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PubMed1995

New advances in the understanding of sporadic inclusion-body myositis and hereditary inclusion-body myopathies.

This review emphasizes new advances in seeking the pathogenic mechanisms of sporadic inclusion-body myositis and hereditary inclusion-body myopathy syndromes. Clinical and pathologic similarities and differences between sporadic and hereditary forms are described. Hypotheses are presented regarding the possible causes and consequences of abnormally accumulated intramyofiber beta-amyloid precursor protein (beta APP) (including beta-amyloid protein and C- and N-terminal epitopes), hyperphosphorylated tau, alpha 1-antichymotrypsin, apolipoprotein E, prion protein, ubiquitin, nicotinic acetylcholine receptor and its 43-kD associated protein, fibroblast growth factor, and transforming growth factor-beta. Also increased are beta APP mRNA and prion protein mRNA. Striking similarities between the pathology of muscle specimens from sporadic inclusion-body myositis and samples from the brains of patients with Alzheimer's disease in regard to Congo red positivity and accumulations of several proteins are discussed. Because most of the proteins that pathologically accumulate throughout the abnormal muscle fibers also accumulate focally at normal human neuromuscular junctions, the possible "junctionalization" of nonjunctional nuclei as a pathogenic mechanism in the muscle fiber is discussed.

Diagnosis, Differential↗
PubMed1995

T cell-mediated immune mechanisms in myositis.

Advances in molecular biologic techniques and the availability of novel immunologic reagents have allowed new approaches to understanding the pathogenesis of human autoimmune diseases, including the idiopathic inflammatory myopathies. Indirect evidence that autoreactive T cells mediate muscle inflammation in the human myositis syndromes has been strengthened by recent studies describing restricted T cell receptor gene expression in certain clinical and/or serologic groups of myositis patients. These findings are supported by other investigations documenting abnormal patterns of cytokine, adhesion molecule, and major histocompatibility complex antigen expression within inflammatory lesions. The major challenge of future studies is to identify the specific antigen(s) responsible for initiating and perpetuating these harmful immune responses.

Amino Acid Sequence↗
PubMed1997

Myositis associated with graft-versus-host disease.

A polymyositis syndrome has been described as a rare complication in patients who develop chronic graft-versus-host disease after allogeneic bone marrow transplantation. Chronic graft-versus-host disease is a cellular immune-mediated donor bone marrow versus patient rejection reaction, which may also lead to an autoimmune pathologic process. Chronic graft-versus-host disease-related polymyositis appears to be very similar to idiopathic myositis in its clinicopathologic presentation; chronic graft-versus-host disease-related myositis responds well to prednisone but cyclosporine may be an important component of second-line therapy due to its efficacy in controlling underlying chronic graft-versus-host disease.

Bone Marrow Transplantation↗
PubMed2001

Myositis in children.

Idiopathic inflammatory myositis in children includes multiple disease entities, but is primarily made up of juvenile dermatomyositis and, to a lesser degree, juvenile polymyositis. Much new information has been published in the last few years about these diseases, including the epidemiology, pathogenesis, clinical diagnosis, and outcomes and treatment. This includes information on onset of symptoms, potential inciting agents, and regional differences. Exciting data have emerged in our understanding of the immune response gene associations and the description of chimerism in children with these disorders. Finally, new advances in clinical evaluations and outcomes have been described as well as new treatment protocols to provide a more effective therapy with less toxicity. Continued investigation is needed to further understand these diseases, but great strides are being made in our understanding and ability to care for children with idiopathic inflammatory myositis.

Adolescent↗
PubMed2001

Assessment of inflammatory myositis.

The assessment of disease activity and damage in patients with idiopathic inflammatory myositis remains inadequate. In this review we describe current approaches to the assessment of disease activity and damage. These include serologic, biochemical, radiographic, and histologic approaches. None of these methods are individually effective in differentiating disease activity from damage. Invariably combinations of these methods are needed to comprehensively assess the extent of both muscular and extramuscular disease. Recent developments (in serology, imaging, and genetics) are mentioned. These may prove to be useful in assessing the full extent of idiopathic inflammatory myositis; however, they are not widely available at present and are confined to research studies

Blood↗
PubMed1993

Myositis ossificans masquerading as a recurrent spindle cell lipoma of the orbit.

A spindle cell lipoma developed in the right orbit of a 43-year-old woman whose medical history was only significant for chronic asthma. Sixteen months after nearly complete excision of the tumor, the patient presented with eyelid swelling, orbital pain, and a calcified apical mass as demonstrated by computerized tomography (CT). Recurrent tumor or a second neoplasm was suspected. Histologic examination revealed myositis ossificans. To our knowledge, this is the first reported case of myositis ossificans in the orbit.

Adult↗
PubMed1987

Gallium uptake in myositis ossificans. Potential pitfalls in diagnosis.

Seven cases of gallium uptake in myositis ossificans are described. Gallium scans are done frequently in paraplegics, quadriplegics, and comatose patients to look for occult infection. It is important to be aware of possible gallium uptake in myositis ossificans, particularly in the extremities, which is frequent in these patients. Gallium uptake may be present prior to any abnormalities seen on plain films or CT scans. It is important to correlate roentgenograms with abnormal gallium scans, particularly in the extremities, to avoid potential pitfalls in diagnosis and prevent unnecessary antibiotic treatment. A bone scan should be obtained whenever possible, particularly when roentgenograms are negative, to confirm the diagnosis.

Adult↗
PubMed1995

Thallium-201 uptake in myositis ossificans. Potential pitfall in diagnosis.

Myositis ossificans is primarily a disorder of adolescents or adults, whereby an area of muscle mass undergoes progressive ossification. Its radiographic and scintigraphic appearances have been well documented. In this paper, a case of histopathologically demonstrated myositis ossificans in the proximal thigh with unexpected TI-201 accumulation was presented.

Adult↗
PubMed2000

Becker muscular dystrophy associated with focal myositis on bone scintigraphy.

METHODS: Bone scintigraphy was performed on a 6-year-old boy with possible sacroiliitis. RESULTS: Extraosseous uptake in the region of the paraspinal, psoas and calf muscles was noted and suggested myositis. Subsequently, his creatine kinase level was found to be elevated. Results of a neuromuscular examination, muscle biopsy with dystrophin immunostaining, and a multiplex polymerase chain reaction led to the diagnosis of Becker muscular dystrophy. CONCLUSIONS: A patient with Becker muscular dystrophy had several distinct areas of myositis localized by bone scintigraphy. Pathologic features including focal myonecrosis and regeneration may contribute to this scintigraphic picture.

Bone and Bones↗
PubMed1993

The treatment of inclusion body myositis: a retrospective review and a randomized, prospective trial of immunosuppressive therapy.

We have sought to examine the response to immunosuppressive therapeutic intervention in inclusion body myositis (IBM) in a retrospective review of prior responses to therapy and in an open, randomized crossover trial. We collected information on the response to prior therapy on 25 patients, and for prospective therapy on 11 of these patients. All met criteria for a definite idiopathic inflammatory myopathy and had biopsy-proven IBM. Clinical and laboratory results were assessed by interviews of patients and by chart review in the retrospective trial. Manual muscle strength was assessed by a single trained observer; the patients' activities of daily living were assessed by questionnaire; and serum tests of muscle-associated enzymes were measured in the prospective trial. In the retrospective review, prednisone appeared to have been of some, albeit modest, clinical benefit in 10 of 25 (40%) patients. Other therapies, primarily azathioprine and methotrexate, also appeared to have halted the progression of weakness in 8 of 35 trials (23%). In the prospective study, combination therapy of oral azathioprine and methotrexate and a biweekly infusion of high-dose intravenous methotrexate with leucovorin rescue were given for 3 to 6 months in an open, crossover design. Both the oral and the intravenous regimens were clinically effective in some patients. There was clinical improvement in 3 trials, stabilization in 11 trials, and worsening in 5 trials, out of a total of 19 completed (22 intended) trials. The presence of active inflammation at entry into the prospective therapeutic protocol, either directly observed on muscle biopsy or indirectly indicated by serum creatine kinase level, may have been associated with clinical improvement. A complete laboratory response with normalization of creatine kinase and other muscle-associated enzymes did not, however, significantly predict clinical responsiveness in the prospective trial. In this first report, to our knowledge, of a prospective trial of immunosuppressive therapy for this disease, stabilization and even slight improvement of strength and functional abilities appeared to be achieved in some patients. We believe that prednisone and other immunosuppressive therapies were of modest benefit in about half of patients with inclusion body myositis, especially those with some evidence of active inflammation. Stabilization of an otherwise inexorably deteriorating course appears, therefore, to be an attainable goal in some patients with IBM.

Adult↗
PubMed1989

Spontaneous streptococcal gangrenous myositis: survival with early debridement.

Spontaneous gangrenous myositis caused by Streptococcus pyogenes is usually fatal, but no longer uniformly so. There appears to be a spectrum of disease due to beta-hemolytic streptococci, from necrotizing fasciitis to pyomyositis to spontaneous gangrenous myositis. Survival is possible with early surgical debridement, reexploration at 24 to 36 hours, and intensive supportive care.

Aged↗
PubMed2002

Adductor myositis as a cause of childhood hip pain.

Two cases of bacterial adductor myositis that presented as painful hips in boys aged 4 and 9 years are reported. Clinically, there was severe pain and a high pyrexia, a raised C-reactive protein and positive blood cultures but a negative hip ultrasound. Urgent magnetic resonance imaging demonstrated changes throughout the adductor muscles in keeping with bacterial myositis. Both boys settled with intravenous antibiotic therapy. We propose that magnetic resonance imaging is a valuable tool in the assessment of infection around the hip and should be indicated when other investigations have excluded a septic arthritis but the child remains unwell.

Anti-Bacterial Agents↗
PubMed1996

The immunopathologic and inflammatory differences between dermatomyositis, polymyositis and sporadic inclusion body myositis.

In polymyositis and sporadic inclusion body myositis, clonal expansion of CD8+ cells which are primed to recognize previously unknown muscle antigens occurs. Compared with sporadic inclusion body myositis, however, in which the T-cell response may not be antigen driven, there is in polymyositis an overexpression of certain T-cell receptor gene families among the autoinvasive T-cells. Although studies on the endomysial expression of cytokines and cell adhesion molecules have provided additional support for the concept of an ongoing immune process, the site of sensitization and the mechanism by which the autoimmune process is triggered remains to be established. In dermatomyositis, a multiorgan disease, evidence exists that the complement-mediated microvascular injury by the putative antibody may not be limited to the endomysial vessels but may also involve the blood vessels in the dermis. The antigenic target on the endothelial cell in dermatomyositis patients and the pathogenic role of the recently studied anti-Mi-2 antibody directed against a helicase are still to be determined.

Dermatomyositis↗
PubMedDate not supplied

Proliferative myositis: value of imaging.

Imaging findings in a 52-year-old woman with proven proliferative myositis are described. US revealed preservation of continuous muscle bundles and patchy areas of hyperechogenicity, containing hypoechoic lines. MRI showed at T2-w sequences an ill-defined, hyperintense, intramuscular lesion, containing isointense lines. Subtotal enhancement, a nonenhancing geometrical web, and fascial enhancement were noted. In patients with painful growing masses, US and MRI correlation may suggest the diagnosis of proliferative myositis leading to biopsy, thus avoiding mutilating surgery.

Contrast Media↗
PubMed1998

Traumatic myositis ossificans of the quadriceps in infants.

Acute traumatic myositis ossifications is uncommon and usually occurs in adolescents and young adults after a significant direct blow to the affected muscle. It is extremely rare in infants, and we have been able to find only two other cases in the English literature. We present two cases of traumatic myositis ossificans in infants who, except for the use of diagnostic ultrasonography, would have been misdiagnosed as having osteomyelitis (Patient 1) and a possible malignancy (Patient 2).

Acute Disease↗
PubMed1983

Battered child syndrome simulating myositis.

The battered child syndrome is not an unusual problem to be encountered in a clinical orthopedic practice. The manifestations in abused children may be varied and many, necessitating that orthopedists be familiar with the ways in which affected children present to reduce the unfortunate consequences that occur when abuse is not initially recognized. Our purpose in reporting the case of an abused child is to attract the attention of the orthopedic surgeon, who may be the initial physician to see a battered child, to an unusual presentation of this syndrome which simulated myositis. The association of myositis with battered child syndrome has not been previously encountered in the orthopedic literature.

Battered Child Syndrome↗
PubMed1984

Myositis ossificans of the upper extremity: a long-term follow-up.

The long-term results of nonoperative treatment of myositis ossificans traumatica have been infrequently reported. We reviewed 83 cases of myositis ossificans seen at the Mayo Clinic from 1950 to 1979 in patients up to 21 years of age. The upper extremity was involved in 31 patients. Follow-up averaged 13 years for 23 of the (74%) patients studied. Football injuries had occurred in 20 of these patients, and the diagnosis was made an average of 3.3 weeks from the time of the initial injury. Observation was the only treatment in 18 of the 31 cases. At follow-up two-thirds of the patients with upper extremity involvement had no problems associated with the disorder, and one-third described some difficulties. Nonoperative treatment remains an accepted management approach for this problem.

Adolescent↗