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PubMed2001

Inclusion body myositis mimicking motor neuron disease.

OBJECTIVE: To describe the clinical and electrophysiologic features of patients with inclusion body myositis that was misinterpreted as motor neuron disease. PATIENTS AND METHODS: We retrospectively retrieved the medical records of 70 patients with a pathologic diagnosis of inclusion body myositis. From this group, we selected those who had been first diagnosed as having motor neuron disease or amyotrophic lateral sclerosis. We reviewed the clinical, electrophysiologic, laboratory, and morphologic studies. RESULTS: Nine (13%) of 70 patients with inclusion body myositis had been diagnosed as having motor neuron disease. Six of the 9 patients had asymmetric weakness; in 4 the distal arm muscles were affected. Eight patients had finger flexor weakness. Tendon reflexes were preserved in weak limbs in 6, hyperactive in 2, and absent in 1. Four patients had dysphagia. Fasciculation was seen in 2 patients. None had definite upper motor neuron signs or muscle cramps. Routine electromyographic studies showed fibrillation potentials and positive sharp waves in all 9. Fasciculation potentials were seen in 7 and long-duration polyphasic motor unit potentials were seen in 8. There was no evidence of a myogenic disorder in these 9 patients. Muscle biopsy was done because of slow progression or prominent weakness of the finger flexors and was diagnostic of inclusion body myositis. A quantitative electromyogram was myopathic in 4 of the 5 patients studied. CONCLUSIONS: Inclusion body myositis may mimic motor neuron disease. Muscle biopsy and quantitative electromyographic analysis are indicated in patients with atypical motor neuron disease, especially those with slow progression or early and disproportionate weakness of the finger flexors.

Aged↗
PubMed1992

Management of dysphagia in inclusion body myositis.

Inclusion body myositis is an inflammatory myopathy in which dysphagia has been considered a rare finding. However, recent literature finds dysphagia an increasingly common symptom as more cases of inclusion body myositis are identified. Unlike some inflammatory myopathic disorders, inclusion body myositis is resistant to treatment with corticosteroids, and therefore, the otolaryngologist may be consulted regarding surgical options for relief of dysphagia. A patient is described in whom severe progressive dysphagia associated with inclusion body myositis developed. Impaired pharyngeal wall motion and cricopharyngeal achalasia were demonstrated by videofluoroscopic evaluation, and the patient was successfully treated by cricopharyngeal myotomy. The pathophysiologic nature of inclusion body myositis and the mechanisms of cervical dysphagia in the inflammatory myopathies are reviewed.

Aged↗
PubMed2006

Lyme myositis.

Myositis has been reported as a rare manifestation of Lyme disease, and Lyme myositis can be an important consideration in the differential diagnosis of unusual cases, especially in patients who live in or travel to endemic areas. We report the case of a 43-year-old man who presented with focal myositis of the proximal lower extremity and was subsequently diagnosed as having Lyme myositis. The patient had previously experienced a febrile illness and rash, but had no ongoing symptoms of Lyme disease. Myositis was confirmed by magnetic resonance imaging and muscle biopsy; Borrelia burgdorferi infection was confirmed by Lyme serology and polymerase chain reaction testing of synovial fluid and biopsy material. The current case is reviewed in the context of findings from previous case descriptions.

Adult↗
PubMed1992

Magnetic resonance imaging of myositis ossificans: analysis of seven cases.

Myositis ossificans typically presents as soft tissue swelling with progressive ossification on radiographs. Since magnetic resonance imaging (MRI) is commonly used to evaluate soft tissue masses, we analyzed eight MR examinations in seven patients with myositis ossificans to determine if typical patterns were present. One acute lesion had homogeneous intermediate signal intensity on T1-weighted images and high signal intensity on T2-weighted images. Two subacute lesions had low signal intensity margins with slightly increased signal intensity centers on T1-weighted images and very high signal intensity on T2-weighted images. Five chronic lesions had two different patterns. All five were well-defined with low signal intensity borders. Three had signal intensity patterns characteristic of fat on T1-weighted and T2-weighted images. The other two lesions had intermediate signal intensity on T1-weighted images and slightly increased signal intensity on T2-weighted images. We conclude that typical MR appearances of myositis ossificans do exist. A low signal intensity rim is a common finding. However, these patterns are not unique to myositis ossificans and resemble those that have been reported in other lesions. It is important to be aware of the spectrum of MR findings of myositis ossificans when considering the differential diagnosis of a soft tissue mass.

Acute Disease↗
PubMed1987

Additional biochemical criteria in the differential diagnosis of myositis.

Thirty-six biopsy specimens of human biceps and vastus lateralis muscles were examined by histometric analysis and determination of enzyme activities (phosphorylase, triosephosphate dehydrogenase, 3-hydroxacyl-CoA-dehydrogenase, lactate dehydrogenase, hexose isomerase, citrate synthetase, 6-phosphogluconate dehydrogenase). The series included 13 specimens from patients suffering from a benign form of muscular dystrophy (limb girdle and Becker type of muscular dystrophy) and 12 specimens from patients with an acute (n = 5) or chronic (n = 7) form of myositis. Muscle fibres were atrophic in myositis and hypertrophic (with an increased variation of fibre diameters) in muscular dystrophies, as has been shown previously. When myositis samples were compared with either normal or dystrophic muscles, a highly significant lowering of glycolytic enzyme activity was found in chronic myositis, while the activity of 6-phosphogluconate dehydrogenase was elevated to highly significant levels. Measurements of the latter enzyme's activity might be of additional value in differentiating chronic forms of myositis from benign muscular dystrophies.

Adolescent↗
PubMed2005

[PM-Scl antibody positive systemic sclerosis associated with inclusion-body myositis].

We describe a 72- year-old patient with a ten year history of anti-PM-Scl positive systemic sclerosis associated with inclusion-body myositis. While the association of dermatomyositis and polymyositis with anti-PM-Scl positive systemic sclerosis is frequently reported, inclusion-body myositis was, to the best of our knowledge, only previously described once in association with anti-PM-Scl-positive systemic sclerosis. The distinction between inclusion-body myositis and other forms of inflammatory myopathy, like the histopathologically well distinguishable polymyositis or dermatomyositis, is relevant because of the poor response of inclusion- body myositis to immunosuppressive treatment. Our case underlines that in patients with anti-PM-Scl-positive systemic sclerosis and treatment resistant progressive myopathy the diagnosis of inclusion body myositis should be considered.

Aged↗
PubMed2006

Myositis specific autoantibodies.

Recent studies have continued to examine the clinical associations of the group of autoantibodies that occurs predominantly in patients who have myositis (antibodies to aminoacyl-tRNA synthetases, to signal recognition particle [SRP], and to the nuclear helicase Mi-2). The anti-synthetase syndrome has been further studied, and the value of tacrolimus in treatment of the associated interstitial lung disease has been noted. The low frequency of myositis specific autoantibodies in non-myositis neuromuscular disorders has been more clearly demonstrated. The clinical associations of anti-Mi-2 and anti-SRP were further studied, and patients with antibodies without myositis were reported. Evidence suggested that ultraviolet light exposure may influence the expression of dermatomyositis and anti-Mi-2. A new classification for myositis using overlap clinical features and autoantibodies was proposed. A new autoantibody, anti-caDM-140, was described, associated with clinically amyopathic dermatomyositis and interstitial lung disease. The possibility was raised that increased antigen expression in regenerating muscle may help to perpetuate the disease. These antibodies continue to be the subject of active investigation.

Amino Acyl-tRNA Synthetases↗
PubMed1994

Functional reorganization in the rat dorsal horn during an experimental myositis.

In anaesthetized rats, the influence of an acute inflammation (2-8 h duration) of the gastrocnemius-soleus (GS) muscle on the excitability of dorsal horn neurones was studied using a mapping procedure. One of the main effects of the myositis was that the neurone population responding to GS A-fibre input increased in size. The increase was most marked in the lateral segments L6-L3 which received little input from the GS muscle in control animals. Excitability testing showed a myositis-induced lowering in threshold, combined with an increase in latency, jitter and input convergence. This suggests that new oligo- or polysynaptic connections become functional under the influence of a myositis. Neuronal effects induced by C fibres in the GS nerves were not significantly altered by a myositis, but C fibre-induced activations from the peroneal and sural nerves increased in the lateral dorsal horn. The results show that an acute myositis leads to marked changes in the functional connectivity of the dorsal horn within a few hours. The main increase in excitability took place in the lateral dorsal horn, where many neurones acquired a new input from the GS muscle. This mechanism may be involved in the spread or referral of muscle pain.

Animals↗
PubMed1995

[Non-traumatic myositis ossificans circumscripta].

BACKGROUND: Myositis ossificans circumscripta is a benign lesion with an acute course and may simulate a malignant tumor. It usually follows trauma to soft tissue. CASE REPORTS: Case n. 1. A 13 year-old girl was admitted because of a painful inflammatory tumour in the left thigh. Initial X-rays were normal. Ultrasound imaging showed a heterogeneous echogenic mass with several extending shadow cones in the distal part of the vastus medialis muscle resembling a calcifying hematoma. Twenty days later, X-rays showed a vague calcified peripheral rim in the medial distal part of the thigh. White blood count was normal, and blood sedimentation rate was 46 millimeters in the first hour. CT scan showed a transparent zone between the lesion and the adjacent bone and a lucent central area, surrounded by a dense outer area consistent with myositis ossificans. Histological examination of the excised mass confirmed myositis ossificans. Two years later, the patient was asymptomatic and X-rays showed no ossification. Case n. 2. A 14 year-old girl suffered from pain in the right anterior hip area since 10 days. She denied any trauma. A firm mass was palpable in the anterior superior iliac spine area and X-rays revealed a calcific density. CT scan showed a dense bony mass in the right gluteus medius muscle clearly separated from the adjacent bony pelvis by a soft tissue plane. Histological examination of the excised mass confirmed myositis ossificans. One year later, the patient was asymptomatic and X-rays of the pelvis showed no ossification. CONCLUSION: Myositis ossificans circumscripta is rare in children. CT scan suggests the benign nature of the lesion by demonstrating integrity of bony cortex and characteristic disposition of calcifications. The biopsy is not necessary if the diagnosis is certain. Surgery permits to reduce the evolution.

Adolescent↗
PubMed1994

Coxsackie B1 virus-induced murine myositis: relationship of disease severity to virus dose and antiviral antibody response.

Inoculation of Coxsackie B1 virus (CB1) in newborn CD1 Swiss mice induces a chronic myositis of proximal hindlimb muscles (CB1 myositis). To study the possible role of the virus dose, and of antiviral antibodies in the development of CB1 myositis, we infected groups of newborn mice with six CB1 doses, ranging from 30 to 10,000 plaque forming units (pfu); after 4 and 8 weeks we determined morbidity and antiviral antibody titer, and quantified histopathological changes. At 4 weeks, morbidity and mononuclear cell infiltration differed significantly for the various groups, with the most prominent changes in 300 pfu animals. At 4 and 8 weeks diseased animals had significantly higher antibody titers than clinically unaffected animals, and at 4 weeks myopathic and infiltrative changes correlated positively with the serum antibody titer. Our data indicate that the virus dose plays a pathogenic role in CB1 myositis, and they suggest further study on the role of humoral immune mechanisms in the early phase of CB1 myositis.

Animals↗
PubMed1988

Orbital myositis involving the oblique muscles. An echographic study.

Idiopathic orbital myositis is a common nonspecific orbital inflammatory syndrome that can involve one or more of the extraocular muscles. Oblique muscle involvement is infrequently reported, possibly because such involvement is difficult to identify clinically or by computed tomography (CT). The authors reviewed seven cases of orbital myositis involving the oblique muscles solely or in association with rectus muscle involvement. Standardized echography demonstrated homogeneous low-reflective enlargement, diagnostic of myositis, of the superior and inferior oblique muscles and tendons along their courses. The finding of massive inferior chemosis was associated with involvement of the inferior oblique muscle. Although the disease typically responds to therapy with systemic steroids, residual gaze restriction may occur. The oblique muscles may be frequently involved in idiopathic orbital myositis when evaluated by standardized orbital echography. In these cases, echography provides rapid, accurate, and reliable confirmation of the diagnosis of myositis, differentiates other orbital inflammatory diseases, and provides an objective measure of the therapeutic response.

Adult↗
PubMed1997

Current status in the treatment of orbital myositis.

PURPOSE: Orbital myositis is described as exquisitely sensitive to therapy with systemic corticosteroids. However, it may turn into a recurrent or chronic disease, requiring repeated courses or maintenance of corticotherapy that often is complicated with serious side effects. The authors conducted this study to evaluate their experience and the current status in the literature as to treatment of orbital myositis with corticosteroids and corticosteroid-sparing alternatives. METHODS: The clinical records and computed tomography scans of patients with orbital myositis who presented at the Orbital Center Amsterdam between 1977 and 1991 were studied for the clinical course and therapeutic outcome. Recurrences were defined as a new attack of the same or another extraocular muscle as the primary affected, in the same or the other orbit. RESULTS: The study group comprised 16 patients. All patients responded to initial treatment with oral corticosteroids (14/16) or nonsteroidal anti-inflammatory drugs (NSAID) (2/16). Of these 16 patients, 9 (56%) had 1 or more recurrences. Recurrences responded to repeated courses of corticosteroids, NSAID, or 20 Gy irradiation. Despite radiotherapy in six patients, recurrences continued to occur within a mean period of 2.7 years (range, 0.5 month-8.8 years). The mean follow-up after initial therapy in general was 9.7 years (range, 2.6-16.9 years), and after radiotherapy, in particular, was 7.4 years (range, 4.1-10.5 years). CONCLUSIONS: Orbital myositis responds well to oral corticosteroids, but recurs in 50% of the cases. Based on a long-term follow-up of six patients who received irradiation of 20 Gy, radiotherapy appears ineffective in stopping recurrent orbital myositis.

Administration, Oral↗
PubMed1998

Relationship between neuronal activity and substance P-immunoreactivity in the rat spinal cord during acute and persistent myositis.

The spinal level of substance P (SP) is assumed to be an important determinant of neuronal activity under pathophysiological conditions. In rat dorsal horn neurones, impulse activity was studied during a carrageenan-induced acute (2-8 h) and a Freund's adjuvant-induced persistent (12 days) myositis and compared with the spinal substance P-immunoreactivity (SP-IR) of the same animals. Myositis-induced changes in responsiveness of the neurones reached a maximum within 2-8 h, whereas background activity of the neurones was highest after 12 days of myositis. The area of SP-IR in the superficial dorsal horn decreased during acute and persistent myositis and the integrated density of the staining was largely unchanged. The difference in time-course between neuronal activity and SP-IR suggest that during persistent myositis factors other than SP gain more influence on the behaviour of the neurones.

Acute Disease↗
PubMed2001

Macrophagic myofasciitis associated with inclusion body myositis: a report of three cases.

We describe three patients with macrophagic myofasciitis and inclusion body myositis. All patients fulfilled diagnostic criteria for inclusion body myositis and myopathologic criteria for macrophagic myofasciitis. In the three cases macrophagic myofasciitis complicated the evolution of a known and painless inclusion body myositis and was diagnosed in a repeated deltoid biopsy because of the appearance of myalgia during the course of inclusion body myositis in all cases. The unexpected appearance of myalgia during the course of painless inclusion body myositis must arouse the suspicion of an association of another inflammatory muscle disease, macrophagic myofasciitis.

Adult↗
PubMed2006

Clinical implications of autoantibody screening in patients with autoimmune myositis.

OBJECTIVE: To evaluate the clinical usefulness of serum autoantibody profiling in patients with autoimmune myositis. METHODS: We retrospectively studied 74 consecutive patients: 68 had definite or probable myositis according to Bohan-Peter criteria, six suffered from antisynthetase syndrome with subclinical myopathy. Myositis specific antibodies (MSA) (anti-ARS, -SRP, -Mi-2) were determined by RNA immunoprecipitation or immunoblot, myositis associated antibodies (MAA) (anti-RoRNP, -U1RNP, -PM/Scl, -Ku) by immunoblot. RESULTS: Forty-three patients (58%) were positive for MSA: anti-Jo-1 in 15/27 polymyositis (PM) (55%), 4/33 dermatomyositis (DM) (12%), 1/8 overlap (12%) and 2/6 antisynthetase syndrome (33%); anti-ARS non-Jo-1 in 1/27 PM (4%), 2/33 DM (6%) and 4/6 antisynthetase syndrome (67%); anti-Mi-2 in 1/27 PM (4%) and 11/33 DM (33%); anti-SRP in 3/27 PM (11%) and 1/33 DM (3%). One patient was anti-Jo-1/Mi-2 positive, one anti-Jo-1/SRP positive. Moreover, 27 patients (36%) were positive for MAA: anti-Ro/SSA in 8/27 PM (30%), 7/33 DM (21%), 1/8 overlap (12%), and 3/6 antisynthetase syndrome (50%); anti-U1RNP in 1/27 PM (3.7%), 1/33 DM (3%), and 2/8 overlap (25%); anti-PM/Scl in 2/8 overlap (25%), anti-Ku in 2/8 overlap (25%). Anti-Jo-1 was predominantly associated with PM, anti-Mi-2 was almost exclusively found in DM patients. Anti-ARS antibodies were closely associated with interstitial lung disease and polyarthritis; notably, anti-ARS non-Jo-1 was more frequent in patients without overt muscle alterations. Anti-Ro/SSA antibody was not associated with any disease subset, but significantly more frequent in antisynthetase syndrome. CONCLUSIONS: Searching for MSA and MAA in patients with autoimmmune myositis is recommended because of its diagnostic and clinical value. Anti-ARS non-Jo-1 antibodies seem to preferentially target patients with pulmonary fibrosis without overt myopathy.

Adolescent↗
PubMed1996

Proteasome alpha-type subunit C9 is a primary target of autoantibodies in sera of patients with myositis and systemic lupus erythematosus.

Autoantibodies occur in low frequencies among patients with myositis characterizing only distinct subsets of this disease. Most of these known antibodies are directed to enzymatically active complexes. The 20S proteasome represents an essential cytoplasmatic protein complex for intracellular nonlysosomal protein degradation, and is involved in major histocompatibility complex class I restricted antigen processing. In this study we investigated whether the 20S proteasome complex is an antibody target in myositis and in other autoimmune diseases. 34 sera of poly/dermatomyositis patients were assayed for antiproteasomal antibodies using enzyme-linked immunosorbent assay, immunoblot, and two-dimensional non-equilibrium pH gradient electrophoresis (NEPHGE). Sera was from patients with systemic lupus erythematosus (SLE), mixed connective tissue disease, and rheumatoid arthritis; healthy volunteers served as controls. In 62% (21/34) of the cases sera from patients with myositis and in 58% (30/52) of the cases sera from patients with SLE reacted with the 20S proteasome. These frequencies exceeded those of sera from patients with mixed connective tissue disease, rheumatoid arthritis, and healthy controls. The alpha-type subunit C9 of the 20S proteasome was determined to be the predominant target of the autoimmune sera in myositis and SLE. Lacking other frequent autoantibodies in myositis, the antiproteasome antibodies are the most common humoral immune response so far detected in this disease entity.

Antibodies, Monoclonal↗
PubMed2003

Tuberculous myositis: a rare but existing clinical entity.

OBJECTIVES: To investigate the clinical characteristics of patients with tuberculous myositis. METHODS: From January 1996 to March 2001, patients with positive cultures of Mycobacterium tuberculosis or histology-proven caseous granulomatous inflammation from muscular specimens were identified and their medical records were reviewed. RESULTS: Thirty-five patients were identified. Infection-related myositis was initially suspected in 20 patients (57.1%). The routes of infection were contiguous spread in 22 patients (62.8%), haematogenous spread in 10 (28.6%) and traumatic inoculation in three (8.6%). Five patients (14.3%), including the three who had received corticosteroids, died of uncontrolled sepsis. The computed tomography or the magnetic resonance imaging of the involved muscles showed findings suggestive of tuberculous myositis in 15 patients (42.9%). CONCLUSIONS: Tuberculosis should be considered as one of the possible aetiologies of myositis, especially among patients with suggestive radiographic findings or in endemic areas of tuberculosis. Patients who develop tuberculous myositis after using corticosteroids have poor prognoses.

Adolescent↗
PubMed1992

Proliferative fasciitis and myositis of childhood.

Eleven cases of proliferative fasciitis and myositis in children, ages 2.5 months to 13 years, are presented. Eight lesions averaging 2.3 cm in size occurred in the extremities, two in the head and neck region and one on the chest wall. Like proliferative fasciitis and myositis in adults, these lesions consisted of admixtures of large polygonal to spindled, ganglion-cell-like fibroblasts with vesicular nuclei and prominent inclusion-like nucleoli. Seven of 11 lesions were initially diagnosed as sarcomas, most commonly rhabdomyosarcoma. Four patients were treated by wide excision (three with regional lymphadenectomy), three received chemotherapy, and one was given radiation therapy. There were some histologic differences from adult-type proliferative fasciitis and myositis. The childhood lesions were generally well circumscribed, lobulated, extremely cellular with less collagen production, and often associated with acute inflammation and microscopic foci of necrosis. Immunohistochemical comparison with adult proliferative fasciitis and myositis showed similar immuneprofiles; the ganglion-like cells stained for vimentin and actin and focally with KP1, suggesting myofibroblastic and histiocytic features. None of the lesions stained for keratin, desmin, or S-100 protein. Ultrastructural examination of two cases revealed cells with a constellation of fibroblastic, myofibroblastic, and histiocytic features. Follow-up of seven patients, averaging 58 months from diagnosis, confirmed that all are alive and well. Recognition of this cellular variant of proliferative fasciitis and myositis is important to prevent misdiagnosis as a sarcoma and unnecessary, excessive therapy.

Actins↗