Myopathies featuring non-caseating granulomas: Sarcoidosis, inclusion body myositis and an unfolding overlap

Alhammad, Reem; Liewluck, Teerin

doi:10.1016/j.nmd.2018.10.007

Cited by 19 publications

(27 citation statements)

References 25 publications

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“…Granulomatous myositis is a myopathic syndrome associated with non-specific epithelioid granulomas in striated muscle [ 2 , 4 , 7 , 8 ]. This rare entity is most frequently related to sarcoidosis, but other uncommon causes have been reported [ 4 ].…”

Section: Discussionmentioning

confidence: 99%

A Pediatric Case of Granulomatous Myositis and Response to Treatment

Abdul-Aziz

Sioufi

Pokorny

et al. 2021

Cureus

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Section: Discussionmentioning

confidence: 99%

A Pediatric Case of Granulomatous Myositis and Response to Treatment

Abdul-Aziz

Sioufi

Pokorny

et al. 2021

Cureus

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“…These patients responded poorly to immunosuppressive treatment, similar to IBM [ 18 ]. The presence of congophilic inclusions and p62-positive deposits was associated with a treatment-refractory course of disease in granulomatous myositis [ 1 ]. Further, patients with granulomatous myositis that fulfilled diagnostic criteria for IBM had anti-cN-1A-antibodies in 43% of cases, a frequency comparable to ‘pure’ IBM patients [ 18 ].…”

Section: Muscular Sarcoidosis Granulomatous Myositis and Ibmmentioning

confidence: 99%

Inclusion body myositis and associated diseases: an argument for shared immune pathologies

Nelke

Kleefeld

Preuße

et al. 2022

acta neuropathol commun

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Inclusion body myositis (IBM) is the most prevalent idiopathic inflammatory myopathy (IIM) affecting older adults. The pathogenic hallmark of IBM is chronic inflammation of skeletal muscle. At present, we do not classify IBM into different sub-entities, with the exception perhaps being the presence or absence of the anti-cN-1A-antibody. In contrast to other IIM, IBM is characterized by a chronic and progressive disease course. Here, we discuss the pathophysiological framework of IBM and highlight the seemingly prototypical situations where IBM occurs in the context of other diseases. In this context, understanding common immune pathways might provide insight into the pathogenesis of IBM. Indeed, IBM is associated with a distinct set of conditions, such as human immunodeficiency virus (HIV) or hepatitis C—two conditions associated with premature immune cell exhaustion. Further, the pathomorphology of IBM is reminiscent of other muscle diseases, notably HIV-associated myositis or granulomatous myositis. Distinct immune pathways are likely to drive these commonalities and senescence of the CD8+ T cell compartment is discussed as a possible mechanism of pathogenesis. Future effort directed at understanding the co-occurrence of IBM and associated diseases could prove valuable to better understand the enigmatic IBM pathophysiology.

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“…An association has been observed between sIBM and granulomatous disease. Approximately 1% of patients with histological features of sIBM have granulomas on muscle biopsy, in addition to the classic histological findings of sIBM 21 . A proportion of these patients also have systemic sarcoidosis 21–24 .…”

Section: Sarcoidosismentioning

confidence: 99%

“…Approximately 1% of patients with histological features of sIBM have granulomas on muscle biopsy, in addition to the classic histological findings of sIBM 21 . A proportion of these patients also have systemic sarcoidosis 21–24 . Other patients display the clinical phenotype of sIBM (including both finger flexor and quadriceps weakness) and show granulomas on muscle biopsy (Figure 2C), but lack the histological findings of sIBM 25,26 .…”

Section: Sarcoidosismentioning

confidence: 99%

“…This pattern of finger flexor and quadriceps weakness is not the one commonly observed in sarcoid myopathy, which more typically manifests as a focal myopathy, a proximal myopathy, 27 or asymptomatic granulomas (present in 50–80% of patients with sarcoidosis) 28 . Overall, among patients with granulomatous inflammation on biopsy, those demonstrating clinical or pathological features of sIBM respond poorly to immunotherapy 21,24 . Nonetheless, a trial of corticosteroid treatment in patients with granulomas on muscle biopsy and sIBM‐like phenotypes can be considered, as clinical improvement has been reported in rare patients and may suggest a contribution of granulomatous inflammation to the weakness 25,26 …”

Section: Sarcoidosismentioning

confidence: 99%

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Myopathies with finger flexor weakness: Not only inclusion‐body myositis

2020

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Muscle disorders are characterized by differential involvement of various muscle groups. Among these, weakness predominantly affecting finger flexors is an uncommon pattern, most frequently found in sporadic inclusion-body myositis. This finding is particularly significant when the full range of histopathological findings of inclusion-body myositis is not found on muscle biopsy. Prominent finger flexor weakness, however, is also observed in other myopathies. It occurs commonly in myotonic dystrophy types 1 and 2. In addition, individual reports and small case series have documented finger flexor weakness in sarcoid and amyloid myopathy, and in inherited myopathies caused by ACTA1, CRYAB, DMD, DYSF, FLNC, GAA, GNE, HNRNPDL, LAMA2, MYH7, and VCP mutations. Therefore, the finding of finger flexor weakness requires consideration of clinical, myopathological, genetic, electrodiagnostic, and sometimes muscle imaging findings to establish a diagnosis. K E Y W O R D S distal myopathy, finger flexor weakness, inclusion-body myositis, myopathy with rimmed vacuoles, myotonic dystrophy 2 | SPORADIC INCLUSION-BODY MYOSITIS sIBM is the most common acquired myopathy after 50 years of age. 3 Its pattern of upper limb muscle involvement has been extensively characterized by both clinical examination and imaging, 4-11 and primarily involves finger flexor muscles. The flexor digitorum profundus (FDP) and flexor pollicis longus are most frequently and most severely affected (Figure 1A,B), with weakness demonstrated in approximately 90% of sIBM patients. 5-7 Within the FDP itself, different fingers can be involved to different degrees. 12 The FDP inserts onto the distal

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Myopathies featuring non-caseating granulomas: Sarcoidosis, inclusion body myositis and an unfolding overlap

Cited by 19 publications

References 25 publications

A Pediatric Case of Granulomatous Myositis and Response to Treatment

A Pediatric Case of Granulomatous Myositis and Response to Treatment

Inclusion body myositis and associated diseases: an argument for shared immune pathologies

Myopathies with finger flexor weakness: Not only inclusion‐body myositis

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