al. A Primary Testicular Diffuse Large B-cell Lymphoma Belonging to the Germinal Center B-cell-like Group. J Clin Med Res. 2010;2(1):47- 49
  • 11. Curling TB. Practical Treatise on the diseases of the testis and of the spermatic cord and scrotum. London, Churchill 1866;605
  • 12. Zucca E, Conconi A, Mughal TI, Sarris AH, Seymour JF, Vitolo U, et al. Patterns of outcome and prognostic factors in primary large-cell lymphoma of the testis in a survey by the International Extranodal Lymphoma Study Group. J Clin Oncol 2003;21(1):20- 27
  • 13. Gundrum JD, Mathiason MA, Moore DB, Go RS. Primary testicular diffuse large B-cell lymphoma: a population-based study on the incidence, natural history, and survival comparison with primary nodal counterpart before and after the introduction of rituximab. J Clin Oncol 2009;27(31):5227-5232
  • 14. Doll DC, Weiss RB. Malignant lymphoma of the testis. Am J Med 1986;81(3):515524
  • 15. Li D, Xie P, Mi C. Primary testicular diffuse large Bcell lymphoma shows an activated B-cell-like phenotype. Pathol Res Pract 2010;206(9):611-615
  • 16. Kemmerling R, Stintzing S, Muhlmann J, Dietze O, Neureiter D. Primary testicular lymphoma: a strictly homogeneous hematological disease? Oncol Rep 2010;23(5):1261-1267
  • 17. Booman M, Douwes J, Glas AM, de Jong D, Schuuring E, Kluin PM. Primary testicular diffuse large B-cell lymphomas have activated B-cell-like subtype characteristics. J Pathol 2006;210(2):163-171
  • 18. Wang Y, Li ZM, Huang JJ, Xia Y, Li H, Li YJ, et al. Three prognostic factors influence clinical outcomes of primary testicular lymphoma. 2013;34(1):55-63
  • 19. Licci S, Morelli L, Covello R. Primary mantle cell lymphoma of the testis. Ann Hematol 2011;90(4):483- 484
  • 20. Sugimoto K, Koike H, Esa A. Plasmablastic lymphoma of the right testis. Int J Urol 2011;18(1):85-86
  • 21. Moertel CL, Watterson J, McCormick SR, Simonton SC. Follicular large cell lymphoma of the testis in a child. Cancer 1995;75(5):1182-1186
  • 22. Horne MJ, Adeniran AJ. Arch Pathol Lab Med. 2011;135(10):1363-1367
  • 23. Ulbright TM. The most common, clinically significant misdiagnoses in testicular tumor pathology, and how to avoid them. Adv Anat Pathol 2008;15(1):1827
  • 24. Sussman EB, Hajdu SI, Lieberman PH, Whitmore WF. Malignant lymphoma of the testis: a clinicopathologic study of 37 cases. J Urol 1977;118(6):10041007
  • 25. Moller MB, d’Amore F, Christensen BE. Testicular lymphoma: a population based study of incidence, clinicopathological correlations and prognosis. The Danish Lymphoma Study Group, LYFO. Eur J Cancer 1994;30A(12):17601764
  • 26. Bhatia K, Vaid AK, Gupta S, Doval DC, Talwar V. Primary testicular non-Hodgkin’s lymphoma-a review article. Sao Paulo Med J 2007;125(5):286-288
  • 27. Darby S, Hancock BW. Localised non-Hodgkin lymphoma of the testis: the Sheffield Lymphoma Group experience. Int J Oncol 2005;26(4):1093-1099
  • 28. Miller TP, Dahlberg S, Cassady JR, Adelstein DJ, Spier CM, Grogan TM, et al. Chemotherapy alone compared with chemotherapy plus radiotherapy for localized intermediate- and high-grade non-Hodgkin’s lymphoma. N Engl J Med 1998;339(1):21-26
  • 29. Reyes F, Lepage E, Ganem G, Molina TJ, Brice P, Coiffier B, et al. ACVBP versus CHOP plus radiotherapy for localized aggressive lymphoma. N Engl J Med. 2005;352:11971205
  • 30. Ergun O, Kosar A, Bas E, Bircan S, Alanoglu EG. Primer testis lenfomasy: bir olgu sunumu ve rituximab ile kombine kemoterapinin etkinli?i. Turkiye Klinikleri J Med Sci 2011;31(3):748-751
Süleyman Demirel Üniversitesi Tıp Fakültesi Dergisi-Cover
  • ISSN: 1300-7416
  • Yayın Aralığı: Yılda 4 Sayı
  • Başlangıç: 1994
  • Yayıncı: SDÜ Basımevi / Isparta