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PubMed2002

[A patient of recurrent orbital myositis with good response to high-dose intravenous immunoglobulin (i.v.-i.g.) therapy].

We report a 62-year old woman with orbital myositis who had a favorable response to intravenous immunoglobulin (i.v.-IG) administration in preventing disease recurrence. She had been suffering from frequent relapses of swelling and redness of the left eye with increasing pain and diplopia caused by restricted eye movement of the left eyeball. T2-weighted magnetic resonance image of the orbit showed enlargement of the left medial rectus muscle. She was treated with 1 g of methylprednisolone per day for 3 days. One mg/kg per day of oral prednisolone was subsequently started with non-steroid anti-inflammatory drugs, which resulted in improvement. However, her symptoms were aggravated while the drug was tapered off even though she used a high dose of oral prednisolone. High dose of i.v.-IG (400 mg/kg per day) was then administered for five days. Since the treatment, she has been free from recurrences of the disease for over one year, suggesting that i.v.-IG can prevent the recurrence of orbital myositis. Some reports have suggested that i.v.-IG treatment is useful to prevent recurrence of other forms of inflammatory myositis, such as dermatomyositis or polymositis. Overproduction of the pro-inflammatory cytokines (IL-1, IL-1 beta, INF-alpha) and Th1 cytokines (INF-gamma, IL-2) are related to the deterioration of these diseases. I.v.-IG treatment suppresses the production of the pro-inflammatory cytokines. In our case, serum levels of the pro-inflammatory cytokines were all normal, but the IL-4 level was elevated after the i.v.-IG treatment, suggesting that orbital myositis was probably related to the Th1 dominant disease that was suppressed by IL-4.

Dose-Response Relationship, Drug↗
PubMed1998

Association of HLA class I and class II alleles with myositis in Japanese patients.

OBJECTIVE: To investigate the correlation of HLA class I and class II antigens and alleles with various forms of myositis in Japanese patients. METHODS: Eighty-four Japanese patients with myositis [22 with polymyositis (PM), 46 with dermatomyositis (DM), 16 with myositis overlapping with other collagen vascular diseases] were typed serologically for HLA-A, B, C antigens. HLA-DRB1, DQA1, and DQB1 alleles were determined by polymerase chain reaction dependent DNA typing methods. Fifty-eight Japanese controls were typed serologically while HLA-DRB1, DQA1, and DQB1 allele typing was carried out in 175, 95, and 104 controls, respectively. RESULTS: HLA-B7 was higher in patients than controls [20.2 vs 6.9% in controls: p=0.02, odds ratio (OR)=3.4]. The increase of HLA-B7 was largely dependent on the increase in overlap patients (37.5%; p=0.005, OR=8.1). HLA-A24 and B52 were significantly decreased in PM as compared to DM, while CW3 was significantly increased in PM versus DM. DRB1*08 alleles were significantly increased in patients (36.9 vs 20.5% in controls; p=0.004, OR=2.3), especially in PM and DM. DQA1*0501 and DQB1*0301 were significantly decreased in patients [4.8 vs 13.7% in controls; p=0.04, OR=0.32, and 8.3 vs 20.2% in controls; p=0.02, OR=0.36, respectively]. CONCLUSION: HLA-class I and class II alleles associated with Japanese patients with myositis may be different from those associated with Caucasian patients.

Alleles↗
PubMed1998

[Focal myositis: report of a case in pediatric age and review of the literature].

INTRODUCTION AND CLINICAL CASE: We present the case of a 13 years old boy with a year-old history of a painful tumour on the external and distal third of the right thigh. The imaging tests suggested a soft tissue tumour but the muscle biopsy revealed the typical findings of focal myositis. Focal myositis is a benign inflammatory pseudotumour of the skeletal muscle recognized as a distinct clinicopathological entity, but there have been descriptions of the cases initially diagnosed as focal myositis that have latter behaved as evident polymyositis. We have not found laboratory data to support a diagnosis of polymyositis in our patient. CONCLUSION: Although focal myositis is considered a self-limited disease, we believe a long-term clinical and biochemical follow-up is warranted, to confirm the absence of recurrence signs and/or the development of a generalized myopathy.

Adolescent↗
PubMed1982

Viral studies in benign acute childhood myositis.

Thirty-five cases of benign acute childhood myositis followed infections with influenza A or B. Two children had recurrent myositis associated with infections of different influenza types. None of the children had acute-phase antibody titers to the infecting type of influenza. These results are consistent with the proposal that benign acute childhood myositis can occur only with the initial infection of a viral type.

Acute Disease↗
PubMed1991

Familial inclusion body myositis among Kurdish-Iranian Jews.

We report two cases of adult-onset, slowly progressive limb-girdle muscle weakness with a remarkable sparing of quadriceps muscles that developed in patients from different families of Iranian-Kurdish-Jewish origin. Each patient had a similarly affected sibling. The findings by means of muscle biopsies showed abnormalities typical of inclusion body myositis, including abundant lined vacuoles and characteristic cytoplasmic inclusions of 15- to 18-nm filaments. Remarkably, many vacuolated muscle fibers showed immunoreactivity to neural cell adhesion molecule, a fetal muscle antigen. The common origin of these patients from an isolated ethnic group with frequent consanguinity and the familial incidence is indicative of a genetic causation or predisposition, probably with an autosomal recessive inheritance. This familial myopathy is one of several clinical syndromes that share the typical pathological findings of inclusion body myositis. The pathogenic relationship between these different familial forms and the more common sporadic form of inclusion body myositis is not known.

Adult↗
PubMed2002

Orbital myositis posing as cluster headache.

OBJECTIVE: To describe the case of a patient with recurrent orbital myositis who was thought to have cluster headaches for 6 years. DESIGN AND SETTING: Case report in an outpatient neuro-ophthalmology clinic. PATIENT: A 24-year-old man developed unilateral supraorbital pain, lacrimation, conjunctival hyperemia, nasal congestion, proptosis, and painful eye movements. The pain intensity varied over the course of each day and disappeared after 1 month. He had multiple attacks responsive to prednisone that were separated by months over the ensuing 6 years. Neuroimaging revealed an enlarged extraocular muscle. CONCLUSIONS: Overlap in symptoms between recurrent orbital myositis and cluster headache delayed the diagnosis in this patient. Orbital myositis should be considered in patients with atypical cluster headache characterized by proptosis, painful eye movements, and pain that does not completely resolve after 3 hours.

Adult↗
PubMed1982

Idiopathic orbital myositis: report of six cases.

Idiopathic orbital myositis, a subgroup of inflammatory orbital pseudotumor, occurred in six patients with acute onset periorbital pain, diplopia, and in most cases, eyelid swelling. Proptosis, ductional restrictions, and responsiveness to oral prednisone administration were characteristic. Computed tomography and orbital ultrasonography showed enlargement of one or more extraocular muscles, with sparing of other orbital soft tissues. One patient had involvement of all recti muscles in both orbits. The distinction of orbital myositis from Graves' ophthalmopathy is made on clinical grounds, with acute orbital pain and corticosteroid responsiveness being uncharacteristic of the latter. The possibility of orbital myositis being an immune-mediated process is discussed.

Adult↗
PubMed1983

Orbital inflammation, myositis, and systemic lupus erythematosus.

A 39-year-old man with systemic lupus erythematosus (SLE) experienced three episodes of acute orbital inflammation, a primary feature of each being a markedly enlarged extraocular muscle (myositis). The muscles of the upper trunk were concomitantly inflamed, and full-thickness skin and muscle biopsy specimens demonstrated an inflammatory myositis. The case demonstrates that orbital proptosis and extraocular myositis can be features of SLE.

Adult↗
PubMed1991

Focal myositis. A new cause for the pediatric neck mass.

Focal myositis is an inflammatory pseudotumor of skeletal muscle that may involve the head and neck. It can closely mimic either a neoplastic or infectious process. We present the case of a 7-year-old boy with a 2-week history of a painful, unilateral neck swelling, fever, and torticollis. He failed to respond to antibiotic therapy and required an open neck exploration. An incisional biopsy of the indurated, inflammatory tissue adherent to his sternocleidomastoid muscle showed focal myositis. Focal myositis is an unusual, but important possibility in the differential diagnosis of any neck mass and especially one in a child. Although its cause is unknown, it is a self-limited disease and neither excisional biopsy nor radical resection is justified.

Abscess↗
PubMed1999

Focal myositis presenting with radial nerve palsy.

Focal myositis is a rare inflammatory pseudotumor of skeletal muscle which usually has a benign course. We report a 56-year-old woman with a painful mass in the left arm with a radial nerve palsy. Magnetic resonance imaging (MRI) of the left arm showed a mass in the triceps muscle that was suggestive of a soft-tissue sarcoma. Electromyography showed a severe radial neuropathy involving both motor and sensory axons. An open biopsy showed focal myositis. Treatment with corticosteroids resulted in complete disappearance of the mass clinically and by MRI, without recurrence for more than 2 years. Radial nerve function also recovered completely. As a treatable cause of focal neuropathy, focal myositis should be included in the differential diagnosis of a muscle mass.

Electromyography↗
PubMed1983

Proliferative myositis of the masseter muscle. A case report and a review of the literature.

A case of proliferative myositis of the masseter muscle in a 62-year-old woman is presented and pertinent literature is reviewed. This benign pseudosarcomatous proliferative lesion has not been previously reported in any of the muscles of mastication. Clinically the patient presented with pain and difficulty in opening of the mouth. On initial examination, a malignant salivary gland tumor was suspected. The proliferative myositis exhibits a disturbing microscopic appearance characterized by the presence of numerous hyperchromatic fibroblasts and oval or spindle-shaped giant cells which resemble malignant rhabdomyoblasts. These lesions have been often misdiagnosed as rhabdomyosarcoma and other types of sarcomas. Awareness of the microscopic characteristics of proliferative myositis and the possibility of its occurrence in the perioral soft tissues is important in avoiding misdiagnosis of this benign condition.

Cell Division↗
PubMed1998

Frequent occurrence of anti-tRNA(His) autoantibodies that recognize a conformational epitope in sera of patients with myositis.

OBJECTIVE: To investigate the incidence of autoantibodies directed to deproteinized transfer RNA(His) (tRNA(His)) in anti-Jo-1 positive myositis patients and to determine the major B cell epitope. METHODS: One hundred sixty-seven myositis sera were screened by immunoblotting and enzyme-linked immunosorbent assay for the presence of anti-Jo-1 antibody. Autoantibodies directed to deproteinized RNA were detected by immunoprecipitation. Ribonuclease cleavage experiments were performed to determine the tRNA(His)-specific features important for recognition. RESULTS: Approximately one-third of the anti-Jo-1 positive sera also contained autoantibodies recognizing tRNA(His). This recognition was independent of modified bases, but the presence of stabilizing Mg2+ ions appeared to be essential for efficient immunoprecipitation. Transfer RNA(His)-specific features in the anticodon loop were not protected from ribonuclease cleavage by bound antibodies, while protection of bases located in the D and T loops was observed. CONCLUSION: A significant number of anti-Jo-1 positive myositis sera contain anti-tRNA(His) activity. Formation of the major autoepitope on tRNA(His) is strongly dependent on proper folding of this molecule mediated by an interaction between D and T loops which is stabilized by either modified residues or Mg2+ ions.

Autoantibodies↗
PubMed1984

Monoclonal antibody analysis of mononuclear cells in myopathies. II: Phenotypes of autoinvasive cells in polymyositis and inclusion body myositis.

In 6 cases of polymyositis and 6 of inclusion body myositis, phenotypes of mononuclear cells focally surrounding and invading muscle fibers were analyzed. By localizing the T8, T4, and Ia markers with direct immunofluorescence and acid phosphatase enzyme cytochemically in the same sections, five different phenotypes were simultaneously identified in a given section: T8+ and T4+ cells that were either activated (Ia+) or not activated (Ia-), and acid phosphatase--reactive and Ia+ macrophages. This approach permitted the separate and quantitative assessment of the distributions of the different phenotypes among the invading versus the surrounding cells. In both polymyositis and inclusion body myositis, the invading cells were selectively enriched in the T8+ phenotype. One-third of all invading cells and one-half of the invading T8+ cells were activated. T4+ cells were more abundant among the surrounding than the invading cells, and only a small proportion of the T4+ cells were activated. These findings are especially significant in view of the cytotoxic capability of the T8+ cells and because histocompatibility factors permit T8+ but not T4+ cells to recognize an antigen on muscle fibers. Macrophages accounted for 21 to 31% of the cells invading or surrounding nonnecrotic fibers. For purposes of comparison, we also analyzed mononuclear cells in necrotic fibers: 80% of these cells were macrophages, and only 20% were T cells. The findings indicate that in polymyositis and inclusion body myositis, nonnecrotic muscle fibers are injured by autoinvasive T8+ cells that act in concert with macrophages. Further, the findings strongly imply previous sensitization of clones of T cells to muscle fiber-associated surface antigen(s).

Adolescent↗
PubMed1989

Clinical heterogeneity and treatment response in inclusion body myositis.

Inclusion body myositis has been described as an inflammatory myopathy with distinctive clinical and pathologic features that is refractory to treatment. Ten cases of inclusion body myositis, as defined by histopathologic findings, were reviewed to determine whether the clinical characteristics are different in patients whose disease has been defined by light and electron microscopic studies compared with those whose disease has been defined by light microscopic studies alone. The clinical characteristics of both groups of patients were similar, and 2 patients have had excellent responses to treatment. Although inclusion body myositis represents a histologic subset of polymyositis, from a clinical perspective, it must be considered a nonspecific designation. Despite a generally poor prognosis, therapeutic intervention is still warranted.

Adult↗
PubMed2002

Differences in idiopathic inflammatory myopathy phenotypes and genotypes between Mesoamerican Mestizos and North American Caucasians: ethnogeographic influences in the genetics and clinical expression of myositis.

OBJECTIVE: As part of a larger, worldwide study of the ethnogeography of myositis, we evaluated the clinical, serologic, and immunogenetic features of Mestizo (Mexican and Guatemalan) and North American Caucasian patients with idiopathic inflammatory myopathy (IIM). METHODS: Clinical manifestations, autoantibodies, HLA-DRB1 and DQA1 alleles, and immunoglobulin Gm/Km allotypes were compared between 138 Mestizos with IIM and 287 Caucasians with IIM, using the same classification criteria and standardized questionnaires. RESULTS: IIM in Mestizo patients was characterized by a higher proportion of dermatomyositis (69% of adult Mestizos versus 35% of adult Caucasians; P < 0.001) and anti-Mi-2 autoantibodies (30% versus 7% of adults, respectively, and 32% versus 4% of children, respectively; P < 0.01). Genetic risk factors also differed in these populations. Whereas Mestizos had no HLA risk factors for IIM, HLA-DRB1*0301, the linked allele DQA1*0501, and DRB1 alleles sharing the first hypervariable region motif (9)EYSTS(13) were major risk factors in Caucasian patients with IIM. Furthermore, different HLA-DRB1 and DQA1 alleles were associated with anti-Mi-2 autoantibodies (DRB1*04 and DQA1*03 in Mestizos and DRB1*07 and DQA1*02 in Caucasians). Immunoglobulin gamma-chain allotypes Gm(1), Gm(17) (odds ratio for both 11.3, P = 0.008), and Gm(21) (odds ratio 7.3, P = 0.005) and kappa-chain allotype Km(3) (odds ratio 7.3, P = 0.005) were risk factors for IIM in Mestizos; however, no Gm or Km allotypes were risk or protective factors in Caucasians. In addition, Gm and Km phenotypes were unique risk factors (Gm 1,3,17 5,13,21 and Gm 1,17 23 21 and Km 3,3) or protective factors (Km 1,1) for the development of myositis and anti-Mi-2 autoantibodies (Gm 1,2,3,17 23 5,13,21) in adult Mestizos. CONCLUSION: IIM in Mesoamerican Mestizos differs from IIM in North American Caucasians in the frequency of phenotypic features and in the immune-response genes predisposing to and protecting from myositis and anti-Mi-2 autoantibodies at 4 chromosomal loci. These and other data suggest the likelihood that the expression of IIM is modulated by different genes and environmental exposures around the world.

Adult↗
PubMed1992

Antibodies to glycyl-transfer RNA synthetase in patients with myositis and interstitial lung disease.

OBJECTIVE: We have previously described anti-EJ antibodies, and provided evidence that these antibodies react with glycyl-transfer RNA (gly-tRNA) synthetase. The aim of the present study was to identify patients with anti-EJ antibodies and describe the clinical associations of the antibody, in particular, whether it is associated with the syndrome of myositis and interstitial lung disease (ILD) that has been previously associated with autoantibodies to the aminoacyl-tRNA synthetases for histidine, threonine, and alanine. METHODS: Sera from patients with suspected or proven polymyositis or dermatomyositis (DM), sera with anticytoplasmic patterns, and control sera were tested for anti-EJ antibodies by immunoprecipitation (IPP). Positive sera and controls were tested for the ability to inhibit gly-tRNA synthetase by preincubation of the enzyme source with the serum. RESULTS: Anti-EJ antibodies were demonstrated in the sera of 5 patients, by IPP of characteristic tRNAs and protein. Original serum EJ and each of the new sera significantly inhibited the enzymatic activity of gly-tRNA synthetase but not histidyl-tRNA synthetase. All 5 of the new patients had inflammatory myopathy, a typical DM rash, and ILD. One, who had an overlap syndrome with systemic lupus erythematosus, had anti-EJ at least 4 months before the development of clinical myositis. Arthritis and Raynaud's phenomenon, other features associated with antisynthetases, were also seen. CONCLUSION: Anti-EJ is associated with the syndrome of myositis and lung disease that is seen in association with other antisynthetases. The finding of specific inhibition of gly-tRNA synthetase by all anti-EJ-positive sera strongly supports the identification of EJ antigen as gly-tRNA synthetase.

Adult↗
PubMed2004

Myositis in a patient with large granular leukocyte leukemia.

We describe the case of a 58-year-old patient with subacute progressive weakness in both legs accompanied by recurrent opportunistic infections. White cell count was normal, but immunophenotyping revealed an increased number of CD8(+) T cells and deficiency of natural killer cells, B cells, and CD4(+) T cells in the peripheral blood. Large granular leukocyte (LGL) leukemia was diagnosed based on a clonal T-cell receptor rearrangement. Muscle biopsy demonstrated severe myositis with extensive CD8(+) T-cell infiltrates. Since no evidence of microbial muscle infection was found, autoimmune myositis was diagnosed. Immunosuppressive treatment resulted in clinical improvement and normalization of creatine kinase (CK) serum levels. The immunological phenotype of the patient and the positive response to treatment adds further to the concept that CD8(+) T cells mediate disease in autoimmune myositis.

CD4-Positive T-Lymphocytes↗
PubMed2006

Eosinophilia, myositis, and myasthenia gravis associated with a thymoma.

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.

Aged↗