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PubMed1998

Fibrodysplasia (myositis) ossificans progressiva. Clinical lessons from a rare disease.

Fibrodysplasia (myositis) ossificans progressiva is a rare dominantly inherited disorder, in which defects in skeletal patterning particularly affecting the big toes, are associated with progressive endochondral ossification of the large striated muscles in a specific order leading to prolonged disability. A recent series of 28 patients studied for as many as 24 years exemplifies the presentation and course of this disease. Painful swelling of muscles (myositis) leading to ossification began at a mean age of 4.6 years (range, 0-16 years) initially in the neck and upper spine (in 25 subjects) and later around the hips, other major joints, and jaw. The rate and extent of disability was unrelated to the time of onset. No form of treatment produced consistent benefit. Despite the unique clinical features, the initial diagnosis of fibrodysplasia ossificans progressiva was often wrong and usually considerably delayed. Mistaken histologic diagnoses such as soft tissue sarcoma or fibromatosis could lead to inappropriate treatment.

Adolescent↗

PubMed1977

Alternating proptosis. A case report of acute orbital myositis defined by the computerized tomographic scan.

A 27-year-old woman had a two-week course of acute painful right proptosis with ptosis, medial conjunctival injection, and restriction of eye movements--particularly abduction. One month later, a similar remitting painful left proptosis with complete limitation of abduction developed. Computerized tomographic x-ray scanning showed marked contrast enhancement of both medial rectus muscles, documenting the presumptive diagnosis of acute orbital myositis without recourse to invasive diagnostic techniques. Myositis is a common component of idiopathic orbital inflammatory disease (orbital pseudotumor), but awareness of the rare patient with acute inflammation clinically localized to the extraocular muscles will decrease confusion with cranial nerve and cavernous sinus disease.

Acute Disease↗

PubMed1980

Myositis, myoglobinemia, and myoglobinuria associated with enterovirus echo 9 infection.

A 30-year-old man had a "flu-like" syndrome followed by clinical and laboratory evidence of myositis localized in both thighs. Serum neutralization test results were compatible with enterovirus echo 9 infection. Severe myoglobinuria dominated the clinical picture. We discuss other possible manifestations of enterovirus echo 9 infections. To our knowledge, this report is the first describing myositis caused by enterovirus echo 9.

Echovirus 9↗

PubMed1984

Inclusion body myositis and chronic immune thrombocytopenia.

We report a case of inclusion body myositis and chronic immune thrombocytopenia with serum platelet antibodies and circulating immune complexes. Although immune mechanisms probably play an important role in the pathogenesis of inclusion body myositis, a viral etiology cannot be excluded.

Antibodies↗

PubMed1987

Phorbol ester-induced orbital myositis.

A single injection of TPA (12-0-tetradecanoyl-phorbol-13-acetate) into the superior rectus muscle of New Zealand white rabbits produced inflammation and edema, followed by fibrosis and muscle restriction. Strain gauge measurements showed a 60% increase in muscle restriction at 24 hours and an increase greater than 400% at 12 weeks. Clinical and histologic characteristics paralleled those of human idiopathic orbital myositis and thyroid orbital myopathy. The combination of initial muscular inflammation and enlargement, subsequent fibrosis and restricted extraocular motility, and histologic evidence of these changes creates a useful model of orbital myositis.

Animals↗

PubMed1979

Primary suppurative myositis in children.

Primary suppurative myositis is rare in the United States when compared with the incidence of disease in the tropics. Clinically, it may mimic many of the more common diseases, such as hematoma, osteomyelitis, arthritis, or appendiceal abscess. It usually has a benign course, with complete recovery after appropriate treatment. Prolonged morbidity and an increased mortality may result from unfamiliarity with this entity. Six children had primary suppurative myositis; one died as a result of perforation of a psoas abscess into the dural sac causing staphylococcal meningitis.

Child↗

PubMed1977

Myositis ossificans traumatica. Association with hemophilia (factor XI deficiency) in a football player.

A case report of an outstanding college football halfback with partial factor XI deficiency, recurrent ecchymosis, and myositis ossificans is reviewed. The association of multiple areas of myositis ossificans and partial clotting-factor deficiency has not been noted in the past. The importance of considering this diagnosis in participants in contact sports with the above findings is emphasized.

Adult↗

PubMed2000

Fine-needle aspiration findings in nodular myositis: a case report.

Fine-needle aspiration (FNA) is frequently the initial diagnostic modality for the workup of suspected metastatic disease in subcutaneous or deeper soft tissues. The technique is less well-accepted for the diagnosis of primary soft-tissue lesions. Occasionally during the investigation of suspected metastatic disease, primary processes of the soft tissues will undergo FNA. Thus, familiarity with the cytomorphology of soft-tissue reparative and neoplastic lesions is important. A variety of reparative and degenerative entities exist within the soft tissues, one of which is localized nodular myositis. This lesion may be a precursor of generalized polymyositis, including polymyositis associated with graft vs. host disease, or it may remain a localized process. Aspirates from localized nodular myositis demonstrate fragments of striated muscle including degenerating and regenerative myocytes, a mixed inflammatory background, and necrotic debris. Cytologic identification of this lesion can be therapeutically important, particularly in post-bone marrow transplant patients.

Adult↗

PubMed2004

Creutzfeldt-Jakob disease and inclusion body myositis: abundant disease-associated prion protein in muscle.

Pathologicalprion protein (PrP(Sc)) is the hallmark of prion diseases affecting primarily the central nervous system. Using immunohistochemistry, paraffin-embedded tissue blot, and Western blot, we demonstrated abundant PrP(Sc) in the muscle of a patient with sporadic Creutzfeldt-Jakob disease and inclusion body myositis. Extraneural PrP(C)-PrP(Sc) conversion in Creutzfeldt-Jakob disease appears to become prominent when PrP(C) is abundantly available as substrate, as in inclusion body myositis muscle.

Aged↗

PubMed1981

Association of myositis and myocarditis in progressive systemic sclerosis.

A retrospective review of 47 patients with progressive systemic sclerosis revealed a highly significant (P less than 0.001) association between myositis and myocarditis. Three patients developed clinically overt myocarditis as an early feature of their disease. These patients had an inflammatory myopathy indistinguishable from polymyositis. Despite an excellent clinical response to steroids, the late occurrence of life-threatening conduction system defects appeared while they were receiving therapy. Consequently, patients with progressive systemic sclerosis and myositis should be evaluated for a coexisting myocarditis. The findings in these patients lend further support to the concept of a generalized myopathic process among the connective tissue diseases.

Adolescent↗

PubMed1984

Rheumatoid myositis. Clinical and histologic features and possible pathogenesis.

Thirty-one patients with rheumatoid arthritis were consecutively studied for evidence of muscle involvement, using muscle biopsy and electromyography. The patients were initially separated into 4 clinical categories: inactive peripheral joint disease (6 patients); active peripheral joint disease (10 patients); systemic disease and a disproportionately elevated sedimentation rate for the degree of mild synovitis (SERD) (11 patients); or elevated creatinine phosphokinase level (4 patients). In addition to routine histology, muscle tissue was examined for de novo synthesis of IgM and IgM rheumatoid factor, and by indirect immunofluorescence for the presence of immunoglobulin and complement deposits. Our results indicate that: muscle fiber necrosis occurs frequently in patients with rheumatoid arthritis; rheumatoid myositis, defined as muscle fiber necrosis and mononuclear cell infiltration, is a distinct entity and occurs particularly in patients with SERD or an elevated creatinine phosphokinase level; and only muscle from patients with rheumatoid myositis exhibited de novo synthesis of rheumatoid factor and significant quantities of IgM, indicating that local immune events may be important in the pathogenesis of this entity.

Arthritis, Rheumatoid↗

PubMed1988

Antibody to threonyl-transfer RNA synthetase in myositis sera.

The prevalence and clinical correlations of anti-threonyl-transfer RNA synthetase (anti-PL-7), as well as the relationship of anti-PL-7 to anti-histidyl-transfer RNA synthetase (anti-Jo-1) were studied in 109 sera from patients with myositis. Inhibition of threonine aminoacylation was used to screen for anti-PL-7. Sera from 3 patients, 2 with polymyositis and 1 with polymyositis-overlap syndrome, and a fourth serum from a patient with dermatomyositis, which was previously found to contain anti-PL-7, inhibited greater than 90% of activity (3.7% of 109 sera). All 4 sera reacted strongly in an enzyme-linked immunosorbent assay with enzyme that was either affinity purified with anti-PL-7 or was biochemically purified. There was no indication of cross-reactivity by aminoacylation inhibition or, for most sera, by enzyme-linked immunosorbent assay. Anti-PL-7 is an uncommon myositis-associated antibody that is independent of anti-Jo-1, but is directed at a functionally related enzyme.

Acylation↗

PubMed1996

Focal myositis presenting as pseudothrombophlebitis of the neck in a patient with mixed connective tissue disease.

This report describes a case of focal myositis in a patient with mixed connective tissue disease. The patient presented with diffuse neck swelling and pseudothrombophlebitis of the left internal jugular vein. Other clinical features included a high fever, elevated erythrocyte sedimentation rate, and prompt improvement after administration of high-dose intravenous corticosteroid therapy. Criteria for polymyositis were absent, serum levels of creatine kinase remained normal, and there was no sign of recurrence during 3 years of followup. Results of immunoprecipitation for anti-Jo-1 and other myositis-specific autoantibodies remained negative in serial serum samples obtained before, during, and after the episode.

Adult↗

PubMed2006

HLA polymorphisms in African Americans with idiopathic inflammatory myopathy: allelic profiles distinguish patients with different clinical phenotypes and myositis autoantibodies.

OBJECTIVE: To investigate possible associations of HLA polymorphisms with idiopathic inflammatory myopathy (IIM) in African Americans, and to compare this with HLA associations in European American IIM patients with IIM. METHODS: Molecular genetic analyses of HLA-A, B, Cw, DRB1, and DQA1 polymorphisms were performed in a large population of African American patients with IIM (n = 262) in whom the major clinical and autoantibody subgroups were represented. These data were compared with similar information previously obtained from European American patients with IIM (n = 571). RESULTS: In contrast to European American patients with IIM, African American patients with IIM, in particular those with polymyositis, had no strong disease associations with HLA alleles of the 8.1 ancestral haplotype; however, African Americans with dermatomyositis or with anti-Jo-1 autoantibodies shared the risk factor HLA-DRB1*0301 with European Americans. We detected novel HLA risk factors in African American patients with myositis overlap (DRB1*08) and in African American patients producing anti-signal recognition particle (DQA1*0102) and anti-Mi-2 autoantibodies (DRB1*0302). DRB1*0302 and the European American-, anti-Mi-2-associated risk factor DRB1*0701 were found to share a 4-amino-acid sequence motif, which was predicted by comparative homology analyses to have identical 3-dimensional orientations within the peptide-binding groove. CONCLUSION: These data demonstrate that North American IIM patients from different ethnic groups have both shared and distinct immunogenetic susceptibility factors, depending on the clinical phenotype. These findings, obtained from the largest cohort of North American minority patients with IIM studied to date, add additional support to the hypothesis that the myositis syndromes comprise multiple, distinct disease entities, perhaps arising from divergent pathogenic mechanisms and/or different gene-environment interactions.

Adenosine Triphosphatases↗

PubMed1994

Fine-needle aspiration cytology of myositis ossificans: a case report.

This report describes the cytological findings of a case of myositis ossificans in a 16-yr-old girl. Clinically and radiologically she was suspected to have a malignant soft tissue tumor. This was not confirmed by fine-needle aspiration cytology and a diagnosis of benign lesion, probably myositis, was given. This diagnosis was confirmed by histological examination.

Adolescent↗

PubMedDate not supplied

Squamous cell carcinoma masquerading as focal myositis of the tongue.

The diagnosis of squamous cell carcinoma of the head and neck is not usually a problem for the otolaryngologist/head and neck surgeon. However, we describe a patient whose ultimate diagnosis of squamous cell carcinoma required 16 months and eluded several clinicians, despite an aggressive diagnostic regimen. The diagnostic difficulty was due to the fact that the small, centrally located tumor was surrounded by an intense inflammatory reaction that histologically mimicked a recently described rare entity of the head and neck, focal myositis. Our purpose is to make the otolaryngologist/head and neck surgeon aware of this previously unreported presentation of squamous cell carcinoma of the tongue; to review the pertinent findings associated with focal myositis; and to reinforce the need to maintain a constant vigil for the diagnosis of cancer, in spite of negative pathologic confirmation, when dictated by the clinical situation.

Biopsy↗

PubMed2002

Camptocormia in Parkinson's disease mimicked by focal myositis of the paraspinal muscles.

We report on a 63-year-old man with idiopathic Parkinson's disease who developed kyphosis and a severe forward flexion of the thoracolumbar spine. A typical feature was an increase during walking or standing and it completely disappeared in the supine position, mimicking the clinical phenomenon of camptocormia (bent spine). In addition to the abnormal posture, a weakness of the erector spinal muscles, local pain, reddening, and elevated temperature of the paraspinal muscles were evident. Creatine kinase was initially elevated, electromyography showed spontaneous activity and a myopathic pattern. Magnetic resonance imaging and bioptic examinations revealed a focal myositis of the paraspinal muscles. This case indicates that camptocormia can be mimicked by focal myositis of paraspinal muscles and must be included in the differential diagnosis, especially when additional symptoms as inflammatory signs or weakness are present.

Anti-Inflammatory Agents↗

PubMed2007

Granulomatous myositis: a clinical study of thirteen cases.

Granulomatous myositis (GM) is a rare condition that has generally been described in association with sarcoidosis. In the absence of sarcoidosis or other underlying disease, a diagnosis of isolated GM is considered. Only one study has focused on the clinical difference between isolated GM and sarcoid myopathy (SM). We report 13 cases of symptomatic GM; 8 had sarcoidosis. All patients with sarcoidosis had predominantly proximal, symmetrical lower-limb weakness, and 3 subsequently developed upper-limb or distal involvement. Three of the five patients with isolated GM had predominantly distal muscle involvement, and two had dysphagia. Corticosteroid treatment was followed by prolonged improvement in only one patient with sarcoidosis. One patient had acute sarcoid myositis and benefited from methotrexate; other immunosuppressants and etanercept proved ineffective in chronic sarcoid myopathy. Three of the five patients with isolated GM responded to corticosteroid treatment. When last examined, three patients with sarcoidosis had severe disability, whereas patients with isolated GM showed milder weakness. Thus, SM was frequently associated with severe disability and rarely improved after corticosteroid treatment, whereas most patients with isolated GM improved.

Aged↗