The risk of cancer in patients with primary Sjögren syndrome: a single-center study from Turkey

The risk of cancer in patients with primary Sjögren syndrome: a single-center study from Turkey

Background: The aim of this study is to determine the risk of cancer in patients with primary Sjögren syndrome (pSS) from a single center in Turkey. Methods: Clinical data of the subjects with pSS were retrospectively analyzed. The incidence of cancer for general population was obtained from GLOBOCAN 2018. Age- and sex-specific standardized incidence ratios (SIR) of solid and hematological cancers were calculated compared with the general population. Results: Four hundred thirty patients with pSS were included in the study. The majority of the patients were female (n = 396, 92.1%), and the mean age was 58.6 ± 12.0 years. Thirty-four patients (7.9 %) were diagnosed with cancer (26 solid and 8 hematological) during follow-up. The SIR for all cancers was 2.45 (95% CI, 1.625–3.275). The SIR was 2.42 (95% CI, 1.542–3.298) for solid cancers and 8.42 (95% CI, 2.394 - 14.446) for hematological cancers. The most diagnosed malignancies were breast cancer (n = 6), ovarian cancer (n = 6), and non-Hodgkin lymphoma (NHL) (n = 4). There was an increased risk for ovarian cancer (SIR 12.76, 95% CI, 2.545–22.975). The SIR values were 2.08 (95% CI, 0.419-3.741) and 10.81 (95% CI, 0.216–21.404) for breast cancer and NHL, respectively. Conclusion: The risk of hematological and solid cancers was higher in the patients with pSS when compared to general population. In our pSS cohort, the risk for ovarian cancer was found to be increased, which has not been previously reported in the literature.

___

  • 1. Fox RI. Sjögren’s syndrome. Lancet 2005; 366:321-331. doi:10.1016/S0140-6736(05)669905
  • 2. Ramos-Casals M, Solans R, Rosas J, Camps MT, Gil A et al. GEMESS Study Group. Primary Sjögren syndrome in Spain = clinical and immunologic expression in 1010 patients. Medicine (Baltimore)2008 Jul; 87(4):210-219. doi: 10.1097/ MD.0b013e318181e6af
  • 3. Yazisiz V, Avci AB, Erbasan F, Kiriş E, Terzioğlu E. Diagnostic performance of minor salivary gland biopsy, serological and clinical data in Sjögren’s syndrome: a retrospective analysis. Rheumatolology International 2009; 29(4):403-409. doi: 10.1007/s00296-008-0698-1
  • 4. Yazisiz V, Arslan G, Ozbudak IH, Turker S, Erbasan F et al. Lung involvement in patients with primary Sjögren’s syndrome: what are the predictors? Rheumatology International 2010;1317-1324. doi: 10.1007/s00296-009-1152-8
  • 5. Bombardieri M, Argyropoulou OD, Ferro F, Coleby R, Pontarini E et al. One year in review 2020: pathogenesis of primary Sjögren’s syndrome. Clinical and Experimental Rheumatology 2020; 38(4): Suppl 126:3-9
  • 6. Sarigul M, Yazisiz V, Bassorgun CI, Ulker M, Avci AB et al. The numbers of Foxp3 + Treg cells are positively correlated with higher grade of infiltration at the salivary glands in primary Sjogren’s syndrome. Lupus 2010; 19(2):138-145. doi: 10.1177/0961203309348234
  • 7. Yazisiz V, Göçer M, Erbasan F, Uçar İ, Aslan B et al. Survival analysis of patients with Sjögren’s syndrome in Turkey: a tertiary hospital-based study. Clinical Rheumatology 2020; 39(1):233-241. doi: 10.1007/s10067-019-04744-6
  • 8. Zhang W, Feng S, Yan S, Zhao Y, Li M et al. Incidence of malignancy in primary Sjogren’s syndrome in a Chinese cohort. Rheumatology (Oxford)2010Mar;49(3):571-577. doi: 10.1093/rheumatology/kep404. Epub 2009 Dec 29
  • 9. Liang Y, Yang Z, Qin B, Zhong R. Primary Sjogren’s syndrome and malignancy risk: a systematic review and meta-analysis. Annals of the Rheumatic Diseases 2014 Jun; 73(6): 1151-1156. doi: 10.1136/annrheumdis-2013-203305. Epub 2013 May 17
  • 10. Brom M, Moyano S, Gandino IJ, Scolnik M, Soriano ER. Incidence of cancer in a cohort of patients with primary Sjögren syndrome in Argentina. Rheumatolology International 2019 Oct; 39(10): 1697-1702. doi: 10.1007/s00296-019-04433-2. Epub 2019 Aug 30
  • 11. Kassan SS, Thomas TL, Moutsopoulos HM, Hoover R, Kimberly RP et al. Increased risk of lymphoma in sicca syndrome. Annals of Internal Medicine 1978; 89: 888–892
  • 12. Ekström Smedby K, Vajdic CM, Falster M, Engels EA, Martínez-Maza O et al. Autoimmune disorders and risk of non-Hodgkin lymphoma subtypes: a pooled analysis within the InterLymph Consortium. Blood 2008 Apr 15; 111(8): 4029-4038. doi: 10.1182/blood-2007-10-119974. Epub 2008 Feb 8
  • 13. Zintzaras E, Voulgarelis M, Moutsopoulos HM. The risk of lymphoma development in autoimmune diseases: a metaanalysis. Archives of Internal Medicine 2005 Nov 14; 165(20): 2337-2344. doi: 10.1001/archinte.165.20.2337
  • 14. Nishishinya MB, Pereda CA, Muñoz-Fernández S, PegoReigosa JM, Rúa-Figueroa I et al. Identification of lymphoma predictors in patients with primary Sjögren’s syndrome: a systematic literature review and meta-analysis. Rheumatology International 2015 Jan; 35(1): 17-26. doi: 10.1007/s00296-014- 3051-x. Epub 2014 Jun 5
  • 15. Theander E, Henriksson G, Ljungberg O, Mandl T, Manthorpe R et al. Lymphoma and other malignancies in primary Sjögren’s syndrome: a cohort study on cancer incidence and lymphoma predictors. Annals of the Rheumatic Diseases 2006 Jun; 65(6): 796-803. doi: 10.1136/ard.2005.041186. Epub 2005 Nov 10
  • 16. Brito-Zerón P, Kostov B, Fraile G, Caravia-Durán D, Maure B et al; SS Study Group GEAS-SEMI (2017). Characterization and risk estimate of cancer in patients with primary Sjögren syndrome. Journal of Hematology and Oncology 2017; 10(1): 90. doi: 10.1186/s13045-017-0464-5
  • 17. Weng M-Y, Huang Y-T, Liu M-F, Lu TH. Incidence of cancer in a nationwide population cohort of 7852 patients with primary Sjogren’s syndrome in Taiwan. Annals of the Rheumatic Diseases 2012; 71: 524–527. doi: 10.1136/ annrheumdis-2011-200402
  • 18. Hemminki K, Liu X, Ji J, Försti A, Sundquist J et al. Effect of autoimmune diseases on risk and survival in female cancers. Gynecologic Oncology 2012; 127: 180–185. doi: 10.1016/j.ygyno.2012.07.100
  • 19. Lewandowska AM, Rudzki M, Rudzki S, Lewandowski T, Laskowska B. Environmental risk factors for cancer-review paper. Annals of Agricultural and Environmental Medicine 2019; 26(1): 1-7. doi: 10.26444/aaem/94299
  • 20. Voulgarelis M, Tzioufas AG, Moutsopoulos HM. Mortality in Sjogren’s syndrome. Clinical and Experimental Rheumatology 2008; 26(5 Suppl 51): 66–71
  • 21. Risselada AP, Kruize AA, Goldschmeding R, Lafeber FPJG, Bijlsma JWJ et al. The prognostic value of routinely performed minor salivary gland assessments in primary Sjögren’s syndrome. Annals of the Rheumatic Diseases 2014; 73(8): 1537-1540. doi: 10.1136/annrheumdis-2013-204634
  • 22. Lazarus MN, Robinson D, Mak V, Møller H, Isenberg DA. Incidence of cancer in a cohort of patients with primary Sjogren’s syndrome. Rheumatology 2006; 45(8): 1012-1005. doi: 10.1093/rheumatology/kei281
  • 23. Shiboski SC, Shiboski CH, Criswell L, Baer A, Challacombe S et al. Sjögren’s International Collaborative Clinical Alliance (SICCA) Research Groups. American College of Rheumatology classification criteria for Sjögren’s syndrome: a data-driven, expert consensus approach in the Sjögren’s International Collaborative Clinical Alliance cohort. Arthritis Care and Research (Hoboken) 2012 Apr; 64(4): 475-487. doi: 10.1002/ acr.21591
  • 24. Global Cancer Observatory, International Agency for Research on Cancer, World Health Organization Cancer Today. http:// gco. iarc.fr/today /home. Accessed 02 July 2020
  • 25. Igoe A, Merjanah S, Scofield RH. Sjögren Syndrome and Cancer. Rheumatic Disease Clinics of North America 2020 Aug; 46(3): 513-532. doi: 10.1016/j.rdc.2020.05.004
  • 26. Pego-Reigosa JM, Restrepo Vélez J, Baldini C, Rúa-Figueroa Fernández de Larrinoa Í. Comorbidities (excluding lymphoma) in Sjögren’s syndrome. Rheumatology 2019; (Oxford)15: key329. doi: 10.1093/rheumatology/key329
  • 27. Kang J, Kim H, Kim J, Choi S, Jung SY et al. Risk of malignancy in Korean patients with primary Sjögren’s syndrome. International Journal of Rheumatic Diseases 2020; 23(9): 1240- 1247. doi: 10.1111/1756-185X.13927
  • 28. Nocturne G, Pontarini E, Bombardieri M, Mariette X. Lymphomas complicating primary Sjögren’s syndrome: from autoimmunity to lymphoma. Rheumatology 2019; (Oxford). kez052. doi: 10.1093/rheumatology/kez052
  • 29. Kauppi M, Pukkala E, Isoma ki H. Elevated incidence of hematologic malignancies in patients with Sjögren’s syndrome compared with patients with rheumatoid arthritis (Finland). Cancer Causes Control 1997; 8:201–4
  • 30. Fragkioudaki S, Mavragani CP, Moutsopoulos HM. Predicting the risk for lymphoma development in Sjogren syndrome: An easy tool for clinical use. Medicine 2016; 95(25): e3766. doi: 10.1097/MD.0000000000003766
  • 31. Brito-Zerón P, Acar-Denizli N, Ng WF, Horváth IF, Rasmussen A et al; Sjögren Big Data Consortium. Epidemiological profile and north-south gradient driving baseline systemic involvement of primary Sjögren’s syndrome. Rheumatology 2020; 59(9): 2350-2359. doi: 10.1093/rheumatology/kez578
  • 32. Noureldine HA, Nour-Eldine W, Hodroj MH, Noureldine MHA, Taher A et al. Hematological malignancies in connective tissue diseases. Lupus 2020; 29(3): 225-235. doi: 10.1177/0961203319899986
  • 33. Aggarwal R, Anaya JM, Koelsch KA, Kurien BT, Scofield RH. Association between Secondary and Primary Sjögren’s Syndrome in a Large Collection of Lupus Families. Autoimmune Disease 2015; 2015:298506. doi: 10.1155/2015/298506
  • 34. Ladouceur A, Tessier-Cloutier B, Clarke AE, Ramsey-Goldman R, Gordon C et al. Cancer and Systemic Lupus Erythematosus. Rheumatic Disease Clinics of North America 2020; 46(3):533- 550. doi: 10.1016/j.rdc.2020.05.005
  • 35. Bernatsky S, Ramsey-Goldman R, Foulkes WD, Gordon C, Clarke AE. Breast, ovarian, and endometrial malignancies in systemic lupus erythematosus: a meta-analysis. British Journal of Cancer 2011; 104(9): 1478-1481. doi: 10.1038/bjc.2011.115
  • 36. Moghadam-Kia S, Oddis CV, Ascherman DP, Aggarwal R. Risk Factors and Cancer Screening in Myositis. Rheumatic Disease Clinics of North America 2020; 46(3): 565-576. doi: 10.1016/j. rdc.2020.05.006
  • 37. Hashimoto A, Arinuma Y, Nagai T, Tanaka S, Matsui T et al. Incidence and the risk factor of malignancy in Japanese patients with systemic sclerosis. Internal Medicine 2012; 51(13): 1683- 1688. doi: 10.2169/internalmedicine.51.7686
  • 38. De Cock D, Hyrich K. Malignancy and rheumatoid arthritis: Epidemiology, risk factors and management. Best Practice and Research Clinical Rheumatology 2018; 32(6): 869-886. doi: 10.1016/j.berh.2019.03.01
  • 39. Aus AT. Can we prevent ovarian cancer? Ceska Gynekologie 2020; 85(1): 49-58
  • 40. Baandrup L, Kjaer SK, Olsen JH, Dehlendorff C, Friis S. Lowdose aspirin use and the risk of ovarian cancer in Denmark. Annals of Oncology 2015; 26(4): 787-792. doi: 10.1093/ annonc/mdu578
  • 41. Pan SY, Ugnat AM, Mao Y. Physical activity and the risk of ovarian cancer: a case-control study in Canada. International Journal of Cancer 2005; 117(2): 300-307. doi: 10.1002/ijc.21157
  • 42. Iversen L, Fielding S, Lidegaard Ø, Mørch LS, Skovlund CW et al. Association between contemporary hormonal contraception and ovarian cancer in women of reproductive age in Denmark: prospective, nationwide cohort study. British Medical Journal 2018; 362: k3609. doi: 10.1136/bmj.k3609
Turkish Journal of Medical Sciences-Cover
  • ISSN: 1300-0144
  • Yayın Aralığı: Yılda 6 Sayı
  • Yayıncı: TÜBİTAK
Sayıdaki Diğer Makaleler

Medically important Candida spp. identification: an era beyond traditional methods

Jülide Sedef GÖÇMEN, Elvan HORTAÇ İŞTAR, Şükran YAVUZDEMİR, Zeynep Ceren KARAHAN, Alper TEKELİ, Ebru EVREN, Yasemin YAVUZ

Assessment of subclinical cardiovascular alterations in nonfunctioning adrenal incidentalomas

Büşra CAN, Bülent CAN, Pelin KARACA ÖZER, Özge TELCİ ÇAKLILI

Radiomics analysis of uterine tumors in 18F-fluorodeoxyglucose positron emission tomography for prediction of lymph node metastases in endometrial carcinoma

Çiğdem SOYDAL, Mine ARAZ, Bulut VARLI, Salih TAŞKIN, Uğur Fırat ORTAÇ, Batuhan BAKIRARAR

Classical heart rate variability and nonlinear heart rate analysis in mice under Napentobarbital and ketamine/xylazine anesthesia

Hasan KAZDAĞLI, H. Fehmi ÖZEL, Şüheda ALPAY, Mustafa ÖZBEK, Mürüvvet ALENBEY

The risk of cancer in patients with primary Sjögren syndrome: a single-center study from Turkey

Funda ERBASAN, Tahir Saygın ÖĞÜT, Ece ÇELİK, Veli YAZISIZ, Bengisu ASLAN, Mustafa Ender TERZİOĞLU, Melis DİLBİL

Conservative management of patients with endometrial intraepithelial neoplasia (EIN): Factors that could affect response and pregnancy rates.

Christos IAVAZZO, Ioannis GKEGKES

Effectiveness of a supervised group exercise therapy based on the biopsychosocial model introduced simultaneously with anti-TNF therapy in anti-TNF-naive patients with active ankylosing spondylitis

Jale KARAKAYA, Edibe ÜNAL, Nur Banu KARACA, Umut KALYONCU, Sedat KİRAZ

The clinical follow-up and management of COVID-19 in children and adolescents with an immunocompromised state or a malignancy

Özlem TÜFEKÇİ, Dilek İNCE, Şefika AKYOL, Meral TORUN BAYRAM, Refik Emre ÇEÇEN, Ayşe ÇAKIL GÜZİN, Hatice KARAOĞLU ASRAK, İrem Ceren ERBAŞ, Şilem ÖZDEM ALATAŞ, Nurşen BELET

Pertussis prevalence among adult patients with acute cough

Serhat ÜNAL, Pınar ZARAKOL, Hatice BÖLEK, Mine Durusu TANRIÖVER, Ahmet İLBAY, Ezgi Çalışkan GÜZELCE

Test-retest reliability and minimal detectable change for measures of wearable gait analysis system (G-Walk) in children with cerebral palsy

Gamze ÇOBANOĞLU, Gökhan YAZICI, Melek VOLKAN YAZICI