Case Reports Open access

Subclinical TRAPS treated with canakinumab

Authors

Published
3 August 2021
Section
Case Reports
DOI
10.4081/reumatismo.2021.1406
Pages
131-134

Abstract

Tumor necrosis factor receptor–associated periodic syndrome (TRAPS) is a rare autoinflammatory disease characterized by recurrent episodes of fever and systemic inflammation. We describe the case of a 19-year-old patient who was referred to our attention with frequent subclinical TRAPS episodes characterized by mild arthralgias and crampy abdominal pain, without fever. Inflammatory markers, including serum amyloid A which increases the risk of long-term amyloidosis, were persistently high also when the patient was in good general conditions. Therapy with human anti-interleukin 1β monoclonal antibody, canakinumab, led to disease control and normalization of the inflammatory markers, which are currently still normal. This clinical case supports the need to treat also subclinical TRAPS. In this respect, canakinumab is effective and reduces the risk of developing amyloidosis.

Keywords

References

  1. Lachmann HJ. Periodic fever syndromes. Best Pract Res Clin Rheumatol. 2017; 31: 596-609. DOI: https://doi.org/10.1016/j.berh.2017.12.001
  2. Lachmann HJ, Papa R, Gerhold K, et al. The phenotype of TNF receptor-associated autoinflammatory syndrome (TRAPS) at presentation: a series of 158 cases from the Eurofever/EUROTRAPS international registry. Ann Rheum Dis. 2014; 73: 2160-7. DOI: https://doi.org/10.1136/annrheumdis-2013-204184
  3. Gattorno M, Hofer M, Federici S, et al. Eurofever Registry and the Paediatric Rheumatology International Trials Organisation (PRINTO). Classification criteria for autoinflammatory recurrent fevers. Ann Rheum Dis. 2019; 78: 1025-1032. DOI: https://doi.org/10.1136/annrheumdis-2019-215048
  4. Gattorno M, Obici L, Cattalini M, et al. Canakinumab treatment for patients with active recurrent or chronic TNF receptor-associated periodic syndrome (TRAPS): an open-label, phase II study. Ann Rheum Dis. 2017; 76: 173-178. DOI: https://doi.org/10.1136/annrheumdis-2015-209031
  5. De Benedetti F, Gattorno M, Anton J, et al. Canakinumab for the Treatment of Autoinflammatory Recurrent Fever Syndromes. N Engl J Med. 2018; 378: 1908-1919. DOI: https://doi.org/10.1056/NEJMoa1706314
  6. Lachmann HJ, Quartier P, So A, Hawkins PN. The emerging role of interleukin-1β in autoinflammatory diseases. Arthritis Rheum. 2011; 63: 314-24. DOI: https://doi.org/10.1002/art.30105
  7. Miyamae T, Hanaya A, Kawamoto M, et al. Diagnostic rate of autoinflammatory diseases evaluated by fever patterns in pediatric- and adult-onset patients. J Clin Rheumatol. 2020; 26: 60-62. DOI: https://doi.org/10.1097/RHU.0000000000000929
  8. Procopio V, Manti S, Bianco G, et al. Genotype-phenotype correlation in FMF patients: A "non classic" recessive autosomal or "atypical" dominant autosomal inheritance?. Gene. 2018; 641: 279-286. DOI: https://doi.org/10.1016/j.gene.2017.10.068
  9. Quillinan N, Mohammad A, Mannion G, et al. Imaging evidence for persistent subclinical fasciitis and arthritis in tumor necrosis factor receptor-associated periodic syndrome (TRAPS) between febrile attacks. Ann Rheum Dis. 2010; 69: 1408-9. DOI: https://doi.org/10.1136/ard.2009.118661
  10. Kallinich T, Haffner D, Rudolph B, et al. "Periodic fever" without fever: two cases of non-febrile TRAPS with mutations in the TNFRSF1A gene presenting with episodes of inflammation or monosymptomatic amyloidosis. Ann Rheum Dis. 2006; 65: 958-60. DOI: https://doi.org/10.1136/ard.2005.043570
  11. Kallinich T, Briese S, Roesler J, et al. Two familial cases with tumor necrosis factor receptor-associated periodic syndrome caused by a non-cysteine mutation (T50M) in the TNFRSF1A gene associated with severe multiorganic amyloidosis. J Rheumatol. 2004; 31: 2519-22. DOI: https://doi.org/10.1055/s-2003-45080
  12. Scarpioni R, Ricardi M, Albertazzi V. Secondary amyloidosis in autoinflammatory diseases and the role of inflammation in renal damage. World J Nephrol. 2016; 5: 66-75. DOI: https://doi.org/10.5527/wjn.v5.i1.66

Metrics

Citations and online attention from Dimensions, Altmetric and PlumX.

PlumX Metrics

License

PAGEPress has chosen to apply the Creative Commons Attribution NonCommercial 4.0 International License (CC BY-NC 4.0) to all manuscripts to be published.