Skip to main content
SpringerLink
Log in

Rituximab treatment for Sjogren syndrome-associated non-Hodgkin’s lymphoma: case series

  • Short Communication
  • Published:
Rheumatology International Aims and scope Submit manuscript

Abstract

Five per cent of patients with primary Sjogren’s syndrome (pSS) develop malignant non-Hodgkin’s lymphoma (NHL), usually of the mucosa-associated lymphoid tissue (MALT) and most frequently located in the major salivary glands. Rituximab (RTX), a chimeric monoclonal antibody against the CD20 molecule expressed on the surface of mature B cells that has been approved for the treatment of NHL, has been used to treat pSS-associated lymphoma. We have described two cases: one with MALT lymphoma in the parotid glands and the other with a rare thymus lymphoma accompanied by the rare complication of a bullous pneumopathy. Both were treated with RTX at haematological doses, which was unsuccessful in the patient with a salivary lymphoma; in the case of the patient with a thymus lymphoma, the mediastinum mass disappeared and did not relapse. Both patients experienced an improvement in the subjective symptoms of dryness, and their Schirmer’s test and scialoscintigraphy results stabilised. The pulmonary bullae remained unchanged.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Log in via an institution

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Fig. 1
Fig. 2

References

  1. Vitali C, Bombardieri S, Jonsson R, Moutsopoulos HM, Alexander EL, Carsons SE et al (2006) Classification criteria for Sjogren’s syndrome: a revised version of the European criteria proposed by the American-European Consensus Group. Ann Rheum Dis 61:554–558

    Article  Google Scholar 

  2. Masaki Y, Sugai S (2004) Lymphoproliferative disorders in Sjogren’s syndrome. Autoimmun Rev 3:175–182

    Article  PubMed  Google Scholar 

  3. Talal N (1992) Sjogren’s syndrome: historical overview and clinical spectrum of disease. Rheum Dis Clin North Am 18:507–515

    PubMed  CAS  Google Scholar 

  4. Voulgarelis M, Dafni UG, Isenberg DA, Moutsopoulos HM (1999) Malignant lymphoma in primary Sjogren’s syndrome: a multicenter, retrospective, clinical study by the European Concerted Action on Sjogren’s Syndrome. Arthritis Rheum 42:1765–1772

    Article  PubMed  CAS  Google Scholar 

  5. McLaughlin P, Grillo-Lopez AJ, Link BK, Levy R, Czuczman MS (1998) Rituximab chimeric anti-CD20 monoclonal antibody therapy for relapsed indolent lymphoma: half of patients respond to a four-dose treatment program. J Clin Oncol 16:2825–2833

    PubMed  CAS  Google Scholar 

  6. Sakamoto O, Saita N, Ando M, Kohrogi H, Suga M, Ando M (2002) Two cases of Sjogren’s syndrome with multiple bullae. Intern Med 41:124–128

    Article  PubMed  Google Scholar 

  7. Tokuyasu H, Watanabe E, Okazaki R, Kawasaki Y, Kikuchi R, Isowa N (2007) Sjogren’s syndrome with multiple bullae caused by lymphocytic interstitial pneumonia. Lung 185:187–188

    Article  PubMed  Google Scholar 

  8. Theander E, Henriksson G, Ljungberg O, Mandl T, Manthorpe R, Jacobsson LT (2006) Lymphoma and other malignancies in primary Sjogren’s syndrome: a cohort study on cancer incidence and lymphoma predictors. Ann Rheum Dis 65:796–803

    Article  PubMed  CAS  Google Scholar 

  9. Di LC, Mariuzzi L, De GA, Beltrami CA (1996) B cell lymphoma of the thymus and salivary gland. J Clin Pathol 49:595–597

    Article  Google Scholar 

  10. Isaacson PG, Chan JK, Tang C, Addis BJ (1990) Low-grade B-cell lymphoma of mucosa-associated lymphoid tissue arising in the thymus. A thymic lymphoma mimicking myoepithelial sialadenitis. Am J Surg Pathol 14:342–351

    Article  PubMed  CAS  Google Scholar 

  11. Yamasaki S, Matsushita H, Tanimura S, Nakatani T, Hara S, Endo Y, Hara M (1998) B-cell lymphoma of mucosa-associated lymphoid tissue of the thymus: a report of two cases with a background of Sjogren’s syndrome and monoclonal gammopathy. Hum Pathol 29:1021–1024

    Article  PubMed  CAS  Google Scholar 

  12. De Vita S, Boiocchi M, Sorrentino D, Carbone A, Avellini C, Dolceti R et al (1997) Characterization of prelymphomatous stages of B cell lymphoproliferation in Sjogren’s syndrome. Arthritis Rheum 40:318–331

    Article  PubMed  Google Scholar 

  13. Ramos-Casals M, Brito-Zeron P, Yague J, Akasbi M, Bautista R, Ruano M, Claver G, Gil V, Font J (2005) Hypocomplementaemia as an immunological marker of morbidity and mortality in patients with primary Sjogren’s syndrome. Rheumatology (Oxford) 44:89–94

    Article  CAS  Google Scholar 

  14. Quartuccio L, Fabris M, Salvin S, Maset M, De Marchi G, De Vita S (2009) Controversies on rituximab therapy in sjogren syndrome-associated lymphoproliferation. Int J Rheumatol 2009:424935

  15. Pijpe J, van Imhoff GW, Spijkervet FK, Roodenburg JL, Wolbink GJ et al (2005) Rituximab treatment in patients with primary Sjogren’s syndrome: an open-label phase II study. Arthritis Rheum 52:2740–2750

    Article  PubMed  CAS  Google Scholar 

Download references

Author information

Authors and Affiliations

  1. Rheumatology Unit of DIMIMP, University of Bari, Bari, Italy

    M. Covelli, E. Lanciano, P. Tartaglia, V. Grattagliano & G. Lapadula

  2. Radiology Unit of DIMIMP, University of Bari, Bari, Italy

    G. Angelelli

  3. Rheumatology Unit, L. Sacco University Hospital, Milan, Italy

    F. Atzeni & P. Sarzi-Puttini

  4. Experimental Medicine and Rheumatology, William Harvey Research Institute, Queen Mary, University of London, London, UK

    F. Atzeni

Authors
  1. M. Covelli
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. E. Lanciano
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. P. Tartaglia
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. V. Grattagliano
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. G. Angelelli
    View author publications

    You can also search for this author in PubMed Google Scholar

  6. F. Atzeni
    View author publications

    You can also search for this author in PubMed Google Scholar

  7. P. Sarzi-Puttini
    View author publications

    You can also search for this author in PubMed Google Scholar

  8. G. Lapadula
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to M. Covelli.

Rights and permissions

Reprints and permissions

About this article

Cite this article

Covelli, M., Lanciano, E., Tartaglia, P. et al. Rituximab treatment for Sjogren syndrome-associated non-Hodgkin’s lymphoma: case series. Rheumatol Int 32, 3281–3284 (2012). https://doi.org/10.1007/s00296-011-2036-2

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00296-011-2036-2

Keywords

Search

Navigation