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PubMed1999

Recurrent orbital myositis: report of a familial incidence.

BACKGROUND: In orbital myositis, painful diplopia develops owing to an enlargement of the extraocular muscles. Diagnosis is established based on history, clinical manifestations, and therapeutic response to steroids, with the findings of magnetic resonance imaging providing additional information. OBSERVATION: We observed a family in which 4 members had an ophthalmopathy suggestive of orbital myositis. The affected members are a sibling pair (female and male) and 2 children of the brothers of their father's father. CONCLUSION: The familial incidence suggests a potential genetic predisposition in the development of orbital myositis.

Adolescent↗
PubMed2001

[Ocular myositis. A rare differential diagnosis of sinus-induced orbital complications].

Orbital myositis is an uncommon subgroup of the nonspecific orbital inflammatory syndromes (previously termed orbital pseudotumor) and presents with eyelid swelling and redness, conjunctival chemosis, pain, proptosis, and diplopia. The disease is to date of unknown origin; autoimmune processes are suspected for the etiology. In the case of an otherwise healthy young male patient (age 28 years), the coexistence of chronic sinusitis primarily led to the diagnosis of sinugen orbital cellulitis. Despite antibiotic drug administration and surgical drainage of the paranasal sinuses the symptoms persisted. A second computed tomography revealed fusiform, inflammatory enlargement of the m. rectus lateralis. This muscle showed a restrictive paresis so that initially the m. rectus medialis was suspected to be paretic. The patient responded dramatically to administration of prednisolone within 2 days. The differential diagnosis between a sinugen orbital complication and orbital myositis is significant because corticosteroids are contraindicated for orbital cellulitis whereas they remain the therapy of choice for orbital myositis.

Adult↗
PubMed2001

Alveolitis may persist during treatment that sufficiently controls muscle inflammation in myositis.

Eight patients with dermato- and polymyositis (DM/PM) and two further cases with sclerodermamyositis overlap syndrome were investigated. These patients showed signs of lung manifestation by noninvasive methods. Bronchoalveolar lavage (BAL) was performed to detect alveolitis. Four of the eight DM/PM patients showed elevated neutrophil counts. All of these and one additional case had increased lymphocyte counts. Five of the DM/PM patients showed higher total cell numbers than five healthy controls. One of the cases with scleroderma-myositis overlap syndrome also developed lymphocyte alveolitis. We conclude that signs of alveolitis are often present in patients with myositis, even though the myositis was adequately controlled by corticosteroid therapy and, in four cases, with corticosteroid plus azathioprine. The need for further follow-up studies to determine the effectiveness of intensified corticosteroid/cytostatic treatment in these patients is emphasised.

Adult↗
PubMedDate not supplied

Myositis in Kawasaki disease.

Myositis is recognized as one of several neurologic complications encountered in Kawasaki disease. We report an unusual patient with Kawasaki disease which was complicated by severe myositis and respiratory failure secondary to weakness. Comparison of our patient with previously reported cases leads us to conclude that myositis in Kawasaki disease usually is present within 1-3 weeks of illness; the symptoms usually are mild and probably remain unnoticed by most physicians, but weakness can be severe and respiratory failure may occur. The degree of creatine kinase elevation may be useful in predicting the severity of myopathy and in alerting the physician to the need for close respiratory monitoring. Therapy is supportive and complete recovery is to be expected.

Child↗
PubMed2006

A systematic review of adult-onset clinically amyopathic dermatomyositis (dermatomyositis siné myositis): a missing link within the spectrum of the idiopathic inflammatory myopathies.

OBJECTIVE: Classical dermatomyositis (CDM) patients display the hallmark cutaneous manifestations of dermatomyositis (DM), proximal muscle weakness, and laboratory evidence of myositis. The epidemiology and management of both adult-onset and juvenile-onset CDM has been well characterized. However, the clinical significance of the hallmark inflammatory cutaneous manifestations of DM occurring in individuals who have no clinically significant muscle weakness and normal muscle enzymes for prolonged periods of time (ie, 6 months or longer) has not been clear. The term amyopathic DM (ADM) (synonymous with DM siné myositis) has been proposed to draw attention to such individuals. A related form of DM, "hypomyopathic DM" [HDM], is the presence of DM skin disease for 6 months or longer in individuals who have no muscle weakness but who are found to have some evidence of muscle inflammation upon testing (muscle enzyme levels, electromyogram, muscle biopsy, muscle magnetic resonance imaging [MRI]). Clinically amyopathic DM (CADM) is a designation that has been proposed for patients having either ADM or HDM. The clinically amyopathic component of this designation was coined to emphasize the fact that the only clinical problem being experienced by these patients at the time of diagnosis is their DM skin disease. Our personal experience suggests that the CADM subphenotype might be more prevalent in adults than has been thought previously. To test this hypothesis and address questions relating to the optimal management and prognosis of such patients, we have systematically reviewed the published literature in this area. METHODS: We carried out a systematic review of the published literature on adult-onset CADM as defined in Table 1 through May 1, 2004. RESULTS: We identified 291 adult-onset CADM cases (18 years or older) reported from over 19 countries. The average duration of DM skin disease was 3.74 years (range, 6 months [by definition] to > 20 years), and 73% were female. Among 37 patients with HDM who were identified, the average duration of disease was 5.4 years, and none had developed clinically significant weakness at the time of the reports. Thirty-seven of the reported CADM patients developed muscle weakness greater than 6 months after onset of their skin disease (15 months to 6 years). For the sake of this discussion, such patients have been analyzed under the designation of "CADM --> CDM." Somewhat surprisingly, 36/291 (13%) of the identified published CADM patients developed interstitial lung disease. Incidental to our review, we also identified 10 published cases of individuals having DM skin disease and interstitial lung disease without muscle weakness, 7 of whom died from interstitial lung disease less than 6 months after onset of their DM skin disease (the term pre-myopathic DM coined by others has been used here to refer to such patients). In addition, an associated internal malignancy was found in 41/291 (14%) of the identified CADM cases. A positive antinuclear antibody was reported in 63% and myositis-specific autoantibodies (eg, Jo-1, Mi-2) in only 3.5% of the reported CADM patients in which such data were available. CONCLUSIONS: The results of this analysis suggests that the CADM subphenotype is more common than has been thought previously and that such patients may comprise a relatively high proportion of DM patients followed by dermatologists. Some CADM patients also have been observed to develop overt proximal muscle weakness years after onset of their DM skin disease. In addition, CADM patients appear to be at risk of developing the same potentially fatal disease associations/complications for which CDM patients are at risk (eg, interstitial lung disease and internal malignancy). Population-based studies of the epidemiology and optimal management of CADM patients, including efforts to identify risk factors associated with potentially fatal outcomes such as late-onset muscle weakness, interstitial lung disease, and malignancy, are needed. As an incidental finding to this literature review, we also identified a small number of reported cases of often-fatal interstitial lung disease occurring shortly after the onset of DM skin disease (< 6 months) in the complete absence of muscle weakness. This subphenotype, referred to as "pre-myopathic DM," is one with which dermatologists should be aware as early diagnosis and aggressive management can be lifesaving.

Adult↗
PubMed2004

Orbital myositis associated with Borrelia burgdorferi (Lyme disease) infection.

OBJECTIVE: To report on the clinical findings in a patient with isolated left inferior rectus myositis associated with serologically confirmed Borrelia burgdorferi infection. DESIGN: Interventional case report. TESTING: Comprehensive clinical, laboratory, and imaging evaluation. RESULTS: Contrast-enhanced computed tomography showed a swollen inferior rectus muscle with infraorbital soft tissue swelling in a patient with diplopia and prior symptoms consistent with manifestations of Lyme disease. Positive serum and cerebrospinal fluid antibodies to B. burgdorferi by enzyme-linked immunoassay were confirmed by Western blot, and the cerebrospinal fluid/serum antibody ratio was elevated. No alternative cause for orbital myositis was found, and treatment with antibiotics resulted in a complete recovery. CONCLUSIONS: Orbital myositis should be added to the expanding list of ophthalmic manifestations of Lyme disease. Correct diagnosis and appropriate antibiotic therapy may reduce the likelihood of further neurologic or ophthalmologic sequelae.

Adult↗
PubMed2003

An unusual presentation of herpes zoster ophthalmicus: orbital myositis preceding vesicular eruption.

PURPOSE: To present a case of orbital myositis associated with herpes zoster ophthalmicus. DESIGN: Observational case report. METHODS: A 47-year-old woman with acute retrobulbar eye pain and diplopia preceding the vesicular rash of herpes zoster ophthalmicus was evaluated and treated. RESULTS: Magnetic resonance imaging showed enlargement and enhancement of extraocular muscles consistent with an inflammatory myopathy. Following acyclovir and prednisone treatment, all symptoms resolved, and neuralgia did not develop. CONCLUSIONS: Herpes zoster may cause symptoms and signs of orbital myositis before eruption of cutaneous skin lesions and thus should be considered in the differential diagnosis of an acute orbital myositis.

Acyclovir↗
PubMed2006

[Bilateral idiopathic orbital myositis].

INTRODUCTION: Orbital myositis is an idiopathic inflammation of the extraoccular muscles. It is often included under the broad description of orbital pseudotumor. It is commonly a unilateral affection. CASE REPORT: We report one case of bilateral orbital myositis in a 45-year-old woman. The first symptoms were diplopia and acute bilateral orbital pain exacerbated by eye movements. Physical examination especially neurological was normal. Serum creatine kinase and biological thyroid function were normal. CT scan found a bilateral enlargement of rectus oculi medialis and lateralis. Clinical improvement was obtained a few days after oral corticosteroid treatment with remission of all signs. CONCLUSION: Orbital myositis is a rare entity. 90-95p.cent of cases are unilateral. Bilateral forms are exceptional and in this case require search for specific etiologies particularly thyroid ophthalmopathy. There is controversy concerning treatment options but corticosteroids are still the most common first choice therapy with good outcome.

Adult↗
PubMed1998

Presumed isolated inflammation of the superior oblique muscle in idiopathic orbital myositis.

OBJECTIVE: To describe isolated involvement of the superior oblique muscle in association with idiopathic orbital myositis. DESIGN: A case report with review of the literature. PARTICIPANT: A 57-year-old man with acquired strabismus participated. INTERVENTION: Oral steroid therapy was administered. MAIN OUTCOME MEASURES: Clinical and radiographic features before and after treatment were measured. RESULTS: Computed tomographic scanning showed isolated enlargement of the superior oblique muscle consistent with orbital myositis. Clinical and radiographic abnormalities quickly improved after oral steroid therapy. CONCLUSIONS: Orbital pseudotumor may present as a myositis isolated to the superior oblique muscle. This extremely rare condition has been reported in only three patients, each of whom lacked early diagnosis or treatment. Early recognition may improve chances for a successful clinical outcome.

Humans↗
PubMed2001

Experimental allergic myositis in SJL/J mouse. Reappraisal of immune reaction based on changes after single immunization.

SJL/J mice have been subjected to immunization with wide varieties of antigens to produce models of autoimmune disorders including experimental myositis. They also have a defect in dysferlin gene and spontaneously develop muscle fiber degeneration, a condition akin to limb-girdle type muscular dystrophy and Miyoshi myopathy. To know whether muscle inflammation of SJL mice after immunization with muscle fractions really represents immune-mediated myositis or no more than an epiphenomenon of muscle degeneration due to dysferlin defect, we studied immunological parameters after immunization with rabbit myosin B fraction. Initial infiltration of macrophages and CD4+ lymphocytes on day 11 was followed by increase in number of CD8+ cells. Such increase was not observed in the nontreated and adjuvant controls. Some infiltrating cells were interferon gamma (IFN-gamma) positive. Furthermore, increased expression of the signal transducers and activator of transcription 1 (STAT-1) and interferon regulatory factor 1 (IRF-1) mRNA was shown in the first 2 weeks. These results indicate Th1 system activity in the muscle, rather than simple dysferlin deficiency, particularly 1-3 weeks after immunization. Thus it is concluded that an immune-mediated myositis is taking place at this stage. This model can be helpful in understanding pathomechanisms involved in the early stage of human myositides. It has also important implications concerning immune reactions associated with transplantation or gene therapy for muscular dystrophies.

Animals↗
PubMed2002

Clinical features and outcomes of juvenile dermatomyositis and other childhood onset myositis syndromes.

The childhood myositis syndromes, primarily JDM, are relatively homogeneous diseases that have a good outcome in most cases, but are chronic, with poor outcomes, in a substantial number of cases. Because of the rarity of childhood myositis, and because there has not yet been a unified evidence-based approach to therapy, optimal treatment remains unknown. In clinic-based series the disease seems to be homogeneous but there are patients with rare clinical features, (e.g., cardiac, pulmonary, and neurologic disease), that impact prognosis. Racial, genetic, and other factors may lead to geographic variations in clinical presentations and outcomes, and perhaps further exploration of these influences will lead to a better understanding of the clinical features and outcomes seen in children. Our treatments are based on accumulated experience, but proper clinical trials have not been done. Ongoing registry studies, development of validated activity and damage assessment tools, large clinical trials, and continued investigation into the pathogenesis of the childhood myositis syndromes should lead to improved understanding and better treatments.

Algorithms↗
PubMed1994

Dermatologic aspects of myositis.

Dermatologists have long recognized that patients occasionally exhibit the pathognomic skin changes of dermatomyositis without clinical evidence of myositis for much longer than is typical for the classic form of this disease. The term dermatomyositis siné myositis has been used to refer to this condition in the past, although there is virtually no written record of this entity in the English-language medical literature. During the past 5 years, the disease course in such patients has been formally described in several case reports and small retrospective series, often under the designation amyopathic dermatomyositis. In this review, we discuss clinical issues relating to this poorly understood illness, including the possibilities of the existence of subclinical myositis, risk of occult malignancy, and risk of systemic disease manifestations normally associated with the classic form of dermatomyositis, such as interstitial lung disease. In addition, we address other issues of recent interest pertaining to the cutaneous manifestations of the inflammatory myopathies, such as unusual types of skin lesions, exacerbation of disease activity by ultraviolet light exposure, relative risk of intradermal bovine collagen injections, and management of corticosteroid-resistant skin lesions.

Dermatomyositis↗
PubMedDate not supplied

Systemic muscular inflammation in a patient with recurrent orbital myositis.

We report a 53-year-old Japanese woman who had recurrent orbital myositis for 14 years. She exhibited mild muscle weakness in proximal limbs 13 years after the onset of orbital myositis. An electromyogram revealed myopathic potentials and denervation potentials in proximal limb muscles. Quadriceps biopsy showed infiltrates of mononuclear cells around intramuscular vessels and mild degenerative changes in muscle cells. These findings indicate that the present case belongs to the spectrum of localized nodular myositis.

Adult↗
PubMed2006

[Two cases of orbital myositis with 67Ga accumulation in the orbits].

We reported two patients of orbital myositis with abnormal accumulation of 6 Ga in the bilateral orbits. The first patient, a 34-year-old man, developed bilateral lid swelling and external ophthalmoplegia. T1-weighted MRI of the bilateral orbits showed enlargement of the bilateral extraocular muscles. 67Ga scintigraphy also showed abnormal accumulation in the orbits. The second patient, a 64-year-old woman, developed diplopia caused by restricted abduction of the bilateral eyes. 67Ga scintigraphy showed abnormal accumulation in the bilateral orbits, in spite of no finding in the MRI of the extraocular muscles. Both cases were treated with 1 g of methylprednisolone per day for 3 days and subsequently 1 mg/kg per day of oral prednisolone, which resulted in improvement of opthalmoplegia and disappearance of the abnormal accumulation of 67Ga in the orbits. There is no report about orbital myositis with abnormal accumulation of 67Ga in the orbit. Our cases suggest that 67Ga shows the activity of orbital myositis and can be useful for diagnosis, especially in case of no abnormality in MRI study.

Adult↗
PubMed1994

Multifocal myositis associated with Sarcocystis sp in a horse.

Multifocal myositis was diagnosed in a 7-year-old Quarter Horse gelding on the basis of history and findings on physical examination, serum biochemical analysis, electromyography, and microscopic examination of frozen sections of muscle biopsy specimens. Histologic examination of the muscle specimen revealed multifocal accumulations of histiocytes, lymphocytes, and plasma cells, with attendant myofiber degeneration and necrosis. Parasitic cysts with morphologic characteristics of Sarcocystis sp were found in regions of myocyte degeneration and necrosis, and in regions of normal muscle. Based on a tentative diagnosis of Sarcocystis sp-induced myositis, the horse was treated with trimethoprim/sulfamethoxazole and pyrimethamine for 28 days, phenylbutazone for 5 days, and paddock rest for 30 days. At the end of treatment, the horse had gained 35 kg, its appetite had returned to normal, and muscle mass was returning to normal. Sarcocystis fayeri is the only Sarcocystis sp reported in equine muscle in the United States and is rarely associated with acute myositis or muscle atrophy. The development of clinical signs in this horse could have been the result of an underlying immunosuppression or infection with a particularly pathogenic strain or large infective dose of S fayeri.

Animals↗
PubMed2001

The benefits and limitations of a physical training program in patients with inflammatory myositis.

The clinical features of inflammatory myositis are determined by the severity and extent of muscle weakness and systemic manifestations. The benefits and limitations of physical training programs and rehabilitation strategies depend on the clinical phase of the disease and analysis of underlying impairments responsible for functional limitations in the patient. Patients with early stage disease and severe weakness will be treated differently than patients who have responded to medication and are improving. Not all patients will respond to medications; their therapy programs will have different requirements. This article reviews available data on the physiologic responses to exercise in patients with inflammatory muscle diseases. New data support more aggressive approaches to progressive strengthening exercises for patients with inflammatory myositis.

Activities of Daily Living↗
PubMed2000

Partial laminin alpha2 chain deficiency in a patient with myopathy resembling inclusion body myositis.

It is becoming evident that clinical phenotypes associated with partial laminin alpha2 chain deficiency are variable. We recently observed a 29-year-old man with leukoencephalopathy and vacuolar myopathy resembling inclusion body myositis. Laminin alpha2 immunohistochemical analysis showed reduction of the protein on muscle fiber surfaces. Molecular analysis revealed two novel compound heterozygous mutations in the LAMA2 gene. This is the first report linking a mutation in the LaMA2 gene with leukoencephalopathy and inclusion body-like myositis.

Adult↗
PubMed2005

[The practice guideline 'Dermatomyositis, polymyositis and sporadic inclusion body myositis'].

This guideline presents recommendations for the diagnosis and treatment of dermatomyositis, polymyositis and sporadic inclusion body myositis (sIBM) according to the best available evidence. Characteristic skin abnormalities can be sufficient for the diagnosis of dermatomyositis. In case of doubt, a skin biopsy is advisable. A muscle biopsy is indicated when other examinations are inconclusive and the musculature is involved. The working group considers screening for cancer to be required in adults with dermatomyositis and presents recommendations for the way that this should be done. At least one-third of all patients with polymyositis has, or will develop, an associated inflammatory connective tissue disease. If a patient with a connective tissue disease develops symmetrical, proximal muscle weakness in the course of weeks or months, this may be assumed to be due to polymyositis. In the absence ofpre-existing connective tissue disease, demonstration of a mononuclear cell infiltrate in muscle tissue is a prerequisite for the diagnosis ofpolymyositis. The histopathology of muscle tissue is used as the gold standard for the diagnosis of sIBM. The practice guideline presents criteria for the concept 'activity' of myositis. Disease activity serves as a guideline for the treatment of polymyositis and dermatomyositis. The treatment of choice for dermatomyositis and polymyositis is high-dose prednisone. Physical activity does not have a negative effect on the course of these diseases. The long-term prognosis ofdermatomyositis and polymyositis is not well known. The clinical course of sIBM is slowly progressive.

Anti-Inflammatory Agents↗