Successful treatment of refractory anti-signal recognition particle myopathy using abatacept

Maeshima, Keisuke; Kiyonaga, Yasuhiro; Imada, Chiharu; Iwakura, Mikako; Hamasaki, Hajime; Haranaka, Miwa; Ishii, Koji

doi:10.1093/rheumatology/ket251

Cited by 18 publications

(6 citation statements)

References 10 publications

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“…Furthermore, Musuruana and Cavallasca 5 reported a dramatic response from a patient receiving methotrexate and azathioprine combined with abatacept. In a case report, Maeshima et al 6 found abatacept to be effective in a patient that did not respond to any biological agents, including a TNF blocker and tocilizumab. Nevertheless, none of these studies administered abatacept in the form of a monotherapy.…”

Section: Discussionmentioning

confidence: 99%

“…Even though the literature contains numerous studies (case studies and series of a small number of cases) concerning the use of abatacept in the treatment of PM, none of such studies employed this agent in the form of a monotherapy. [3][4][5][6] In this article, we present a case diagnosed with PM that did not sufficiently respond to neither biological agents, namely rituximab, nor pharmaceutical agents that were indicated for the conventional treatment of the disease and developed femoral head avascular necrosis because of the corticosteroid treatment.…”

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confidence: 99%

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A Case of Refractory Polymyositis Successfully Treated With Abatacept Monotherapy

2020

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Polymyositis (PM) is an autoimmune disease progressing in the form of a break down of the muscles that is induced by chronic inflammation in skeletal muscles. Muscle weakness is painless and concentrates on proximal muscles, involving the pectoral and pelvic girdle. If the disease is not treated properly, it may progress and lead to a considerable decrease in the quality of life. Its conventional treatment involves drugs that suppress inflammation such as steroids, methotrexate, azathioprine, and intravenous immunoglobulin. However, conventional treatment may prove insufficient to halt the progression of the disease and offer only a limited improvement because of the adverse effects it causes in some patients. In this article, we present a 48-year-old female patient diagnosed with PM nearly 13 years ago that did not sufficiently respond to the pharmaceutical agents that were indicated for the conventional treatment of the disease and developed femoral head avascular necrosis because of the treatment and was, in the end, successfully treated with abatacept monotherapy at our clinic.

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Section: Discussionmentioning

confidence: 99%

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confidence: 99%

A Case of Refractory Polymyositis Successfully Treated With Abatacept Monotherapy

2020

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“…The costimulatory molecules CD28 and CTLA‐4 are both upregulated in the muscle tissue of PM and DM patients suggesting that abatacept, an anti‐T cell biologic targeting CD80 and CD86 on antigen‐presenting cells, might be effective in myositis . This agent was effective in a refractory PM patient , a JDM patient with severe, ulcerative rash and calcinosis , and a patient with anti‐SRP‐positive severe PM . It was recently reported that a patient with severe myositis, rheumatoid arthritis, vasculitis and ILD refractory to several immunosuppressive agents and biologic therapies responded to abatacept .…”

Section: Biologic Agents In Myositismentioning

confidence: 99%

Update on the pharmacological treatment of adult myositis

Oddis

2016

J Intern Med

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“…Abatacept use was associated with a favorable outcome in a report of refractory PM [52], while a recalcitrant JDM patient with ulcerative skin disease and progressive calcinosis also improved [53]. In another Japanese case report, abatacept was successful in the treatment of refractory anti-signal recognition particle (anti-SRP) myositis [54]. In a more recent report, a patient with severe myositis in overlap with rheumatoid arthritis, peripheral vasculitis, and interstitial lung disease, who had been refractory to many conventional and biologic therapies, responded well to abatacept with good control of the myositis [55].…”

Section: Abataceptmentioning

confidence: 99%

Modern Therapies for Idiopathic Inflammatory Myopathies (IIMs): Role of Biologics

Moghadam-Kia

Oddis

Aggarwal

2016

Clinic Rev Allerg Immunol

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Despite the lack of placebo-controlled trials, glucocorticoids are considered the mainstay of initial treatment for idiopathic inflammatory myopathy (IIMs) and myositis-associated ILD (MA-ILD). Glucocorticoid-sparing agents are often given concomitantly with other immunosuppressive agents, particularly in patients with moderate or severe disease. As treatment of refractory cases of idiopathic inflammatory myopathies has been challenging, there is growing interest in evaluating newer therapies including biologics that target various pathways involved in the pathogenesis of IIMs. In a large clinical trial of rituximab in adult and juvenile myositis, the primary outcome was not met, but the definition of improvement was met by most of this refractory group of myositis patients. Rituximab use was also associated with a significant glucocorticoid-sparing effect. Intravenous immune globulin (IVIg) can be used for refractory IIMs or those with severe dysphagia or concomitant infections. Anti-tumor necrosis factor (anti-TNF) utility in IIMs is generally limited by previous negative studies along with recent reports suggesting their potential for inducing myositis. Further research is required to assess the role of new therapies such as tocilizumab (anti-IL6), ACTH gel, sifalimumab (anti-IFNα), and abatacept (inhibition of T cell co-stimulation) given their biological plausibility and encouraging small case series results. Other potential novel therapies include alemtuzumab (a humanized monoclonal antibody which binds CD52 on B and T lymphocytes), fingolimod (a sphingosine 1-phosphate receptor modulator that traps T lymphocytes in the lymphoid organs), eculizumab, and basiliximab. The future investigations in IIMs will depend on well-designed controlled clinical trials using validated consensus core set measures and improvements in myositis classification schemes based on serologic and histopathologic features.

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Successful treatment of refractory anti-signal recognition particle myopathy using abatacept

Cited by 18 publications

References 10 publications

A Case of Refractory Polymyositis Successfully Treated With Abatacept Monotherapy

A Case of Refractory Polymyositis Successfully Treated With Abatacept Monotherapy

Update on the pharmacological treatment of adult myositis

Modern Therapies for Idiopathic Inflammatory Myopathies (IIMs): Role of Biologics

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