Signal Recognition Antibody–Positive Myopathy and Response to Intravenous Immunoglobulin G (IVIG)

Garcia-Rosell, Melinda; Moore, Suzanne; Pattanaik, Debendra; Menon, Yamini; Bertorini, Tulio E.; Carbone, Laura D

doi:10.1097/rhu.0b013e31828e6442

Cited by 9 publications

(4 citation statements)

References 13 publications

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“…11 In addition, early treatment with IVIG seems to be associated with a more significant improvement in strength. 8,11,13,[42][43][44] Intravenous immunoglobulin as monotherapy has also been successful in treating rare cases of anti-HMGCR antibodyepositive NAM. 27 Rituximab may be especially beneficial in anti-SRP antibodyepositive NAM.…”

Section: Treatmentmentioning

confidence: 99%

Diagnosis and Management of Immune-Mediated Myopathies

Milone

2017

Mayo Clinic Proceedings

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Immune-mediated myopathies (IMMs) are a heterogeneous group of acquired muscle disorders characterized by muscle weakness, elevated creatine kinase levels, and myopathic electromyographic findings. Most IMMs feature the presence of inflammatory infiltrates in muscle. However, the inflammatory exudate may be absent. Indeed, necrotizing autoimmune myopathy (NAM), also called immune-mediated necrotizing myopathy, is characterized by a necrotizing pathologic process with no or minimal inflammation in muscle. The recent discovery of antibodies associated with specific subtypes of autoimmune myopathies has played a major role in characterizing these diseases. Although diagnostic criteria and classification of IMMs currently are under revision, on the basis of the clinical and muscle histopathologic findings, IMMs can be differentiated as NAM, inclusion body myositis (IBM), dermatomyositis, polymyositis, and nonspecific myositis. Because of recent developments in the field of NAM and IBM and the controversies around polymyositis, this review will focus on NAM, IBM, and dermatomyositis.

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Section: Treatmentmentioning

confidence: 99%

Diagnosis and Management of Immune-Mediated Myopathies

Milone

2017

Mayo Clinic Proceedings

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“…High-dose steroids is typically the first-line treatment for autoimmune necrotizing myopathy but risk of relapse is high with steroid therapy alone 6. A few cases have shown that early use of IVIG or plasmapheresis, as used in our case, is associated with more favourable outcome as compared with steroids alone 8. Other immunosuppressive therapies such as methotrexate, azathioprine, rituximab and cyclophosphamide can be used in refractory cases.…”

Section: Discussionmentioning

confidence: 77%

Autoimmune necrotizing myopathy after statin discontinuation

Abid¹,

Watthanasuntorn

Bischof

2019

BMJ Case Rep

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“…In addition, the early use of IVIG or plasmapheresis have been found associated with strength improvement and favorable outcome. 10 Risk of relapse has been been found to be high during medication dose reduction or total discontinuation. 3 Affected patients have been advised against the use of statin in future life as continuation has been found to be associated with worse outcomes.…”

Section: Discussionmentioning

confidence: 99%

Necrotizing Autoimmune Myopathy: A Rare Variant of Idiopathic Inflammatory Myopathies

Khan

Khalid

Ullah

et al. 2017

Journal of Investigative Medicine High Impact Case Reports

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Idiopathic inflammatory myopathies are an unusual group of myopathies with annual incidence of 1 in 100 000 people in the United States. Necrotizing autoimmune myopathy comprises only 16% of this group. It usually presents with severe proximal weakness, lower extremity weakness, and severe fatigue while very rarely does it present with dysphagia and respiratory muscle weakness. Statin use, cancer, and connective tissue disorder are the usual associated risk factors. Anti-signal recognition particle and 3-hydroxy-3-methylglutaryl-coenzyme A reductase are the 2 most common autoantibodies associated with necrotizing autoimmune myopathy. In this article, we present a very rare case of a 66-year-old male who presented with shortness of breath and dysphagia requiring intubation and ventilator support. Creatine kinase was 23 000, myoglobin was 7000, and ANA was positive. All other autoimmune and infectious workup including Lyme disease was unremarkable. Muscle biopsy turned out remarkable for necrotizing myopathy. No evidence of statin use, active malignancy, or connective tissue disease was found. He was treated with high-dose corticosteroids and a short course of intravenous immunoglobulin with very mild improvement in symptoms. Anti-signal recognition particle and 3-hydroxy-3-methylglutaryl-coenzyme A reductase could not be performed as the patient refused to pursue further medical testing. This is a very rare case of idiopathic inflammatory myopathy presenting with bulbar and respiratory muscle weakness requiring ventilator support.

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Signal Recognition Antibody–Positive Myopathy and Response to Intravenous Immunoglobulin G (IVIG)

Cited by 9 publications

References 13 publications

Diagnosis and Management of Immune-Mediated Myopathies

Diagnosis and Management of Immune-Mediated Myopathies

Autoimmune necrotizing myopathy after statin discontinuation

Necrotizing Autoimmune Myopathy: A Rare Variant of Idiopathic Inflammatory Myopathies

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