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PubMed2004

Inflammatory disorders of muscle: progress in polymyositis, dermatomyositis and inclusion body myositis.

PURPOSE OF REVIEW: To provide an update on the major advances in inflammatory myopathies. RECENT FINDINGS: Polymyositis is an uncommon disorder that can be misdiagnosed when the old, and never validated, criteria of Bohan and Peter are used. New diagnostic criteria were recently introduced, in which the MHC/CD8 complex is considered a specific immunopathological marker because it distinguishes the antigen-driven inflammatory cells that characterize polymyositis and sporadic inclusion-body myositis from the non-specific, secondary inflammation seen in other disorders, such as dystrophies. In sporadic inclusion-body myositis the inflammatory cells invade non-vacuolated fibers, whereas the vacuolated fibers are not invaded by T cells, implying two independent processes, a primary immune process with antigen-driven T cells identical to polymyositis, and a degenerative process in which beta-amyloid and amyloid-related proteins participate in vacuolar degeneration. In polymyositis and sporadic inclusion-body myositis, antigen-specific and clonally expanded autoinvasive T cells persist for years, even in different muscles, as reconfirmed by proof-of-principle techniques involving CDR3 spectratyping combined with laser microdissected single-cell polymerase chain reaction of the T-cell receptor genes. The formation of immunological synapse between autoinvasive T cells and muscle fibers was recently strengthened by the upregulation of co-stimulatory molecules ICOS/ICOS-L and PD-L1. A new, distinct myopathy characterized by T-cell-triggered macrophage hyperactivation has now been recognized in patients with dermatomyositis-like disease. SUMMARY: Despite recent progress, the antigen(s) responsible for T-cell activation in polymyositis and sporadic inclusion-body myositis and the cause of vacuolar degeneration in sporadic inclusion-body myositis remain unclear. Newer, more aggressive immunotherapies may be encouraging, but control trials are needed to prove efficacy.

Amyloid↗

PubMed2005

Interstitial lung disease in polymyositis and dermatomyositis.

PURPOSE OF REVIEW: The purpose of this review is to discuss current concepts regarding management of interstitial lung disease in polymyositis and dermatomyositis. RECENT FINDINGS: Interstitial lung disease seems to be a more frequent manifestation in patients with polymyositis and dermatomyositis than previously reported. Modern technology, including high-resolution computerized tomography in combination with pulmonary function tests provides sensitive tools to detect early signs of interstitial lung disease. By systematic use of these investigations in newly diagnosed polymyositis and dermatomyositis, up to two thirds of patients were discovered to have signs of interstitial lung disease in a recent study. Clinical symptoms such as cough and dyspnea may not be sensitive enough to detect interstitial lung disease. Awareness of this complication in patients with myositis is important, because early diagnosis and management of interstitial lung disease may prevent development of chronic pulmonary fibrosis and thereby prolong patient survival and improve quality of life. Treatment recommendations of interstitial lung disease in polymyositis and dermatomyositis are still limited by absence of controlled trials and could only be based on experiences from small case series and case reports. At least some patients with interstitial lung disease improve with immunosuppressive treatment, but data are limited, and longitudinal studies are needed. SUMMARY: Interstitial lung disease seems to be a common manifestation in patients with polymyositis and dermatomyositis already at diagnosis of the muscle disease. When present, interstitial lung disease has a major effect on morbidity and mortality and should be looked for in these patients using high-resolution computerized tomography and pulmonary function tests early in the disease course, because immunosuppressive treatment may change the course of the lung disease.

Biopsy↗

PubMed2005

Acute polymyositis after donor lymphocyte infusion.

Polymyositis usually occurred along with other manifestations of chronic graft-versus-host disease (GVHD) after allogeneic hematopoietic stem cell transplantation (HSCT) had been reported. However, polymyositis with a sole manifestation of acute GVHD following donor lymphocyte infusion (DLI) is rare. We reported a 45-yr-old man of acute lymphoid leukemia post-allogeneic HSCT 6 months developed acute polymyositis after DLI. He did not develop any symptoms, signs of acute or chronic GVHD following allogeneic HSCT, despite withdraw of immuosuppresive agents, cyclosporin A (CsA). As the DNA-STR of bone marrow analysis showed mixed chimerism, he received the DLI for remission on May 16, 2002. Acute polymyositis developed following DLI 22 d later. The clinical presentation of polymyositis is compatible with a manifestation of acute GVHD following DLI. It also responds to the treatment of steroid and CsA.

Cyclosporine↗

PubMed2001

Polymyositis, pulmonary fibrosis and malignant lymphoma associated with hepatitis C virus infection.

Polymyositis has been associated with various viral infections, and a spectrum of immune-related diseases may occur with hepatitis C (HCV) infection. Both polymyositis and HCV infection may be accompanied by pulmonary fibrosis. An association between polymyositis and malignancy has also been reported. We report a 55-year-old woman accompanied cryoglobulinemia with HCV infection and manifesting polymyositis, pulmonary fibrosis and malignant lymphoma. Steroid therapy was effective to improve interstitial pneumonia, polymyositis, and liver function.

Cryoglobulinemia↗

PubMed1993

Association between bovine collagen dermal implants and a dermatomyositis or a polymyositis-like syndrome.

OBJECTIVE: To determine whether an excess incidence of dermatomyositis or polymyositis or both exist in patients treated with injectable bovine collagen implants and to characterize the clinical picture. DESIGN: Historical cohort study (July 1980 through June 1988). PATIENTS: Patients were identified from personal experience or adverse reaction reports received by the manufacturer. SETTING: An 8-year period in the United States during which approximately 345,000 patients received implants. RESULTS: Eight patients with dermatomyositis and an additional patient with polymyositis were identified from approximately 345,000 patients receiving injectable bovine collagen implants from July 1980 through June 1988. The nine patients with dermatomyositis or polymyositis were diagnosed an average of 6.4 months (range, 0.7 to 24.9 months) after collagen implant or skin test exposure or both. Eight of the nine patients had a delayed-type hypersensitivity response at the test or treatment sites or both, and five of six patients tested were found to have increased serum antibodies to collagen. Compared with the general population, the incidence of dermatomyositis or polymyositis among collagen-treated patients was statistically increased (standardized incidence ratio, 5.05; 95% CI, 2.31 to 9.59; P < 0.0001). A similar analysis of the eight dermatomyositis case patients produced a standardized incidence ratio of 18.8 (CI, 8.1 to 37.0; P < 0.0001). Using a Monte Carlo simulation, an interval of 6.4 months or less from exposure to onset of disease was found to be an extremely rare event, occurring less than 72 times per one million simulation trials (CI, 57 to 91). CONCLUSIONS: Because these data suggest that an immunologic response to bovine type I or type III collagen or both caused this dermatomyositis or polymyositis-like syndrome, the risks versus benefits for the cosmetic use of collagen implants should be reassessed.

Adult↗

PubMedDate not supplied

Polymyositis: rare complication of hepatitis B vaccination. An unusual cause of toxic shock syndrome.

Transient weakness of the legs developed in a 17 year-old male high school student three weeks after the initial injection of a hepatitis B vaccine. Seventeen days after the second injection of the vaccine, low-grade fever, a pruritic maculopapular rash appeared and weakness of the legs recurred. This was associated with elevation of the creatinine kinase to 2,406 U/L. The day after admission he became afebrile and in the subsequent four days the rash improved but leg weakness persisted. One-month later, muscle strength had returned; and the creatinine kinase had returned to normal levels. The only case of dermatomyositis associated with hepatitis B vaccination and the findings in the six reported cases of surface antigen-positive hepatitis associated with polyomyositis or dermatomyositis are briefly reviewed. Hepatitis B vaccination should be encouraged, but it is important to be aware that, rarely, dermatomyositis, polymyositis or neurovascular complications may occur. Polymyositis associated with the administration of the hepatitis B vaccine or with hepatitis B virus infection is a rare occurrence. A Medline Search performed from 1960 to January 2002 associating hepatitis B vaccine or hepatitis B virus with myopathy, myositis, polymyositis and dermatomyositis, showed only one case of dermatomyositis related to the hepatitis B vaccine, and six case reports relating polymyositis to hepatitis B virus infection. We present a case where a causal relationship between polymyositis and hepatitis B vaccination appears quite likely.

Adolescent↗

PubMed2005

[A study of vascular factor in the pathogenesis of polymyositis].

OBJECTIVE: To study ICAM-1, IL-1alpha expression in capillaries and to explore whether capillaries play a role in the pathogenesis of polymyositis. METHODS: Ten cases of patients with polymyositis and 6 patients with other myopathies were collected. CD34, ICAM-1 and IL-1alpha were selected for a quantitative measurement using an image analysis system, and results were presented as the percent of positive staining in the tissue area. RESULTS: We found that the percent of capillaries in tissues from polymyositis was significantly less than that from controls (P = 0.009). ICAM-1 and IL-1alpha expression in endothelial cells of capillaries were stronger in polymyositis patients. CONCLUSION: Our findings indicated that changes of capillaries might participate in the pathogenesis of polymyositis.

Adolescent↗

PubMed1996

[Polymyositis after interferon beta treatment of chronic hepatitis type C].

We reported a case of polymyositis manifested after beta-interferon therapy for chronic viral hepatitis type C. In November 1992, a 62-year-old female was diagnosed as having chronic viral hepatitis type C. In January 1993, she was treated with beta-interferon(total 2.52 x 10(8)IU). In March 1993, after the treatment, she noticed weakness and atrophy of her lower extremities. Neurological examination revealed proximal dominant muscle weakness and atrophy of the extremities. Increased levels of serum CK and aldolase were disclosed on admission. By needle EMG, a low voltage with short duration interference pattern was noted. February 7, by muscle biopsy of the right quadriceps, necrotic myofibers, marked variation in fiber diameter, stromal mononuclear cell infiltration, and endomysial fibrosis was detected. The immunological stain of infiltrating cells in the muscle fibers revealed CD4:CD8 ratio as 64:55, whereas in conventional polymyositis CD8 cells dominate CD4 cells. Diagnosis of polymyositis was made and oral prednisolone 60 mg a day was started. Her symptoms and signs improved gradually and she was discharged after 2 months. Many adverse effects due to immunological disturbances caused by interferon treatment have been reported, but polymyositis due to interferon has been very rare. When interferon will be prescribed, manifestation of polymyositis should be considered.

Chronic Disease↗

PubMedDate not supplied

Long-term prognosis of 69 patients with dermatomyositis or polymyositis.

OBJECTIVES: To assess the long-term prognosis of dermatomyositis and pol myositis. METHODS: 69 patients with dermatomyositis or polymyositis were selected according to the diagnostic criteria of Bohan and Peter and were followed up for a minimum of 6.3 years (for surviving patients) (mean 11.6 years). Clinical and biological features, and pulmonary and muscle parameters were considered as prognostic factors for death. Functional disability was assessed using a 4-stage grading system. RESULTS: 30 deaths (43.5%) occurred mainly due to cardiovascular (8), pulmonary (8), carcinomatous (5) and iatrogenic complications (5). Survival rates were 82.6% at 1 year, 73.9% at 2.66, 7% at 5 and 55.4% at 9. Significant prognostic factors for death (Cox model with time-dependent covariates) were old age (p < 0.0001), dysphonia (p < 0.001), pulmonary interstitial fibrosis (p < 0.02), absence of dysphagia (p < 0.02) and asthenia-anorexia (p < 0.05). Dermatomyositis and polymyositis subgroups had slightly different significant prognostic factors for death: old age, cancer, pulmonary interstitial fibrosis and asthenia-anorexia for dermatomyositis; old age, failure to improve muscle strength in response to treatment after one month, and the absence of myalgia as presenting symptom for polymyositis. At the end of the follow-up, 33/39 surviving patients (84.6%) had no or insignificant muscular disability, whereas 3 children were bedridden due to generalized calcinosis. CONCLUSIONS: High mortality occurred in the first year, and the survival rate decreased continually up to 9 years. The main prognostic factor for death is old age, but dermatomyositis and polymyositis must be considered separately. General features (pulmonary fibrosis, cancer, asthenia-anorexia) are involved in dermatomyositis, whereas muscular symptoms are the most significant in polymyositis. The long-term functional prognosis was fairly good, except for generalized calcinosis, which tended to occur in childhood dermatomyositis.

Adolescent↗

PubMed1998

[Polymyositis-induced respiratory failure in the presence of antecedent pleural effusion].

Pulmonary involvement is generally recognized to be an important determinant of the clinical course in cases of polymyositis. A patient with bilateral pleural effusion as an initial symptom developed respiratory failure due to muscle weakness. Polymyositis was diagnosed on the basis of a muscle biopsy. A chemotherapy regimen consisting of methyl prednisolone (1 g/day for 3 days) followed by prednisolone (60 mg/day) alleviated, the pleural effusion and respiratory failure. Though pleural complications in polymyositis have been described mostly in association with concomitant interstitial lung disease, our patient exhibited only bilateral pleural effusion. Given that pleural effusion can be an initial symptom of polymyositis, it seems necessary that polymyositis be taken into consideration by differential diagnoses of pleural effusion of unknown etiology.

Aged↗

PubMed1990

Polymyositis: a case history approach to the differential diagnosis and treatment.

A wide range of conditions can mimic polymyositis. Thus, diagnosing this condition can be a challenge. Although no single criterion is diagnostic of polymyositis, the following criteria have been proposed and widely used: (1) symmetric proximal muscle weakness; (2) characteristic violaceous rash on the hands, elbows, and knees; (3) increased muscle enzymes in the serum; (4) characteristic electromyographic findings (insertional activity, fibrillation potentials, motor unit potentials of increased frequency and decreased duration, and normal conduction velocity in nerves); and (5) muscle biopsy specimen with characteristic inflammatory and myopathic changes. Although polymyositis primarily involves muscle, up to 20% of patients may have extramuscular problems. The main treatment for polymyositis is high-dose corticosteroids. In corticosteroid-resistant patients, methotrexate is often effective. In this report, case histories are presented to highlight the usefulness and the limitations of the common diagnostic criteria for polymyositis.

Adrenal Cortex Hormones↗

PubMed1990

[A case of polymyositis associated with chronic active hepatitis].

A case of polymyositis associated with chronic active hepatitis was reported. A 53-year-old man, who had no previous history of blood transfusion nor hepatitis, noticed proximal dominant muscle weakness on January 29, 1985. He was admitted to Kyoto National Hospital on February 7, and laboratory studies disclosed the elevation of serum enzyme levels; creatine kinase (CK) 9845 IU/L (normal 54-263), glutamate oxaloacetate transaminase (GOT) 834 IU/L (9-31), glutamate pyruvate transaminase (GPT) 491 IU/L (4-34), lactate dehydrogenase (LDH) 2135 IU/L (248-464). Also serum gamma globulin was high (1.8 g/dl) and LE-like cell was found. The diagnosis of polymyositis was made and prednisolone therapy (60 mg/day) was started on February 23. The elevated serum enzymes decreased gradually, but severe muscle weakness persisted for about one month. On April 3, he was admitted to our hospital. Physical examination revealed moderate proximal dominant muscle weakness without skin eruption, jaundice or hepatosplenomegaly. The serum enzymes were still high; CK 1826, GOT 173, GPT 232 (GOT less than GPT), LDH 1548. However, alkaline phosphatase (ALP) and bilirubin were normal. Hepatitis B surface antigen (HBsAg) was not detected. Antinuclear antibody was positive. The electromyogram study showed myopathic change, and the muscle biopsy demonstrated myopathic change and cell infiltration, compatible with polymyositis. These results suggested liver dysfunction associated with polymyositis. Prednisolone therapy was continued and muscle weakness decreased. From December, 1985, serum enzymes (CK, GOT, GPT, LDH) elevated again and muscle weakness also slightly increased. Anti-smooth muscle antibody was positive. It was suggested that both polymyositis and liver dysfunction deteriorated.(ABSTRACT TRUNCATED AT 250 WORDS)

Autoantibodies↗

PubMed1989

[Spectrum of antinuclear and anti-cytoplasmic antibodies in dermatomyositis and polymyositis overlap syndromes].

Antinuclear antibodies (ANA) of the IgG class were detected in 60% of patients with dermatomyositis. Only in 16% could we demonstrate precipitating antibodies against the nuclear Mi-2 antigen. These antibodies are considered to be serological markers of dermatomyositis. The ANA spectrum in polymyositis-overlap syndrome was considerably more heterogeneous: patients with polymyositis/progressive systemic scleroderma overlap had antibodies against the nucleolar PM-Scl antigen or the nuclear Ku antigen. Cytoplasmic antibodies to Jo-1 were present in polymyositis associated with Sjögren's syndrome and pulmonary fibrosis. Antibodies against ribosomal ribonucleoprotein were found in polymyositis with systemic lupus erythematosus and antibodies to nuclear ribonucleoprotein in polymyositis associated with mixed connective tissue disease. The investigation demonstrates that the characterization of ANA specificities helps to differentiate between dermatomyositis and distinct forms of polymyositis-overlap syndrome.

Adult↗

PubMed1986

[135 cases of polymyositis].

The classification of 135 patients with either dermatomyositis (DM) or polymyositis (PM) showed 34 cases of DM and 56 cases of PM in adults, 6 cases of DM associated with cancer, 9 cases of DM in children, 16 cases of localized PM and 14 cases of an overlapping syndrome. Results of biological tests, erythrocyte sedimentation rate and serum enzyme determinations were inconstantly abnormal. The electromyograms were generally of a myogenic type with spontaneous activity in about half the cases. Muscle biopsy usually showed inflammation necrosis and regeneration, sometimes only of moderate severity only. Results were normal in several cases. In 21 patients only the pathognomonic perifascicular atrophy was reported. Proposed classifications are unsatisfactory. Polymyositis may be considered as a syndrome. Among the primary forms the distinction between acute dermatomyositis and subacute or chronic polymyositis is poorly defined and passage from one disorder to another is frequent. Pseudo-myasthenic forms are not entities and pseudo-myopathic types are actually muscular dystrophies. Associations with polymyositis are common and may consist only in the addition of one sign of no clinical significance (PM "plus"). The polymyositis lesion may be part of a syndrome such as Gougerot-Sjögren's or of another connective tissue disease. A system of diagnostic criteria uses numerical ratings of each criterion as a function of its semiologic importance.

Adrenal Cortex Hormones↗

PubMed1984

Gynecologic carcinoma associated with dermatomyositis-polymyositis.

Dermatomyositis-polymyositis was associated with gynecologic carcinoma in ten patients treated at the Mayo Clinic between 1952 and 1982. Only two patients did not manifest the typical skin changes of acute dermatomyositis. In all ten cases, the onset of dermatomyositis-polymyositis preceded evidence of carcinoma. The intervals between initial symptoms of dermatomyositis-polymyositis and subsequent diagnosis of carcinoma ranged from three months to six years. Usually, the carcinoma became evident within two years. The carcinomas were ovarian in five cases, cervical in three, endometrial in one, and vaginal in one. Ages of the ten patients ranged from 40 to 66 years (mean, 53 years). In most cases, symptoms of dermatomyositis-polymyositis regressed markedly after diagnosis and treatment of the underlying carcinoma. In selected patients for whom more detailed follow-up information was available, the manifestations of dermatomyositis-polymyositis progressed and regressed in synchrony with tumor spread and effective treatment, respectively.

Adenocarcinoma↗

PubMed1983

Radioimmunoassay for antibodies to human skeletal muscle myosin in serum from patients with polymyositis.

Antibodies to human skeletal muscle myosin were detected in 90% of sera from patients with polymyositis by radioimmunoassay using purified human myosin as antigen, and the mean titre was 4.24 X 10(-10)M. The incidence and the mean titre of anti-myosin antibodies were significantly higher than in patients without polymyositis. Anti-myosin antibody titres correlated with steroid therapy in polymyositis patients. Titres in untreated patients with polymyositis correlated with the severity of muscle weakness. Radioimmunoassay for anti-myosin antibodies should prove to be useful in the diagnosis of polymyositis and the evaluation of clinical state.

Adult↗

PubMed1977

Polymyositis.

Polymyositis is a treatable and curable disease. Unfortunately, patients with polymyositis are still frequently diagnosed as having muscular dystrophy. It is important to have a high index of suspicion of polymyositis, and to recognize that the detailed symptoms and signs and the overall picture are different from the relatively clear-cut pattern of the muscular dystrophies. Treatment of polymyositis is rewarding, although it frequently requires considerable resilience from both patient and doctor to achieve the best results. A detailed search for an underlying aetiology should be made, and a carcinoma should be suspected in elderly patients and those with dermatomyositis. In patients in whom polymyositis is related to a carcinoma, the latter may be at a stage where total excision is possible.

Azathioprine↗

PubMed2002

Results and long-term followup of intravenous immunoglobulin infusions in chronic, refractory polymyositis: an open study with thirty-five adult patients.

OBJECTIVE: Polymyositis is a rare inflammatory muscular disease of unknown cause. Corticosteroids and immunosuppressive drugs are the first choice of therapy but are not always effective and may cause serious side effects. Many studies have shown that polyvalent intravenous immunoglobulin (IVIG) may be of interest for the treatment of dermatomyositis. We carried out an open, prospective study to evaluate the efficacy of IVIG in subjects with polymyositis that was refractory to traditional treatments, and we evaluated the benefits of this therapy over a long-term period of followup. METHODS: Thirty-five adult white patients (20 female, 15 male, mean age 43.5 years [SD 16.8]) with chronic, refractory polymyositis were treated with high doses of IVIG, after the patients had received the following traditional treatments: prednisone (n = 35), methotrexate (n = 24), azathioprine (n = 13), cyclophosphamide (n = 4), cyclosporine (n = 7), chlorambucil (n = 1), plasmapheresis (n = 8), lymphopheresis (n = 1), and total body irradiation (n = 1). There had been no changes in the patients' treatment in the 2 months before the initiation of IVIG therapy, and doses were not increased during IVIG treatment. We used preparations of polyvalent human IVIG with increased concentrations of intact IgG. The patients received 1 gm/kg/day for 2 consecutive days per month. The mean course of treatment was 4-6 months. The clinical assessment involved the evaluation of proximal muscle power, muscle disability scale score, and esophageal disorders. The biochemical evaluations carried out before each treatment period were compared by Student's t-test and nonparametric Wilcoxon test. Results were considered to be significant at P = 0.05. RESULTS: In the short-term, significant clinical improvement was noted in 25 of the 35 patients (71.4%). Mean muscle power was estimated before and after IVIG therapy and was found to be significantly improved (P < 0.01). All patients had a significant biochemical response. Mean creatine kinase levels during IVIG therapy decreased significantly before the fourth IVIG perfusion (P < 0.01). Side effects, usually minor, were noted in 6 patients. This benefit allowed the initial prednisone dose to be reduced by >50% in all patients. The mean (+/- SD) followup time for the 25 patients who responded favorably to IVIG treatment was 51.4 +/- 13.1 months. Twelve of these 25 patients remained in full remission following their initial course of IVIG, resulting in complete stoppage of medication in 5 patients or low doses of steroids in 7 patients. The condition of 6 patients remained improved and no other drugs were prescribed, but the patients remained dependent on IVIG infusions. Seven of the 25 patients who responded well to IVIG treatment relapsed at an average of 17.1 months (range 4-23 months) after the discontinuation of IVIG. CONCLUSION: IVIG is an interesting therapy for the treatment of polymyositis, with results showing that the condition of approximately 70% of the patients tested improved. After the discontinuation of the IVIG therapy, the efficacy remained stable in 50% of the patients, with a followup of over 3 years.

Adult↗