Inclusion body myositis with granuloma formation in muscle tissue

Sakai, Kenji; Ikeda, Yoshihisa; Ishida, Chiho; Matsumoto, Y.; Ono, Kenjiro; Iwasa, Kazuo; Yamada, Masahito

doi:10.1016/j.nmd.2015.06.460

Cited by 11 publications

(12 citation statements)

References 18 publications

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“…In most cases we saw myofiber atrophy with subsequent invasion of connective and adipose tissue. The results are in accordance with observations made by others [12,17,27,36,37]. In a few cases hypertrophic myofibers in IBM patients were also reported [12,27].…”

Section: Discussionsupporting

confidence: 93%

“…In our study the clinical features, including weakness of upper and lower extremities, difficulty in climbing stairs as well as patients' ages were typical and similar to other reports [12,27,34]. Moreover, similarly to Hermanns et al [4] and Sakai et al [17] we observed periodical increases in the levels of CK and LDH enzymes in the studied patients' serum. In patients from a study conducted in China the average value of CK was 397.38 IU/L, whereas for LDH it was 218.21 IU/L [12].…”

Section: Discussionsupporting

confidence: 91%

“…We found that skeletal muscle biopsies of IBM patients presented typical changes which have been well described by others, including hypertrophy of connective and adipose tissues, focal occurrence of necrotic fibers, RVs, a pattern of inflammatory cell infiltrations or pathological protein aggregates [3,4,9,12,17,37]. In most cases we saw myofiber atrophy with subsequent invasion of connective and adipose tissue.…”

Section: Discussionsupporting

confidence: 71%

“…Many authors distinguish four basic subgroups of idiopathic inflammatory myopathies: dermatomyositis (DM), polymyositis (PM), immune-mediated necrotizing myopathy (NM) and IBM [6,7,15,16]. However, from a clinical standpoint s-IBM, is distinguishable from the aforementioned diseases by asymmetric weakness of the finger flexor and knee extensor muscles, atrophy of the quadriceps muscles of the thighs with an inability to fully straighten the knees and resistance to immunosuppressive therapy [3,6,7,17]. It should be emphasized that patients demonstrating clinical symptoms typical for IBM with few inflammatory cells or with the presence of RVs, as seen in a muscle biopsy examination, could be difficult to diagnose in terms of histopathology [6].…”

Section: Introductionmentioning

confidence: 99%

“…The most reliable way of identifying this disease is a muscle biopsy followed by histopathological and immunohistochemical assessment [5,14]. Immunohistochemistry (IHC) is a method which enables the detection of various proteins that accumulate in muscle fibers of patients suffering from s-IBM and that are associated with inflammation processes, autophagy or endoplasmic reticulum stress [7,17,22]. Accumulated proteins have the immunoreactivity of betaamyloid, Tau protein and apolipoproteins [20,[23][24][25].…”

Section: Introductionmentioning

confidence: 99%

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Immunohistochemical and ultrastructural analysis of sporadic inclusion body myositis: a case series

et al. 2018

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Sporadic inclusion body myositis (s-IBM) is a progressive, skeletal muscle disease with poor prognosis. However, establishing the final diagnosis is difficult because of the lack of clear biomarkers in the blood serum and very slow development of clinical symptoms. Moreover, most other organs function normally without any disturbance. Here, in patients with this untreatable disease, we have underlined the importance of immunohistochemical and ultrastructural assessment of skeletal muscle in patients diagnosed with s-IBM. The goal of this study was to identify the distribution of specific antigens and to determine morphological features in order to localize pathological protein aggregates, rimmed vacuoles, and loss of myofibrils, which are key elements in the diagnosis of s-IBM. All studied patients were between 48 and 83 years of age and were hospitalized in the Department of Rheumatology and Internal Medicine between 2011 and 2016. Anamneses revealed an accelerated progression of muscle atrophy, weakness of limb muscles, and difficulties with climbing stairs. Based on histopathology and transmission electron microscopy examination, inflammatory infiltrations consisting of mononuclear cells, severe atrophy and focal necrosis of myofibers, splitting of myofilaments, myelinoid bodies and rimmed vacuoles were observed. Primary antibodies directed against CD3, CD8, CD68, cN1A, beta-amyloid, Tau protein and apolipoprotein B made it possible to identify types of cells within infiltrations as well as the protein deposits within myofibers. Using a combination of immunohistochemistry and electron microscopy methods, we were able to establish the correct final diagnosis and to implement a specific treatment to inhibit disease progression.

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Section: Discussionsupporting

confidence: 93%

Section: Discussionsupporting

confidence: 91%

Section: Discussionsupporting

confidence: 71%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

See 3 more Smart Citations

Immunohistochemical and ultrastructural analysis of sporadic inclusion body myositis: a case series

et al. 2018

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Granulomatous myopathy: Sarcoidosis and beyond

Chompoopong

Liewluck

2022

Muscle and Nerve

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Non‐necrotizing granulomatous inflammation is a rare but easily recognized histopathological finding in skeletal muscle biopsy. A limited number of diseases are known to be associated with non‐necrotizing granulomatous myopathy. Once identified, a careful evaluation for evidence of extramuscular granulomatosis and other signs suggestive of sarcoidosis is warranted as about half of the patients have sarcoid myopathy. In addition, the presence of granulomatous myopathy should trigger a search for clinical and pathological clues of inclusion body myositis (IBM), which accounts for most of the remaining patients and can coexist with sarcoidosis. Recognizing the features of IBM in patients with granulomatous myopathy can potentially spare the patients from unnecessary exposure to immunosuppressive therapies. In patients whose granulomatous myopathy remain unexplained, further investigations should aim at identifying myasthenia gravis and other autoimmune disorders, especially those known to cause granulomatous inflammation in other organs. Laboratory investigations should include acetylcholine receptor, antimitochondrial, antineutrophil cytoplasmic, thyroglobulin, and thyroid peroxidase autoantibodies. In the appropriate clinical context, exposure to immune checkpoint inhibitors and chronic graft‐vs‐host disease can be causes of granulomatous myopathy. In cases of unexplained granulomatous myopathy, natural killer/T‐cell lymphoma should be considered and careful histopathological examination for atypical cells and appropriate immunostaining is crucial. Identifying the etiology of granulomatous myopathy in each patient can guide appropriate treatment.

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