Myositis and myopathy of sarcoidosis: A case report

Burr, Tucker; Kazan-Tannus, Joao Fernando

doi:10.1016/j.radcr.2020.12.014

Cited by 3 publications

(2 citation statements)

References 6 publications

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“…The 16-year-old went into remission with the addition of adalimumab while our patient was lost to follow-up. There are reports of successful treatment of adults with sarcoid myositis with glucocorticoids [ 10 , 27 ] with or without methotrexate [ 28 ] as well as other immune modulating agents, including TNF-antagonists.…”

Section: Discussionmentioning

confidence: 99%

Why so low? An unusual case of myositis in a child

et al. 2023

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Background Sarcoidosis is characterized by non-caseating epithelioid granulomas in various tissues throughout the body, most commonly the lung. Non-caseating granulomas may be seen in skeletal muscle, though typically asymptomatic and under-recognized. While rare in children, there is a need to better characterize the disease and its management. Here we present a 12-year-old female with bilateral calf pain who was ultimately found to have sarcoid myositis. Case Presentation A 12-year-old female presented to rheumatology with significantly elevated inflammatory markers and isolated lower leg pain. MRI of the distal lower extremities demonstrated extensive bilateral myositis with active inflammation, atrophy, and to a lesser extent fasciitis. This distribution of myositis in a child garnered a broad differential requiring a systematic evaluation. Ultimately, muscle biopsy revealed non-caseating granulomatous myositis with perivascular inflammation, extensive muscle fibrosis, and fatty replacement of the muscle with a CD4+ T cell predominant, lymphohistiocytic infiltrate consistent with sarcoidosis. Review of histopathology from age 6 of an extraconal mass resected from her right superior rectus muscle further confirmed the diagnosis. She had no other clinical symptoms or findings of sarcoidosis. The patient improved significantly with methotrexate and prednisone, though flared again after self-discontinuation of medications and was subsequently lost to follow-up. Conclusion This is the second reported case of granulomatous myositis associated with sarcoidosis in a pediatric patient, and the first to present with a chief complaint of leg pain. Increased knowledge of pediatric sarcoid myositis within the medical community will enhance recognition of the disease, improve the evaluation of lower leg myositis, and advance outcomes for this vulnerable population.

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Section: Discussionmentioning

confidence: 99%

Why so low? An unusual case of myositis in a child

et al. 2023

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“…Sarcoidosis is characterized by noncaseating granulomas in affected organs [ 3 ]. Involvement of skeletal muscle is called muscular sarcoidosis or sarcoid myopathy [ 3 , 11 ]. This manifestation infrequently accompanies sarcoidosis and presents with a highly variable clinical phenotype, ranging from acute myositis (in younger patients) to a pseudomyopathic form (in older patients) [ 3 ].…”

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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Isolated muscular sarcoidosis presenting as hypercalcaemic renal failure

Karthik,

Roshan,

Jabbar

et al. 2023

BMJ Case Rep

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The case report describes the details of a man in his 40s admitted for evaluation of renal failure. Biochemical testing revealed parathyroid hormone (PTH) -independent hypercalcaemia. The evaluations for the usual causes such as malignancies, granulomatous diseases, multiple myeloma and vitamin D toxicity were negative. Fluorodeoxyglucose Positron Emission Tomography (FDG-PET) scan identified diffuse uptake in the muscles, and the subsequent muscle biopsy showed non-caseating granulomas suggestive of granulomatous myositis, possibly sarcoidosis, in view of raised ACE levels and the absence of other causes. The patient showed a dramatic response to glucocorticoids, with prompt relief of symptoms and normalisation of serum calcium and creatinine. The case highlights the importance of considering sarcoid myositis in the evaluation of hypercalcaemia and the need for prompt initiation of glucocorticoid therapy to achieve favourable outcomes. The successful use of FDG-PET in diagnosing PTH-independent hypercalcaemia suggests its potential as a valuable tool in the diagnostic algorithm for this condition.

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Myositis and myopathy of sarcoidosis: A case report

Cited by 3 publications

References 6 publications

Why so low? An unusual case of myositis in a child

Why so low? An unusual case of myositis in a child

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

Isolated muscular sarcoidosis presenting as hypercalcaemic renal failure

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