Anti-mitochondrial antibody-associated myositis with eosinophilia and dropped head

Shimizu, Hayato; Nishino, Ichizo; Ueda, Tetsuhiro; Kohara, Nobuo; Nishioka, Hiroaki

doi:10.1016/j.ensci.2018.05.004

Cited by 4 publications

(5 citation statements)

References 7 publications

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“…Since corticosteroid therapy improved both the abnormal skeletal muscle findings and ocular symptoms, the skeletal and ocular symptoms may have shared inflammatory pathogenesis. Good responsiveness to corticosteroid therapy is consistent with previous reports on myositis with anti-mitochondrial antibody type 2 (3,5,9). Although we did not perform an EMG study after corticosteroid treatment, the EMG findings may have been normalized by treatment.…”

Section: Discussionsupporting

confidence: 91%

“…The EMG findings were suggestive of a myopathy pattern, while the MRI findings revealed ocular muscle swelling and abnormally high intensities of skeletal muscles. Generally, myositis with anti-mitochondrial antibodies type 2 is characterized by slow-progressive limb, cardiac, and respiratory muscle weaknesses; arrhythmia; liver dysfunction with or without primary biliary cirrhosis; and increased CK levels (5)(6)(7)(8)(9). Our patient did not present with muscle weakness, disturbances of the respiratory or cardiac muscles, or complications of PBC.…”

Section: Discussionmentioning

confidence: 65%

“…Anti-mitochondrial antibody type 2 is an antibody against mitochondria used as a diagnostic marker of primary biliary cirrhosis (PBC) (1,2); furthermore, patients positive for anti-mitochondrial antibody type 2 may present with myositis (3,4). Therefore, myositis with anti-mitochondrial antibody type 2 is often characterized by muscle weakness of the four extremities, cardiac involvement, and dyspnea (5)(6)(7)(8)(9). Although Magrini et al reported PBC patient with ocular myositis (10), there has been few reported cases involving ocular paresis.…”

Section: Introductionmentioning

confidence: 99%

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Myositis with Anti-mitochondrial Antibody Type 2 with Diplopia and Ptosis

et al. 2021

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Anti-mitochondrial antibody type 2 is a diagnostic marker of primary biliary cirrhosis and complicates myositis. Myositis with anti-mitochondrial antibody type 2 is clinically characterized by slowly progressive limb, cardiac, and respiratory muscle weakness as well as serum creatinine kinase elevations. However, there has been few cases with eye symptoms. We herein report a 59-year-old woman with anti-mitochondrial antibody type 2 who presented with diplopia and ptosis. Magnetic resonance imaging revealed bilateral ocular muscle enlargement and abnormally high intensities in the lower limb muscles. Corticosteroid therapy improved these symptoms. Myositis with anti-mitochondrial antibody type 2 can present with eye symptoms.

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Section: Discussionsupporting

confidence: 91%

Section: Discussionmentioning

confidence: 65%

Section: Introductionmentioning

confidence: 99%

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Myositis with Anti-mitochondrial Antibody Type 2 with Diplopia and Ptosis

et al. 2021

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“…Other autoantibodies falling into the group of MAAs have been associated with certain disease courses, and pathological presentations e.g. anti-mitochondrial M2 antibodies in granulomatous diseases (56) and necrotizing myopathy (Tables 1,2). However most of them like anti-PmSCL or anti-SSa or SSb and U1RNP have been described regularly in certain diseases like sclerodermia, Sjögren Syndrome, mixed connective tissue diseases etc., and we hypothesize that myositis may occur during these diseases rather than the antibodies occurring with myositis.…”

Section: Autoantibodies As Biomarkersmentioning

confidence: 99%

Biomarkers in Inflammatory Myopathies—An Expanded Definition

2019

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Biomarkers as parameters of pathophysiological conditions can be of outmost relevance for inflammatory myopathies. They are particularly warranted to inform about diagnostic, prognostic, and therapeutic questions. As biomarkers become more and more relevant in daily routine, this review focusses on relevant aspects particularly addressing myopathological features. However, the level of evidence to use them in daily routine at presence is low, still since none of them has been validated in large cohorts of patients and rarely in independent biopsy series. Hence, they should be read as mere expert opinions. The evaluation of biomarkers as well as key biological parameters is an ongoing process, and we start learning about relevance of them, as we must recognize that pathophysiology of myositis is biologically incompletely understood. As such this approach should be considered an essay toward expansion of the definition “biomarker” to myositis, an emerging field of interest in biomedical research.

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“…Some studies showed a lower chance of limb weakness in AMA-positive myositis, compared to AMA-negative ones [ 6 ]. Paravertebral muscle atrophy presented by head drop [ 7 ] or rectus abdominis involvement without weakness [ 8 ] may be a clinical feature of AMA-positive myositis. This may indicate torso muscle biopsy, other than biceps brachii or quadriceps femoris, to confirm the diagnosis.…”

Section: Discussionmentioning

confidence: 99%

Pathological findings with vacuoles in anti-mitochondrial antibody-positive inflammatory myopathy

Chen,

Zhang,

et al. 2024

BMC Musculoskelet Disord

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Background A few patients with inflammatory myopathy showed anti-mitochondrial antibody (AMA) positivity. This study aimed to report the clinical and pathological findings with vacuoles in 3 cases of such patients. Methods Three cases with myositis from the Myositis Clinical Database of Peking University First Hospital were identified with AMA positivity. Their clinical records were retrospectively reviewed and the data was extracted. All the 3 cases underwent muscle biopsy. Results Three middle-aged patients presented with chronic-onset weakness of proximal limbs, marked elevation of creatine kinase, and AMA-positivity. Two of the 3 cases meet the criteria of primary biliary cholangitis. All the 3 cases presented with cardiac involvement and proteinuria. Two cases developed type 2 respiratory failure. MRI of the thigh muscle showed multiple patches of edema bilaterally in both cases, mostly in the adductor magnus. Pathological findings include degeneration of muscle fibers, diffused MHC-I positivity, and complement deposits on cell membranes. Vacuoles without rims of different sizes were discovered under the membrane of the muscle fibers. A few RBFs were discovered in case 1, while a diffused proliferation of endomysium and perimysium was shown in case 2. Conclusions AMA-positive inflammatory myopathy is a disease that could affect multiple systems. Apart from inflammatory changes, the pathological findings of muscle can also present vacuoles.

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Anti-mitochondrial antibody-associated myositis with eosinophilia and dropped head

Cited by 4 publications

References 7 publications

Myositis with Anti-mitochondrial Antibody Type 2 with Diplopia and Ptosis

Myositis with Anti-mitochondrial Antibody Type 2 with Diplopia and Ptosis

Biomarkers in Inflammatory Myopathies—An Expanded Definition

Pathological findings with vacuoles in anti-mitochondrial antibody-positive inflammatory myopathy

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