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PubMed1992

Lymphocyte subsets in muscle biopsies of human T-lymphotropic virus type I carriers with polymyositis.

Recent epidemiologic studies have shown that human T-lymphotropic virus type I (HTLV-I) is associated with polymyositis. To clarify the histological features of HTLV-I positive polymyositis, we studied muscle tissue from two groups of patients with polymyositis, those seropositive for HTLV-I (5 patients) and those seronegative (5 patients). We examined the lymphocyte subsets in muscle biopsies using monoclonal antibodies. In the endomysium, a variable number of T-lymphocytes and macrophages and a smaller proportion of B-lymphocytes were found in both groups. In both HTLV-I positive and negative patients a variable number of T-lymphocytes and macrophages, with a smaller number of B-lymphocytes, were found in the perimysium. As a whole, the T-lymphocytes were predominantly of the helper/inducer phenotype in both groups. We failed to find any specific phenotype distribution among T-cells infiltrating the muscle of HTLV-I carriers with polymyositis.

Adult↗
PubMed1994

Fatal respiratory failure due to polymyositis.

A patient presenting as severe respiratory failure due to alveolar hypoventilation resulting from respiratory muscle weakness is described. Diagnosis of polymyositis was established by electromyography and muscle biopsy. Steroid therapy was initiated and it ameliorated respiratory failure remarkably. Selective respiratory muscle involvement due to polymyositis has been suggested in this patient. Although fatal respiratory failure is a very rare complication in polymyositis, polymyositis should be considered as one of underlying diseases causing severe respiratory failure.

Fatal Outcome↗
PubMed1995

Cost-effectiveness of MR imaging in evaluating polymyositis.

OBJECTIVE: Polymyositis is an inflammatory disorder of muscle that is often difficult to diagnose. The purpose of this study was to assess the usefulness and cost-effectiveness of MR imaging as a biopsy guide in patients with suspected polymyositis as an alternative to a blind biopsy. SUBJECTS AND METHODS: Our subjects were 25 patients with clinically suspected and ultimately confirmed polymyositis, 14 of whom had preoperative MR imaging at 1.5 T (including axial T1-weighted, axial T2-weighted [fat saturated], coronal T1-weighted, and coronal fast short-T1 inversion recovery images of both entire extremities). Images were prospectively interpreted for a specific location for a biopsy that showed the most muscle edema with the least atrophy. Surgical results and the estimated true cost of medical care (cost-accounting method) were compared between patients with preoperative MR images and patients whose biopsy site was selected on the basis of clinical criteria. RESULTS: Among the 14 patients whose biopsy site was selected on the basis of MR findings, only one biopsy result was false-negative. Patients without imaging had five biopsies with false-negative results. Patients with preoperative MR images averaged actual medical costs of $14,000 compared with $20,000 for those without MR images. Hospitalization time averaged 12 days for patients with MR images for guidance compared with 14 days for those without MR images. The average daily cost was $1,150 for the patients with MR images compared with $1,390 for those without. CONCLUSION: We conclude that MR imaging not only can aid in surgical planning but also can decrease the costs of patient care for patients with suspected polymyositis.

Adult↗
PubMed2002

A case of polymyositis with dilated cardiomyopathy associated with interferon alpha treatment for hepatitis B.

Polymyositis is a rare complication of interferon alpha treatment as a result of immune-modulating role of the drug itself. In this case, interferon alpha induced polymyositis and cardiomyopathy is diagnosed in a 33-yr-old male patient with history of chronic hepatitis B. To treat hepatitis B, interferon alpha was administered until the proximal muscle weakness developed. Thereafter, sixteen cycles of immunoglobulin treatment (400 mg/kg) along with corticosteroids were instituted and led to an improvement in subjective symptoms with decreases in level of CPK and LDH. However, dilated cardiomyopathy has not improved in spite of the cessation of interferon treatment. Unlike the persistently elevated serum HBV DNA level, the serum ALT and AST levels have gradually decreased. Our case shows that clinical symptoms of polymyositis improved with steroid and immunoglobulin treatment without deterioration of the hepatitis B. To our knowledge, this is the first case of polymyositis associated with dilated cardiomyopathy after the administration of interferon in a patient with hepatitis B.

Adrenal Cortex Hormones↗
PubMedDate not supplied

[Polymyositis in childhood].

INTRODUCTION AND OBJECTIVE: Idiopathic inflammatory myopathies are very rare in infancy. We present five cases of polymyositis in children in which the clinical variability and difficulty in diagnosis that occurs with this disorder are clearly seen, and analyze their response to steroid treatment. CLINICAL CASES: We can distinguish two groups of patients: the first is formed of the case of a two and a half month old baby with generalized hypotonia; and the second includes the other four cases, children of between 2 and 8 years old with weakness, which was mainly proximal. One of the second group later developed juvenile chronic arthritis. In all cases there was a raised CPK and a myopathic EMG, with or without spontaneous muscle activity. Muscle biopsy showed inflammatory myopathy. The first group corresponds to so-called infantile polymyositis which is characterized by generalized hypotonia. The second group includes older children in whom the clinical features of the disorder are similar to those in adults. The association of other autoimmune diseases with infantile polymyositis is exceptional. All cases show more or less improvement with corticosteroids. CONCLUSIONS: The polymyositis are extremely rare before puberty and have a broad clinical spectrum. Congenital cases have been described in infants and in older children. The biopsy results are usually unexpected. Treatment with corticosteroids leads to clinical improvement which may be only partial and is less favorable than in the dermatomyositis.

Adrenal Cortex Hormones↗
PubMed2000

Amyloid myopathy masquerading as polymyositis.

OBJECTIVE: It is not well appreciated that the clinical presentation of amyloid myopathy can mimic that of polymyositis. By retrospective clinicopathologic analysis we determined distinctive features of amyloid myopathy that differentiate the 2 diseases. METHODS: Two patients with clinical and histologic evidence of an inflammatory myopathy had fatal outcomes despite appropriate treatment for polymyositis. Their clinical course and original pathologic specimens were reviewed. In addition, original tissue samples were obtained and analyzed using Congo red staining and immunoperoxidase. RESULTS: The initial diagnosis of polymyositis was supported in both cases by muscle biopsies showing inflammatory infiltrates and elevations of creatine phosphokinase and by classic electromyography. Retrospective evaluation of the initial muscle biopsies disclosed subtle but incontrovertible evidence of vascular amyloid. Further analysis of the original specimens confirmed the presence of immunoglobin light chain (AL) amyloid. CONCLUSION: Amyloid myopathy can mimic polymyositis. Both can have similar clinical symptoms, as well as inflammatory infiltrates on muscle biopsy. Failure to recognize amyloid myopathy deprives patients of potentially life prolonging treatment. Congo red staining and immunohistochemical analysis of tissue could prevent misdiagnosis.

Aged↗
PubMed2000

[A case of polymyositis presenting pregnancy with acute respiratory failure].

A 33-year-old pregnant woman developed respiratory difficulty with bilateral pleural effusion 31 weeks into gestation. On admission she had an elevated serum level of creatine kinase, but muscle weakness in the extremities was mild. After an immediate and successful Cesarean section, she developed respiratory failure and mechanical ventilation was required. The patient was diagnosed as having polymyositis from the limb muscle biopsy. She was treated with dexamethasone at a dose of 8 mg, methylprednisolone++, 1 g daily for three days, and then prednisolone 60 mg daily. One week later the serum level of creatine kinase was normalized and the patient was weaned from ventilator support. The dose of prednisolone was tapered 10 mg every week and she was discharged two months after delivery without prednisolone. A chest CT scan revealed no interstitial pneumonitis or aspiration pneumonia, so her respiratory failure seemed to be ascribable to polymyositis-related respiratory muscle weakness and pleural effusion. However, severe involvement of respiratory muscles without generalized marked muscle weakness is extremely rare, and pleural complications have usually been described in association with pulmonary parenchymal diseases. During the following two years polymyositis has not recurred in this patient and there have been no data indicative of other overlapping collagen diseases. Reports of polymyositis which occurred during pregnancy are rare, and the pathogenesis and clinical outcome of these patients need to be carefully investigated.

Acute Disease↗
PubMed2000

[A case of steroid-resistant polymyositis who made a good response to high dose intravenous immunoglobulins (IVIG)].

We report a 69-year-old woman with steroid-resistant polymyositis who made a good response to high dose intravenous immunoglobulins (IVIG). She was diagnosed as polymyositis by muscle weakness of her extremities and bulbar muscles, higher level of serum CK and muscle biopsy findings of her biceps brachii muscle. Because of the coexistence of pulmonary tuberculosis and interstitial pneumonia, she was initially treated with 40 mg (1 mg/kg)/day of oral prednisolone, and methylprednisolone pulse therapy 500 mg/day intravenous drip (d.i.v.) x 3 days, 750 mg/day d.i.v. x 3 days. Since her condition deteriorated in spite of these conventional therapies, IVIG therapy 15 g (375 mg/kg)/day d.i.v. x 3 days was administrated. She showed remarkable improvement of serum CK level and muscle strength. Although corticosteroid is the mainstay of therapy for polymyositis, our case study showed that IVIG is also an efficacious therapy for polymyositis, especially resistant to the conventional treatment.

Aged↗
PubMed2000

[Intravenous immunoglobulins in the treatment of polymyositis and dermatomyositis].

Polymyositis and dermatomyositis are two inflammatory muscle diseases of unknown origin mediated by a dysimmune mechanism via a cytotoxic effect against the muscle fiber in polymyositis and a humoral effect against the muscle vessels in dermatomyositis. First line treatment of polymyositis and dermatomyositis is still based on corticosteroid therapy. In case of failure, corticosteroid dependence, or intolerance, a second line treatment using immunosuppressors or polyvalent human intravenous immunoglobulins (IVIg) is usually associated. We report our experience with IVIg in the treatment of 50 patients with myositis unresponsive to corticosteroids and immunosuppressors. Thirty-five patients had polymyositis and 15 had dermatomyositis. We discuss the possible mechanisms and their precise role in the therapeutic armamentarium in these dysimmune diseases.

Adrenal Cortex Hormones↗
PubMedDate not supplied

Polymyositis as a paraneoplastic manifestation of hepatocellular carcinoma.

A case of hepatocellular carcinoma associated with polymyositis is reported. A 70-year-old man noticed muscular weakness mainly in the proximal limb muscles. The clinical course, a raised level of serum creatine kinase and electromyographic findings suggested polymyositis, and the pathological findings on muscle biopsy were compatible with this diagnosis. Computed tomography of the upper abdomen revealed a mass lesion in segment VII and VIII of the liver, which was diagnosed pathologically as hepatocellular carcinoma. The patient underwent systematic resection of segment VII and the dorsal part of segment VIII of the liver. After surgery, the weakness improved and the serum creatine kinase level normalized without medical treatment for the polymyositis. The relief of neurological symptoms and signs after complete resection of the tumor strongly suggests paraneoplastic polymyositis, which has been described only rarely in association with hepatocellular carcinoma.

Aged↗
PubMed2000

[A patient of infantile polymyositis triggered by respiratory syncytium virus infection].

An 11-month-old boy developed acute polymyositis about 1 week after respiratory syncytium virus (RSV) pneumonia. He was admitted to our hospital because of interstitial pneumonia. RSV infection was confirmed by the presence of its antigen in his nasal discharge. Two weeks later, his chest X-ray findings improved and RSV antigen became negative, but severe generalized muscle weakness developed, causing respiratory failure. Muscle biopsy demonstrated inflammatory cellular infiltration with occasional fiber necrosis. Intravenous steroid pulse therapy was remarkably effective resulting in complete recovery of his muscle power. In this patient polymyositis was preceded with RSV infection, suggesting a close relationship between polymyositis and RSV infection. Although many patients of viral myositis have been reported to be associated with coxsackie B, HCV and HTLV-1 viruses, our patient is the first infantile polymyositis secondary to RSV infection.

Acute Disease↗
PubMed1994

Polymyositis-like syndrome in hypothyroidism: report of two cases.

An increase of muscle enzymes in hypothyroidism has sometimes been correlated with a polymyositis-like syndrome and hypothyroid patients have been misdiagnosed and mismanaged as suffering from polymyositis. Actually, muscle symptoms, such as aches and pain, stiffness, weakness and cramps or, more rarely, hypertrophy, are observed in hypothyroidism and increased serum muscle enzyme values, particularly the level of creatine phosphokinase (CPK), indeed seem to suggest polymyositis. The muscular groups most commonly affected by the above mentioned symptomatology are those of the shoulder and pelvic girdles. In this report two hypothyroid patients complaining of muscle symptoms, whose serum muscle enzymes were particularly elevated, are described. In the first case the patient had been suffering from pain and weakness of the thenar eminence for about 4 months. The clinical features suggested a diagnosis of Carpal Tunnel Syndrome, but thyroid function tests revealed primary hypothyroidism. In the second case the patient had been afflicted with muscular weakness of the shoulder girdle for 2 months and was unable to keep his arms raised over his head. A study of thyroid function demonstrated a picture of hypothyroidism. Both patients were treated with L-thyroxine and in a relatively short time biochemical parameters improved remarkably, and the symptoms disappeared. The hypothesis that a muscular effort of long duration by hypothyroid patients may have been responsible for the muscular damage and the symptoms may explain why only a few hypothyroid patients develop a clinical picture similar to polymyositis.

Adult↗
PubMed1995

[Successful combination therapy of cyclosporine and steroids in two cases with interstitial pneumonitis associated with polymyositis].

Cyclosporine is an immunosuppressive agent which is well-established in the transplantation of organs including kidney, liver and bone marrow. It acts by inhibiting the production of interleukin 2, thereby blocking both the development of cytotoxic lymphocytes, and the proliferation of helper T cells. T cell-mediated muscle damage is thought to be important in the pathogenesis of polymyositis. And activated cytotoxic T cells are thought to play an important role of polymyositis/dermatomyositis with active pneumonitis. It is thereby likely that cyclosporine would be effective in the management of polymyositis with interstitial pneumonitis. We have used cyclosporine in two cases of corticosteroids resistant polymyositis associated with pneumonitis. The first case was admitted because of the relapse of polymyositis. She was partially responded by the high dose of steroid, but showed decreased %DLCO and increased AaDO2 during the therapy. And oral cyclosporine was given with steroid. Within two weeks, serum creatinine kinase level was reduced to normal range, and the improvement of pneumonitis was observed. The second case was admitted because of the flare of pneumonitis. She was treated with high dose of steroid with insufficient response. And cyclosporine was prescribed. Within two weeks of treatment, her symptom was relieved, and blood gas analysis showed an improvement of pulmonary function. And steroid could be tapered. In both cases, the initial dose of cyclosporine was 200 mg/day, and the optimal trough level was thought to be ranged 100 to 150 ng/ml. In the second case, renal dysfunction was observed but it was recovered by the reduction of the dose of cyclosporine. No other side effect was appeared.(ABSTRACT TRUNCATED AT 250 WORDS)

Cyclosporins↗
PubMedDate not supplied

[Two cases of polymyositis with cardiac involvement].

Cardiac involvement in polymyositis is more prevalent than previously assumed. A 59-year-old and a 58-year-old man presented with cardiac involvement preceding skeletal muscular lesions, admitted because of increased levels of muscle-derived enzymes and left ventricular dysfunction with ECG abnormalities. Coronary angiography revealed no stenotic lesions. Right ventricular endomyocardial biopsy disclosed myocarditis. Left ventriculography showed local asynergy of cardiac wall motion. After admission the weakness and atrophy of skeletal muscles progressed gradually and high levels of muscle-derived enzymes persisted. Electromyography and skeletal muscle biopsy confirmed the clinical diagnosis of polymyositis. Both patients were diagnosed as having polymyositis with cardiac involvement, and treatment with steroids was started. Symptoms improved significantly, and the CK enzyme level was reduced effectively. The condition of one patient was well controlled, but the other suffered from repeated heart failure due to severe left ventricular dysfunction. The clinical spectrum of polymyositis is wide and variable. Further studies are needed to evaluate the detection, management, and prognosis of the disease as well as the pathogenesis and to prevent progression of cardiac involvement.

Electrocardiography↗
PubMed1993

Xe-133 muscle blood flow in polymyositis and dermatomyositis.

The histologic picture of polymyositis or dermatomyositis is characterized by muscle fiber degeneration, which is assumed to be caused by a circulatory abnormality. To verify this assumption, we measured the muscle blood flow in patients with polymyositis (or dermatomyositis) and in normal subjects using the inert gas Xe-133. Sixteen patients and 30 age-matched normal volunteers were studied. Muscle blood flow was measured with Xe-133 dissolved in isotonic saline as described by Lassen et al. (1964) and Pozderac et al. (1975). Data were acquired in the frame mode after intramuscular injection (anterior tibial muscle) of 0.3 mCi of Xe-133 in 0.2ml of saline for 20 min. A computer routine was used to calculate the clearance half time (T 1/2) in min and flow rate (Q) in ml/100 gm tissue/min. The student's t test was used to test statistical significance. Our preliminary results suggest: 1. The muscle blood flow in patients with polymyositis and dermatomyositis was significantly lower than the corresponding normal control levels. 2. This findings may be due to decreased in muscle capillary 3. Further studies are warranted to obtain a conclusive understanding of the pathogenesis of polymyositis and dermatomyositis.

Adult↗
PubMed1995

[Recurrent bronchiolitis obliterans organizing pneumonia as first manifestation of polymyositis].

The case of an idiopathic polymyositis preceded by several episodes compatible with the diagnosis of bronchiolitis obliterans organizing pneumonia is herein reported. This kind of pulmonary affection in polymyositis can be simultaneous or later to the diagnosis of the systemic disease, but as the first manifestation is exceptional. In the case we report, paradoxically, when the diagnosis of polymyositis was made the patient had not clinical or radiographic bronchopulmonary affection. If the first clinical manifestation of an autoimmune disease like polymyositis can be an idiopathic bronchiolitis obliterans organizing pneumonia, we think that a prolonged follow up of these patients is necessary.

Anti-Inflammatory Agents↗
PubMed1981

Polymyositis beginning as a focal process.

Six patients with polymyositis initially complained of a single, localized, painful mass involving an extremity. The mass enlarged during the course of a two- to six-week period. Biopsy of the lesion revealed myopathic changes with inflammation, and the diagnosis of benign inflammatory pseudotumor was considered. The mass regressed, but during the next three to six months, a rapidly progressive generalized myopathy developed that caused weakness of the trunk and extremities in association with malaise and weight loss. Subsequent muscle biopsy specimens obtained from a site remote from the original mass were again characterized by lymphocytic infiltration, fiber necrosis, and regenerative activity. Our experience indicates that polymyositis may begin as a focal process that mimics a localized inflammatory pseudoneoplastic reaction. An essential clue to the diagnosis of polymyositis at this early stage is the elevation of ESR and serum creatine phosphokinase levels that does not occur in pseudotumor. The muscle biopsy further serves to distinguish polymyositis from pseudotumor.

Adult↗
PubMed1981

Patients with polymyositis and dermatomyositis who undergo plasmapheresis therapy. Pathologic findings.

Muscle biopsy specimens were studied in 20 of 26 patients who had polymyositis or dermatomyositis prior to treatment with plasmapheresis and immunosuppressive therapy. Morphologic findings from biopsy specimens were similar among patients with polymyositis and dermatomyositis, except that perifascicular atrophy was a prominent feature in patients with dermatomyositis and inconspicuous or absent in patients with polymyositis; immunofluorescent staining of muscle for immunoglobulins and/or complement was noted more frequently for patients with dermatomyositis than those with polymyositis. Patients judged clinically to have highly active disease had the most severe changes, which consisted of muscle necrosis, regeneration, phagocytosis, and inflammation; those with mild and moderately active disease were indistinguishable pathologically. No correlation between duration of disease and degree of pathologic alteration was found. Six of seven patients with both pretreatment and posttreatment biopsies showed marked improvement in the extent of pathologic alteration, and a statistically significant reduction in the degree of muscle atrophy following treatment.

Adolescent↗