Abstract
Infliximab, a monoclonal chimeric anti-tumor necrosis factor alpha (anti-TNF-α) antibody, was tried in two patients suffering from severe refractory juvenile ankylosing spondylarthritis with disease duration of more than 10 years. To assess the response, validated clinical activity parameters were monitored prospectively. In both patients, treatment with infliximab at a dosage of 5 mg/kg body weight already led to considerable improvement with loss of joint pain the day after it was given. Bath Ankylosing Spondylitis Functional Index scores decreased from 5.8 to 0 and 7.2 to 1.0 and the Bath Ankylosing Spondylitis Disease Activity Index from 2.6 to 1.4 and 9.0 to 1.0. In one patient, the response to a single infusion continued for more than 8 months. Because of a recurrence of symptoms in intervals of 2 months, the fourth infusion has now been given to the second patient, resulting in immediate clinical response. No side effects have been noted. Infliximab seems to be a promising agent for treatment of active and refractory juvenile ankylosing spondylitis. Controlled studies and long-term observations are warranted.
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Burgos-Vargas R, Pacheco-Tena C, Vázquez-Mellado J (1997) Juvenile-onset spondyloarthropathies. Rheum Dis Clin North Am 23:569–598
Brewerton DA, James DCO (1975) The histocompatibility antigen HLA-B27 and disease. Semin Arthritis Rheum 4:191
Burgos-Vargas R, Vázquez-Mellado J, Pacheco-Tena C, Hernández-Garduño A, Goycochea-Robles MV (2002) A 26 week randomised, double blind, placebo controlled exploratory study of sulfasalazine in juvenile onset spondyloarthropathies. Ann Rheum Dis 61:941–942
Flato B, Aasland A, Vinje O, Forre O (1998) Outcome and predictive factors in juvenile rheumatoid arthritis and juvenile spondyloarthropathy. J Rheumatol 25:366–375
Minden K, Kiessling U, Listing J et al (2000) Prognosis of patients with juvenile chronic arthritis and juvenile spondyloarthropathy. J Rheumatol 27:2256–2263
Grom AA, Murray KJ, Luyrink L, Emery H, Passo MH, Glass DN et al (1996) Patterns of expression of tumor necrosis factor α, tumor necrosis factor β, and their receptors in synovia of patients with juvenile rheumatoid arthritis and juvenile spondyloarthropathy. Arthritis Rheum 39:1703–1710
Brandt J, Haibel H, Cornely D, Golder W, Gonzalez J, Reddig J, Thriene W, Sieper J, Braun J (2000) Successful treatment of active ankylosing spondylitis with the anti-tumor necrosis factor α monoclonal antibody infliximab. Arthritis Rheum 43:1346–1352
Fries W, Giofre MR, Catanoso M, Lo GR (2002) Treatment of acute uveitis associated with Crohn’s disease and sacroiliitis with infliximab. J Rheumatol 29:118–122
Calin A, Garrett S, Whitelock H, Kennedy LG, O’Hea J, Mallorie P, Jenkinson T (1994) A new approach to defining functional ability in ankylosing spondylitis: the development of the Bath Ankylosing Spondylitis Functional Index. J Rheumatol 21:2281–2285
Garrett S, Jenkinson T, Kennedy LG, Whitelock H, Gaisford P, Calin A (1994) A new approach to defining disease status in ankylosing spondylitis: the Bath Ankylosing Spondylitis Disease Activity Index. J Rheumatol 21:2286–2291
Burgos-Vargas R, Vázquez-Mellado J (1995) The early clinical recognition of juvenile-onset ankylosing spondylitis and its differentiation from juvenile rheumatoid arthritis. Arthritis Rheum 38:835–844
Murray KJ, Grom AA, Thompson SD, Lieuwen D, Passo MH, Glass DN (1998) Contrasting cytokine profiles in the synovium of different forms of juvenile rheumatoid arthritis and juvenile spondyloarthropathy: prominence of interleukin 4 in restricted disease. J Rheumatol 25:1388–1398
Brandt J, Haibel H, Reddig J, Sieper J, Braun J (2002) Successful short term treatment of severe undifferentiated spondyloarthropathy with the anti-tumor necrosis factor-alpha monoclonal antibody infliximab. J Rheumatol 29:118–122
Braun J, Brandt J, Listing J, Zink A, Alten R, Golder W, Gromnica-Ihle E, Kellner H, Krause A, Schneider M, Sörensen H, Zeidler H, Thriene W, Sieper J (2002) Treatment of active ankylosing spondylitis with infliximab: a randomised controlled multicentre trial. Lancet 359:1187–1193
Gorman JD, Sack KE, Davis JC (2002) Treatment of ankylosing spondylitis by inhibition of tumor necrosis factor α. New Engl J Med 346:1349–1356
Kruithof E, van den Bosch F, Baeten D, Herssens A, De Keyser F, Mielants H, Veys EM (2002) Repeated infusions of infliximab, a chimeric anti-TNF-α monoclonal antibody, in patients with active spondyloarthropathy: one year follow up. Ann Rheum Dis 61:207–212
Van den Bosch F, Kruithof E, Baeten D, De Keyser F, Mielants H, Veys EM (2000) Effects of a loading dose regimen of three infusions of chimeric monoclonal antibody to tumor necrosis factor α (infliximab) in spondyloarthropathy: an open pilot study. Ann Rheum Dis 59:428–433
Reiff A, Henrickson M (2001) Prolonged efficacy of etanercept in refractory juvenile ankylosing spondylitis. Arthritis Rheum 44 [Suppl 1]:S292
Horneff G, Burgos-Vargas R (2002) TNF-alpha antagonists for the treatment of juvenile-onset spondylarthritides. Clin Exp Rheumatol 20 [Suppl 28]:137–142
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Schmeling, H., Horneff, G. Infliximab in two patients with juvenile ankylosing spondylitis. Rheumatol Int 24, 173–176 (2004). https://doi.org/10.1007/s00296-003-0378-0
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DOI: https://doi.org/10.1007/s00296-003-0378-0