Successful treatment of tumor necrosis factor receptor-associated periodic syndrome (TRAPS) with tocilizumab: a case report

Akasbi, Nessrine; Soyfoo, Muhammad Shahnawaz

doi:10.5152/eurjrheumatol.2014.14053

Cited by 22 publications

(8 citation statements)

References 9 publications

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“…Levels of IL-6 are elevated in patients with TRAPS; thus, suppressing IL-6 levels may help to control the inflammatory attacks. Several case reports support the efficacy of tocilizumab in TRAPS patients who have failed other biologic treatments [55][56][57].…”

Section: Treatmentmentioning

confidence: 97%

Tumor Necrosis Factor Receptor-Associated Periodic Syndrome (TRAPS)

Papadopoulou

Lachmann

2019

Periodic and Non-Periodic Fevers

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Section: Treatmentmentioning

confidence: 97%

Tumor Necrosis Factor Receptor-Associated Periodic Syndrome (TRAPS)

Papadopoulou

Lachmann

2019

Periodic and Non-Periodic Fevers

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“…Inadequate control of inflammation may increase the morbidity of amyloidosis. Both IL-6 and IL-8 have been reported to be elevated in TRAPS (18), and tocilizumab, an IL-6 receptor antibody, has been reported to be useful as a treatment for patients with TRAPS who have been unsuccessfully treated with TNF inhibitors and IL-1 inhibitors (19)(20)(21).…”

Section: The Role Of Il-6 In Tnf Receptorassociated Periodic Syndromementioning

confidence: 99%

“…Research has found that the treatment of TRAPS with TCZ can halt acute attacks and prevent further attacks for around 6 months ( 19 ). TCZ has been found effective in TRAPS patients who had previously failed to respond to etanercept and infliximab (both TNF-α inhibitors) ( 20 ). A case report from Japan found TCZ therapy effective in a 30-year-old female patient with TRAPS who was refractory to etanercept ( 21 ).…”

Section: Potential Therapeutic Targets and Issues With Il-6 Inhibitor...mentioning

confidence: 99%

Interleukin-6 inhibition in the treatment of autoinflammatory diseases

Koga

Kawakami

2022

Front. Immunol.

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Autoinflammatory diseases are characterized by abnormalities that prevent innate immune cells from producing autoantibodies. While interleukin (IL)-6 is not directly associated with inflammasomes, like IL-1β or IL-18, it plays an important role in the pathogenesis of autoinflammatory diseases. Studies of autoinflammatory diseases, such as familial Mediterranean fever, cryopyrin-associated periodic syndrome, and tumor necrosis factor receptor-associated periodic syndrome, have shown IL-6 to be a promising therapeutic target. It has also been suggested that inhibition of IL-6 may have a therapeutic effect on amyloidosis, which is frequently associated with these chronic inflammatory diseases. In this study, we discuss the most recent research on the role of IL-6 in autoinflammatory diseases and its potential as a therapeutic target in their treatment.

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“…Treatment with IL-6 inhibitor tocilizumab (a humanized monoclonal antibody that competitively inhibits the binding of IL-6 to its receptor) was reported in patients who did not respond to TNF or IL-1 inhibitors [83,84]. Several case reports were published in patients with various TNFR1 variants: one patient with R92Q, the second patient with no identified variant, the third patient with C33Y, and the fourth patient with C96R, were all treated with tocilizumab with complete resolution of their symptoms [83][84][85][86].…”

Section: Traps Treatmentmentioning

confidence: 99%

Revisiting TNF Receptor-Associated Periodic Syndrome (TRAPS): Current Perspectives

Cudrici

Deuitch

Aksentijevich

2020

IJMS

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Tumor necrosis factor receptor-associated periodic syndrome (TRAPS) is an autosomal dominant autoinflammatory syndrome characterized by prolonged and recurrent episodes of fever, abdominal and/or chest pain, arthralgia, myalgia, and erythematous rash. TRAPS is associated with heterozygous variants in the TNFRSF1A gene, which encodes the TNFR1 (tumor necrosis factor receptor 1) receptor. Disease-causing variants are found exclusively in the extracellular domain of TNFR1 and affect receptor structure and binding to the TNF ligand. The precise mechanism of the disease is still unclear, but it is thought that intracellular accumulation of misfolded mutant protein leads to endoplasmic reticulum stress and enhanced inflammatory responses through constitutive activation of various immune pathways. Other possible mechanisms contributing to the disease pathogenesis include defective receptor shedding, TNF-induced cell death, production of reactive oxygen species, and autophagy impairment. Patients’ leucocytes are hyperresponsive to stimulation and produce elevated levels of proinflammatory cytokines. Systemic autoimmune (AA) amyloidosis is an important cause of morbidity and mortality in TRAPS. Over the last two decades, new therapies have changed the progression and outcome of the disease. In this review, we summarize clinical data from 209 patients with validated pathogenic variants reported in the literature and discuss TRAPS diagnosis, pathogenesis, and treatment options.

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Successful treatment of tumor necrosis factor receptor-associated periodic syndrome (TRAPS) with tocilizumab: a case report

Cited by 22 publications

References 9 publications

Tumor Necrosis Factor Receptor-Associated Periodic Syndrome (TRAPS)

Tumor Necrosis Factor Receptor-Associated Periodic Syndrome (TRAPS)

Interleukin-6 inhibition in the treatment of autoinflammatory diseases

Revisiting TNF Receptor-Associated Periodic Syndrome (TRAPS): Current Perspectives

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