Yonsei Med J.  2013 Jul;54(4):895-901. 10.3349/ymj.2013.54.4.895.

Morbidity of Solid Cancer in Behcet's Disease: Analysis of 11 Cases in a Series of 506 Patients

Affiliations
  • 1Department of Dermatology, Ajou University School of Medicine, Suwon, Korea. esl@ajou.ac.kr

Abstract

PURPOSE
Behcet's disease (BD) is rarely reported to be associated with malignancies in the literature. However, the frequency of cancer in BD patients remains unknown. This study evaluated cancer morbidity in BD patients compared with that in the general population of Korea.
MATERIALS AND METHODS
A retrospective chart review was performed on 506 patients visiting our hospital from 1994 to 2011 for BD. We analyzed the standardized morbidity rate (SMR), which is the ratio of observed to expected malignancies. Furthermore, we reviewed cases of solid cancer in BD patients in the literature.
RESULTS
Of the 506 patients with BD, 11 (2.17%) developed cancer. We found a variety of solid cancers without predominance and no hematologic malignancies. The total number of cancers observed was less than expected, which was determined from the statistical data of the National Cancer Information Center of Korea, with an SMR of 0.023 (95% confidence interval, 0.012-0.039).
CONCLUSION
BD may be associated with a lower cancer-related morbidity compared with the general population of Korea.

Keyword

Behcet's disease; malignancy; morbidity; solid cancer

MeSH Terms

Adult
Behcet Syndrome/*complications/drug therapy/epidemiology
Female
Follow-Up Studies
Humans
Male
Middle Aged
Neoplasms/*epidemiology/etiology/surgery
Republic of Korea/epidemiology
Retrospective Studies

Reference

1. Sakane T, Takeno M, Suzuki N, Inaba G. Behçet's disease. N Engl J Med. 1999; 341:1284–1291.
Article
2. Direskeneli H. Autoimmunity vs autoinflammation in Behcet's disease: do we oversimplify a complex disorder? Rheumatology (Oxford). 2006; 45:1461–1465.
Article
3. Pankhurst T, Savage CO, Gordon C, Harper L. Malignancy is increased in ANCA-associated vasculitis. Rheumatology (Oxford). 2004; 43:1532–1535.
Article
4. Szekanecz Z, Szekanecz E, Bakó G, Shoenfeld Y. Malignancies in autoimmune rheumatic diseases - a mini-review. Gerontology. 2011; 57:3–10.
Article
5. Liang JA, Sun LM, Yeh JJ, Lin WY, Chang SN, Sung HC, et al. Malignancies associated with systemic lupus erythematosus in Taiwan: a nationwide population-based cohort study. Rheumatol Int. 2012; 32:773–778.
Article
6. Abu-Shakra M, Guillemin F, Lee P. Cancer in systemic sclerosis. Arthritis Rheum. 1993; 36:460–464.
Article
7. So MW, Koo BS, Kim YG, Lee CK, Yoo B. Idiopathic inflammatory myopathy associated with malignancy: a retrospective cohort of 151 Korean patients with dermatomyositis and polymyositis. J Rheumatol. 2011; 38:2432–2435.
Article
8. Kaklamani VG, Tzonou A, Kaklamanis PG. Behçet's disease associated with malignancies. Report of two cases and review of the literature. Clin Exp Rheumatol. 2005; 23:4 Suppl 38. S35–S41.
9. International Study Group for Behçet's Disease. Criteria for diagnosis of Behçet's disease. Lancet. 1990; 335:1078–1080.
10. Mizushima Y. [Revised diagnostic criteria for Behçet's disease in 1987]. Ryumachi. 1988; 28:66–70.
11. Tagami H, Makimoto K, Yamazaki M, Kodera M. [Unusual case of malignant pharyngeal granuloma--with symptoms resembling Behcet's syndrome]. Hifuka Kiyo. 1970; 65:253–261.
12. Tamaoki N, Habu S, Yoshimatsu H, Tsuchiya M, Watanabe H. Thymic change in Behçet's disease. Keio J Med. 1972; 21:201–213.
Article
13. Hamza MH. Cancer complicating Behçet's disease treated with chlorambucil. Ann Rheum Dis. 1986; 45:789.
14. Kamata T, Yamaguti A, Fusida S, Ishida T, Katou M, Sekino H, et al. A case report of the development of intestinal Behcet disease immediately after sigmoidectomy for sigmoid cancer. Jpn J Gastroenterol Surg. 1988; 21:2331–2334.
Article
15. Oishi S, Koga B, Sasaki M, Umeda T, Sato T. Pheochromocytoma associated with Behçet's disease. Jpn J Clin Oncol. 1989; 19:283–286.
16. Murata I, Omagari K, Nishihata S, Kamiya T, Imanishi T, Tanaka Y, et al. [A case of Behçet disease associated with hepatocellular carcinoma]. Gan No Rinsho. 1989; 35:625–631.
17. Muramatsu M, Kotake S, Yoshikawa K, Sasamoto Y, Matsuda H, Yamawaki S. The development of malignant rhabdoid tumor in a patient with Behçet's disease treated with ciclosporin. Graefes Arch Clin Exp Ophthalmol. 1998; 236:798–799.
Article
18. Bethea L, Khan S. Behçet's disease and pheochromocytoma. J S C Med Assoc. 1999; 95:295–298.
19. Celik I, Altundağ K, Erman M, Baltali E. Cyclophosphamide-associated carcinoma of the urinary bladder in Behçet's disease. Nephron. 1999; 81:239.
Article
20. Akpolat T, Yildiz L, Akpolat I, Bekir Selcuk M, Ozbek N, Yücel I. Small-cell carcinoma as a cause of superior vena cava syndrome in a patient with Behçet's disease. Respiration. 2000; 67:593.
Article
21. Kötter I, Aepinus C, Graepler F, Gärtner V, Eckstein AK, Stübiger N, et al. HHV8 associated Kaposi's sarcoma during triple immunosuppressive treatment with cyclosporin A, azathioprine, and prednisolone for ocular Behçet's disease and complete remission of both disorders with interferon alpha. Ann Rheum Dis. 2001; 60:83–86.
22. Baltaci S, Gögüs C, Karamürsel T, Tulunay O. Invasive bladder carcinoma in a patient with Behçet's disease. Int J Urol. 2003; 10:669–671.
Article
23. Nishimura K, Fujiki R, Hirotsu J, Ueda T, Nakamura E, Yoshidomi M, et al. A case of hilar bile duct cancer with intestinal Behçet's disease. Kurume Med J. 2004; 51:169–173.
24. Satolli F, Venturi C, Vescovi V, Morrone P, De Panfilis G. Merkel-cell carcinoma in Behçet's disease. Acta Derm Venereol. 2005; 85:79.
Article
25. Kwon KM, Jang BK, Chung WJ, Park KS, Cho KB, Hwang JS, et al. [A case of primary hepatic leiomyosarcoma with intrahepatic and abdominal subcutaneous metastasis in Behcet's disease]. Korean J Hepatol. 2005; 11:386–391.
26. Lee JE, Sohn JW, Lee KH, Park YS, Kim KH, Choi JW, et al. Colon Cancer in Behcet's Disease. Yeungnam Univ J Med. 2006; 23:124–130.
27. Kammori M, Tsuji E, Ogawa T, Takayoshi N, Kurabayashi R, Takubo K, et al. The pathological findings of vasculitis simultaneously occurring with carcinoma, invasive breast carcinoma in a patient with Behçet's disease. Breast Cancer. 2006; 13:378–381.
Article
28. Mezalek ZT, Harmouche H, Attar NE, Serraj K, Aouni M, Adnaoui M, et al. Kaposi's sarcoma in association with Behcet's disease: case report and literature review. Semin Arthritis Rheum. 2007; 36:328–331.
Article
29. Meyer J, Wahidi M, Shofer S, Evans J, Crawford J, Kelsey CR. Formation of a bronchoesophageal fistula following concurrent radiation and chemotherapy for lung cancer in the setting of Behçet's disease. J Thorac Oncol. 2008; 3:1361–1362.
Article
30. Chargari C, Kirova YM, Fourquet A, Campana F. Severe acute radiation-related skin toxicity in a breast cancer patient with Behçet's disease. Radiother Oncol. 2009; 91:139.
Article
31. Yamada M, Shiroeda H, Nomura T, Hayashi R, Sato K, Tsutsumi M, et al. Colon cancer arising in an ulcer scar due to intestinal Behçet's disease. Intern Med. 2011; 50:429–432.
32. Ahn JK, Oh JM, Lee J, Koh EM, Cha HS. Behcet's disease associated with malignancy in Korea: a single center experience. Rheumatol Int. 2010; 30:831–835.
33. Cengiz M, Altundag MK, Zorlu AF, Güllü IH, Ozyar E, Atahan IL. Malignancy in Behçet's disease: a report of 13 cases and a review of the literature. Clin Rheumatol. 2001; 20:239–244.
Article
34. Shahram F, Daneshmandi S, Bayan N, Nadji A, Akbarian M, Davatchi F. Behcet's disease and malignancy, report of 11 cases. In : Bang D, Lee ES, Lee S, editors. Behcet's disease. 2000. Seoul: Design Mecca Publishing Co.;p. 515–519.
35. Kural-Seyahi E, Fresko I, Seyahi N, Ozyazgan Y, Mat C, Hamuryudan V, et al. The long-term mortality and morbidity of Behçet syndrome: a 2-decade outcome survey of 387 patients followed at a dedicated center. Medicine (Baltimore). 2003; 82:60–76.
36. Bang D, Lee JH, Lee ES, Lee S. Ten Cases of Behcet's disease associated with Tumor. In : Bang D, Lee ES, Lee S, editors. Behcet's disease. 2000. Seoul: Design Mecca Publishing Co.;p. 526–528.
37. Rubin DC, Shaker A, Levin MS. Chronic intestinal inflammation: inflammatory bowel disease and colitis-associated colon cancer. Front Immunol. 2012; 3:107.
Article
38. Lioté F, Pertuiset E, Cochand-Priollet B, D'Agay MF, Dombret H, Numéric P, et al. Methotrexate related B lymphoproliferative disease in a patient with rheumatoid arthritis. Role of Epstein-Barr virus infection. J Rheumatol. 1995; 22:1174–1178.
39. Harada M, Kumemura H, Fujita A, Yanagimoto C, Harada R, Hashimoto O, et al. A human T-cell lymphotropic virus type-1 (HTLV-1) carrier complicated with various autoimmune diseases including primary biliary cirrhosis. Hepatol Res. 2005; 31:116–119.
Article
40. Chen MR. Epstein-barr virus, the immune system, and associated diseases. Front Microbiol. 2011; 2:5.
Article
41. Zandman-Goddard G, Shoenfeld Y. Infections and SLE. Autoimmunity. 2005; 38:473–485.
Article
42. Kaklamani VG, Sadim M, Koumantaki Y, Kaklamanis P, Pasche B. Role of polymorphisms in Adamantiades-Behçet's disease. J Rheumatol. 2008; 35:2376–2378.
Article
Full Text Links
  • YMJ
Actions
Cited
CITED
export Copy
Close
Share
  • Twitter
  • Facebook
Similar articles
Copyright © 2024 by Korean Association of Medical Journal Editors. All rights reserved.     E-mail: koreamed@kamje.or.kr