• Please log in or register to access this feature.

Case Report

You have full text access to this OnlineOpen article

Role of interleukin-6 in a patient with tumor necrosis factor receptor–associated periodic syndrome: Assessment of outcomes following treatment with the anti–interleukin-6 receptor monoclonal antibody tocilizumab

  1. Prashantha M. Vaitla1,*,
  2. Paul M. Radford2,
  3. Patrick J. Tighe2,
  4. Richard J. Powell2,
  5. Elizabeth M. McDermott1,
  6. Ian Todd2,†,
  7. Elizabeth Drewe1,†

Article first published online: 30 MAR 2011

DOI: 10.1002/art.30215

Issue

Arthritis & Rheumatism

Arthritis & Rheumatism

Volume 63, Issue 4, pages 1151–1155, April 2011

Additional Information

How to Cite

Vaitla, P. M., Radford, P. M., Tighe, P. J., Powell, R. J., McDermott, E. M., Todd, I. and Drewe, E. (2011), Role of interleukin-6 in a patient with tumor necrosis factor receptor–associated periodic syndrome: Assessment of outcomes following treatment with the anti–interleukin-6 receptor monoclonal antibody tocilizumab. Arthritis & Rheumatism, 63: 1151–1155. doi: 10.1002/art.30215

Author Information

  1. 1

    Nottingham University Hospitals National Health Service Trust, Queen's Medical Centre Campus, Nottingham, UK

  2. 2

    University of Nottingham and Queen's Medical Centre, Nottingham, UK

  1. †

    Drs. Todd and Drewe contributed equally to this work.

*Speciality Registrar in Clinical Immunology, Queens Medical Centre Campus, Nottingham University Hospitals NHS Trust, Derby Road, Nottingham NG7 2UH, UK

  1. †

    Drs. Todd and Drewe contributed equally to this work.

Publication History

  1. Issue published online: 3 MAR 2011
  2. Article first published online: 30 MAR 2011
  3. Accepted manuscript online: 10 JAN 2011 11:32AM EST
  4. Manuscript Accepted: 16 DEC 2010
  5. Manuscript Received: 14 MAY 2010

Funded by

  • Jones 1986 Charitable Trust

SEARCH

SEARCH BY CITATION

ARTICLE TOOLS

View Full Article (HTML) Get PDF (112K)

REFERENCES

  • 1
    McDermott MF, Aksentijevich I, Galon J, McDermott EM, Ogunkolande BW, Centola M, et al. Germline mutations in the extracellular domains of the 55kDa TNF receptor, TNFR1, define a family of dominantly inherited autoinflammatory syndromes. Cell 1999; 97: 133–44.
  • 2
    Drewe E, McDermott EM, Powell PT, Isaacs JD, Powell RJ. Prospective study of anti-tumour necrosis factor receptor superfamily 1B fusion protein, and case study of anti-tumour necrosis factor receptor superfamily 1A fusion protein, in tumour necrosis factor receptor associated periodic syndrome (TRAPS): clinical and laboratory findings in a series of seven patients [published erratum appears in Rheumatology (Oxford) 2003;42:711]. Rheumatology (Oxford) 2003; 42: 235–9.
  • 3
    Drewe E, Powell RJ, McDermott EM. Comment on: failure of anti-TNF therapy in TNF receptor 1-associated periodic syndrome (TRAPS). Rheumatology (Oxford) 2007; 46: 1865–6.
  • 4
    Jacobelli S, Andre M, Alexandra JF, Dode C, Papo T. Failure of anti-TNF therapy in TNF receptor 1-associated periodic syndrome (TRAPS). Rheumatology (Oxford) 2007; 46: 1211–2.
  • 5
    Simon A, Bodar EJ, van der Hilst JC, van der Meer JW, Fiselier TJ, Cuppen MP, et al. Beneficial response to interleukin 1 receptor antagonist in TRAPS. Am J Med 2004; 117: 208–10.
  • 6
    Gattorno M, Pelagatti MA, Meini A, Obici L, Barcellona R, Federici S, et al. Persistent efficacy of anakinra in patients with tumor necrosis factor receptor–associated periodic syndrome. Arthritis Rheum 2008; 58: 1516–20.
    Direct Link:
  • 7
    Yokota S, Imagawa T, Mori M, Miyamae T, Aihara Y, Takei S, et al. Efficacy and safety of tocilizumab in patients with systemic-onset juvenile idiopathic arthritis: a randomised, double-blind, placebo-controlled, withdrawal phase III trial. Lancet 2008; 371: 998–1006.
  • 8
    Matsumoto K, Nagashima T, Takatori S, Kawahara Y, Yagi M, Iwamoto M, et al. Glucocorticoid and cyclosporine refractory adult onset Still's disease successfully treated with tocilizumab. Clin Rheumatol 2009; 28: 485–7.
  • 9
    Nowlan ML, Drewe E, Bulsara H, Esposito N, Robins RA, Tighe PJ, et al. Systemic cytokine levels and the effects of etanercept in TNF receptor-associated periodic syndrome (TRAPS) involving a C33Y mutation in TNFRSF1A. Rheumatology (Oxford) 2006; 45: 31–7.
  • 10
    Harambat J, Desjonqueres M, Garaix F, Cochat P, Ranchin B, Cimaz R, et al. First report of rapidly progressive glomerulonephritis in tumor necrosis factor receptor–associated periodic syndrome. Arthritis Rheum 2008; 58: 3275–6.
    Direct Link:
  • 11
    Todd I, Radford PM, Ziegler-Heitbrock L, Ghaemmaghami AM, Powell RJ, Tighe PJ. Elevated CD16 expression by monocytes from patients with tumor necrosis factor receptor–associated periodic syndrome. Arthritis Rheum 2007; 56: 4182–8.
    Direct Link:
  • 12
    Todd I, Radford PM, Draper-Morgan KA, McIntosh R, Bainbridge S, Dickinson P, et al. Mutant forms of tumour necrosis factor receptor I that occur in TNF-receptor-associated periodic syndrome retain signalling functions but show abnormal behaviour. Immunology 2004; 113: 65–79.
    Direct Link:
  • 13
    Rebelo SL, Bainbridge SE, Amel-Kashipaz MR, Radford PM, Powell RJ, Todd I, et al. Modeling of tumor necrosis factor receptor superfamily 1A mutants associated with tumor necrosis factor receptor–associated periodic syndrome indicates misfolding consistent with abnormal function. Arthritis Rheum 2006; 54: 2674–87.
    Direct Link:
  • 14
    Lobito AA, Kimberley FC, Muppidi JR, Komarow H, Jackson AJ, Hull KM, et al. Abnormal disulfide-linked oligomerization results in ER retention and altered signaling by TNFR1 mutants in TNFR1-associated periodic fever syndrome (TRAPS). Blood 2006; 108: 1320–7.
  • 15
    Simon A, Park H, Maddipati R, Lobito AA, Bulua AC, Jackson AJ, et al. Concerted action of wild-type and mutant TNF receptors enhances inflammation in TNF receptor 1-associated periodic fever syndrome. Proc Natl Acad Sci U S A 2010; 107: 9801–6.
View Full Article (HTML) Get PDF (112K)