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Tocilizumab treatment in Felty’s syndrome

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Abstract

Felty’s syndrome (FS) is a deforming disease, characterized by the triad of rheumatoid arthritis (RA), neutropenia, and splenomegaly. Currently, FS patients are treated mainly with immunosuppressants, such as methotrexate and glucocorticoids, which however are not suitable to some patients and may cause severe side effects. Here we report a clinical FS case that was treated with Tocilizumab (TCZ) successfully. The patient had symmetrical swelling and pain of multiple joints, deformity of elbow joints with obvious morning stiffness. Joint color Doppler ultrasound showed synovial hyperplasia and bone erosion of wrist and proximal interphalangeal joints and CT scan suggested splenomegaly. Further examination showed neutropenia and anemia, a high titer of anti-cyclic citrullinated peptide antibody, rheumatoid factor and anti-nuclear antibodies, positive p-ANCA, and elevated IgA and IgG. After treating with TCZ, the patient has been relieved of clinical symptoms. His spleen has recovered to normal size. The absolute neutrophil count (ANC) tended to be stable, and joint erosion did not deteriorate. We have reviewed the literatures on FS treatment with biological agents and found only a few reports using TNF-α antagonist and rituximab treating FS, but none with TCZ. So, it is the first time to report a successful FS case treated with TCZ. This case suggests that the TCZ may be a new choice for FS treatment, under the condition of closely monitoring the ANC.

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References

  1. Balint GP, Balint PV (2004) Felty’s syndrome. Best Pract Res Clin Rheumatol 18(5):631–645. https://doi.org/10.1016/j.berh.2004.05.002

    Article  PubMed  Google Scholar 

  2. Ish-Hurwitz S, Dovrish Z, Edelstein E, Bernheim J, Bernheim J, Hadari R, Amital H (2007) Diffuse disseminated candidiasis in a patient with Felty’s syndrome: a case report. Rheumatol Int 28(1):65–68. https://doi.org/10.1007/s00296-007-0366-x

    Article  PubMed  Google Scholar 

  3. Patel R, Akhondi H (2020) Felty syndrome. Stat Pearls [Internet]. Stat Pearls Publishing, Treasure Island

    Google Scholar 

  4. Wassenberg S, Herborn G, Rau R (1998) Methotrexate treatment in Felty’s syndrome. Br J Rheumatol 37(8):908–911. https://doi.org/10.1093/rheumatology/37.8.908

    Article  CAS  PubMed  Google Scholar 

  5. Xiao RZ, Xiong MJ, Long ZJ, Fan RF, Lin DJ (2014) Diagnosis of Felty’s syndrome, distinguished from hematological neoplasm: a case report. Oncol Lett 7(3):713–716. https://doi.org/10.3892/ol.2013.1779

    Article  PubMed  Google Scholar 

  6. Mahévas M, Audia S, De Lastours V, Michel M, Bonotte B, Godeau B (2007) Neutropenia in Felty’s syndrome successfully treated with hydroxychloroquine. Haematologica 92(7):e78–e79. https://doi.org/10.3324/haematol.11819

    Article  PubMed  Google Scholar 

  7. Talip F, Walker N, Khan W, Zimmermann B (2001) Treatment of Felty’s syndrome with leflunomide. J Rheumatol 28(4):868–870

    CAS  PubMed  Google Scholar 

  8. Yazıcı A, Uçar A, Mehtap Ö, Gönüllü EÖ, Tamer A (2014) Presentation of three cases followed up with a diagnosis of Felty syndrome. Eur J Rheumatol 1(3):120–122. https://doi.org/10.5152/eurjrheumatol.2014.026

    Article  PubMed  PubMed Central  Google Scholar 

  9. Sordet C, Gottenberg JE, Hellmich B, Kieffer P, Mariette X, Sibilia J (2005) Lack of efficacy of rituximab in Felty’s syndrome. Ann Rheum Dis 64(2):332–333. https://doi.org/10.1136/ard.2004.025643

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  10. Heylen L, Dierickx D, Vandenberghe P, Westhovens R (2012) Targeted therapy with rituximab in Felty’s syndrome: a case report. Open Rheumatol J 6:312–314. https://doi.org/10.2174/1874312901206010312

    Article  PubMed  PubMed Central  Google Scholar 

  11. Narváez J, Domingo-Domenech E, Gómez-Vaquero C, López-Vives L, Estrada P, Aparicio M, Martín-Esteve I, Nolla JM (2012) Biological agents in the management of Felty’s syndrome: a systematic review. Semin Arthritis Rheum 41(5):658–668. https://doi.org/10.1016/j.semarthrit.2011.08.008

    Article  CAS  PubMed  Google Scholar 

  12. Becker H, Appel H, Fassbinder T (2014) Rituximab for treatment of Felty’s syndrome. Z Rheumatol 73(5):465–468. https://doi.org/10.1007/s00393-014-1380-5

    Article  CAS  PubMed  Google Scholar 

  13. Tomi AL, Lioté F, Ea HK (2012) One case of Felty’s syndrome efficiently treated with rituximab. Jt Bone Spine 79(6):624–625. https://doi.org/10.1016/j.jbspin.2012.01.013

    Article  Google Scholar 

  14. Wang CR, Chiu YC, Chen YC (2018) Successful treatment of refractory neutropenia in Felty’s syndrome with rituximab. Scand J Rheumatol 47(4):340–341. https://doi.org/10.1080/03009742.2017.1334816

    Article  CAS  PubMed  Google Scholar 

  15. Furst DE, Fleischman R, Kalden J, Kavanaugh A, Sieper J, Mease P, Smolen J, Breedveld F (2013) Documentation of off-label use of biologics in rheumatoid arthritis. Ann Rheum Dis 72(Suppl 2):ii35–ii51. https://doi.org/10.1136/annrheumdis-2013-consensusapp

    Article  CAS  PubMed  Google Scholar 

  16. Hazenberg BP, Van Leeuwen MA, Van Rijswijk MH, Stern AC, Vellenga E (1989) Correction of granulocytopenia in Felty’s syndrome by granulocyte-macrophage colony-stimulating factor. Simultaneous induction of interleukin-6 release and flare-up of the arthritis. Blood 74(8):2769–2770

    Article  CAS  Google Scholar 

  17. Rashba EJ, Rowe JM, Packman CH (1996) Treatment of the neutropenia of Felty syndrome. Blood Rev 10(3):177–184. https://doi.org/10.1016/s0268-960x(96)90024-7

    Article  CAS  PubMed  Google Scholar 

  18. Aghdashi MA, Khadir M, Dinparasti-Saleh R (2019) Antinuclear antibodies and lupus-like manifestations in rheumatoid arthritis and ankylosing spondylitis patients at 4 months’ follow-up after treatment with infliximab and etanercept. Curr Rheumatol Rev 16(1):61–66. https://doi.org/10.2174/1573397115666190506152729

    Article  CAS  Google Scholar 

  19. Ghavami A, Genevay S, Fulpius T, Gabay C (2005) Etanercept in treatment of Felty’s syndrome. Ann Rheum Dis 64(7):1090–1091. https://doi.org/10.1136/ard.2004.032110

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  20. Sheppard M, Laskou F, Stapleton PP, Hadavi S, Dasgupta B (2017) Tocilizumab (Altera). Hum Vaccin Immunother 13(9):1972–1988. https://doi.org/10.1080/21645515.2017.131690

    Article  PubMed  PubMed Central  Google Scholar 

  21. Narváez J, Díaz-Torné C, Magallares B, Hernández MV, Reina D, Corominas H, Sanmartí R, de la Serna AR, Llobet JM, Nolla JM (2015) Comparative effectiveness of tocilizumab with either methotrexate or leflunomide in the treatment of rheumatoid arthritis. PLoS ONE 10(4):e0123392. https://doi.org/10.1371/journal.pone.0123392

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  22. Lazaro E, Morel J (2015) Management of neutropenia in patients with rheumatoid arthritis. Jt Bone Spine 82(4):235–239. https://doi.org/10.1016/j.jbspin.2015.01.005

    Article  CAS  Google Scholar 

  23. Verhoeven F, Guillot X, Prati C, Wendling D (2015) Treatment of pseudo Felty’s syndrome: is there a place for rituximab? Jt Bone Spine 82(3):196–199. https://doi.org/10.1016/j.jbspin.2014.12.001

    Article  CAS  Google Scholar 

  24. Shovman O, Shoenfeld Y, Langevitz P (2015) Tocilizumab-induced neutropenia in rheumatoid arthritis patients with previous history of neutropenia: case series and review of literature. Immunol Res 61(1–2):164–168. https://doi.org/10.1007/s12026-014-8590-4

    Article  CAS  PubMed  Google Scholar 

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Acknowledgements

The authors would like to thank the editors and two reviewers for great assistance with their paper revision. A very special thanks to Dr. Jianying Shen for her hardwork in language editing of this manuscript.

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This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

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Correspondence to Li Dong.

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All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Declaration of Helsinki and its later amendments or comparable ethical standards. All authors fulfilled the ICMJE authorship criteria.

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Informed consent was obtained from the individual participant included in the study. Informed consent was obtained from this patient for publication of this case report and any accompanying images. A copy of this consent is available to the editors of the journal.

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Rongda Li and Qi Wan: First authors.

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Li, R., Wan, Q., Chen, P. et al. Tocilizumab treatment in Felty’s syndrome. Rheumatol Int 40, 1143–1149 (2020). https://doi.org/10.1007/s00296-020-04588-3

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