A case of eosinophilic polymyositis complicated by myasthenia gravis

Ikeda, Yoshio; Tanaka, Makoto; Mizushima, Koichi; Okamoto, Koichi

doi:10.1002/(sici)1097-4598(199810)21:10<1356::aid-mus24>3.0.co;2-z

Cited by 7 publications

(4 citation statements)

References 6 publications

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“…In the present patient, the main inflammation was observed at the orbital periosteum, a finding that was not described in the previous reports. However, the co-occurrence of MG and inflammatory myopathy was described in the previous reports (6). The mechanism underlying these two disorders is unknown; however, an autoimmune process is suspected.…”

Section: Discussionmentioning

confidence: 93%

Idiopathic Orbital Inflammation Appearing on the Affected Side of Preceding Myasthenia Gravis

et al. 2020

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The patient was a 70-year-old man with idiopathic orbital inflammation (IOI) that appeared on the severely affected side of preceding myasthenia gravis (MG). The patient was diagnosed with MG 5 years prior to the onset of IOI. When IOI was diagnosed, an edrophonium test was negative. IOI was considered because he complained of left orbital pain, eyelid swelling, and cerebral MRI exhibited the enhanced lesions along the left orbital periosteum. A biopsy specimen revealed pathological findings compatible with IOI. The administration of corticosteroids was effective for improving the ocular symptoms. IOI should be considered when ocular symptoms deteriorated with soft tissue swelling/pain in MG patients.

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Section: Discussionmentioning

confidence: 93%

Idiopathic Orbital Inflammation Appearing on the Affected Side of Preceding Myasthenia Gravis

et al. 2020

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“…Eosinophils are postulated to cause tissue injury by producing eosinophil‐derived cytotoxic granule proteins such as eosinophilic cationic protein (ECP). Interleukin (IL) 3, IL‐5, and granulocyte macrophage colony–stimulating factor are known as mediators of eosinophilopoiesis,7, 8 and these cytokines also promote the release of ECP from granules in eosinophils. Overproduction of IL‐5 by Th‐2 cells is considered to be the major triggering factor in a subset of eosinophilic disorders 5…”

Section: Discussionmentioning

confidence: 99%

“…Hypereosinophilia has been associated with a wide variety of systemic disorders, including myositis 9, 10, 16. In a minority of patients with myasthenia gravis (MG) associated with thymoma, myositis has been reported 2, 3, 6, 7, 15, 17, 18. The case presented is only the second report of the rare concurrence of hypereosinophilia, myositis, and MG associated with a thymoma.…”

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confidence: 87%

Eosinophilia, myositis, and myasthenia gravis associated with a thymoma

et al. 2006

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Hypereosinophilia has been associated with a wide variety of systemic disorders, including myositis. Myositis develops in a minority of patients with myasthenia gravis associated with a thymoma. We present a patient who developed a life-threatening myopathy in which testing demonstrated the concurrence of hypereosinophilia, myositis, and myasthenia gravis associated with thymoma. Thymoma-associated T-cell abnormalities may well have contributed to this rare association. This case underscores the need to reevaluate constantly the presumed cause of clinical complaints, as more than one cause may be present.

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“…Among the less common etiologies of this finding is eosinophilic polymyositis (EPM). Eosinophilic polymyositis is a rare disease with only a handful of cases [ 1 – 16 ] reported in the literature, mostly in the setting of malignancy [ 11 ], autoimmune disease [ 5 ], genetic abnormalities [ 8 ], hypereosinophilic syndrome (HES) [ 17 ], and even medications [ 1 , 4 , 7 ]. When no etiologic factor can be identified, idiopathic eosinophilic polymyositis is diagnosed.…”

Section: Introductionmentioning

confidence: 99%

Severe Rhabdomyolysis without Systemic Involvement: A Rare Case of Idiopathic Eosinophilic Polymyositis

Farooq

Choksi

Chu

et al. 2015

Case Reports in Rheumatology

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Introduction. Eosinophilic polymyositis (EPM) is a rare cause of rhabdomyolysis characterized by eosinophilic infiltrates in the muscle. We describe the case of a young patient with eosinophilic polymyositis causing isolated severe rhabdomyolysis without systemic involvement. Case Presentation. A 22-year-old Haitian female with no past medical history presented with progressive generalized muscle aches without precipitating factors. Examination of the extremities revealed diffuse muscle tenderness. Laboratory findings demonstrated peripheral eosinophilia and high creatinine phosphokinase (CPK) and transaminase levels. Workup for the common causes of rhabdomyolysis were negative. Her CPK continued to rise to greater than 100,000 units/L so a muscle biopsy was performed which showed widespread eosinophilic infiltrate consistent with eosinophilic polymyositis. She was started on high dose systemic corticosteroids with improvement of her symptoms, eosinophilia, and CPK level. Discussion. This case illustrates a systematic workup of rhabdomyolysis in the presence of peripheral eosinophilia. Many differential diagnoses must be considered before establishing a diagnosis of idiopathic eosinophilic polymyositis. To our knowledge, our case of eosinophilic polymyositis is unique as it presented with severe rhabdomyolysis without another organ involvement. Clinicians should maintain a high index of suspicion for this physically debilitating disease to aid in prompt diagnosis.

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A case of eosinophilic polymyositis complicated by myasthenia gravis

Cited by 7 publications

References 6 publications

Idiopathic Orbital Inflammation Appearing on the Affected Side of Preceding Myasthenia Gravis

Idiopathic Orbital Inflammation Appearing on the Affected Side of Preceding Myasthenia Gravis

Eosinophilia, myositis, and myasthenia gravis associated with a thymoma

Severe Rhabdomyolysis without Systemic Involvement: A Rare Case of Idiopathic Eosinophilic Polymyositis

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