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PubMed2004

Pruritus in adult dermatomyositis.

Dermatomyositis has a significant clinical component of pruritus that has not yet been studied. Pruritus can significantly affect the life of patients. The aim of the present work was to study the degree of pruritus experienced by patients. A four-question survey was sent to patients with documented dermatomyositis. The survey used a 100-mm Visual Analogue Scale (VAS) to describe current, worst and daily pruritus, and the effect this has on daily activities. Twenty-six subjects returned completed questionnaires: four had no pruritus; the majority had a significant amount with means above 50 on the VAS. A mean of 44.6 was found for the effect on daily life. Further studies should be performed to examine the prevalence and severity of pruritus in this population and it's effect on their quality of life. Clinicians must be aware of the significant pruritus and provide adequate therapy to improve quality of life.

Adult↗
PubMed2004

Oligoclonal expansion of circulating and tissue-infiltrating CD8+ T cells with killer/effector phenotypes in juvenile dermatomyositis syndrome.

Although triggering by infectious agents and abnormal immune responses may play some role in the pathogenesis of juvenile dermatomyositis syndrome (JDMS), the precise mechanism of muscle destruction and vascular damage is largely unknown. In this study, we tried to elucidate the role of cytotoxic T cells in two patients with JDMS, who were diagnosed based on the characteristic symptoms, laboratory data, MRI findings and electromyographic patterns. Peripheral blood T cell phenotypes were determined by flow cytometry, using mAbs against specific T cell receptor (TCR) Vbetas. Complementarity-determining region3 (CDR3) size analysis was performed by gene scanning of CDR3 polymerase chain reaction (PCR) amplification products specific for each Vbeta. Subsequently, CDR3 nucleotide sequences were obtained after cloning of the predominant products. The distribution of lymphocytes infiltrating the muscle tissue was analysed by immunohistochemistry. In both patients examined, a unique combination of TCR Vbeta repertoires was increased within the CD8+ T cells. These subpopulations expressed a characteristic phenotype, indicating that they are memory/effector T cells with killer functions. At the same time, immunohistological and molecular biological examinations of the biopsied muscle samples revealed that identical CD8+ T cell clones with identical phenotypes/TCR Vbeta infiltrated within the inflammatory tissue, in particular around vessels. These findings indicate that oligoclonal expansion of CD8+ T cells plays a central role in the pathogenesis of muscle injury in the juvenile form of dermatomyositis syndrome and may provide a useful clinical parameter of disease activity and responsiveness to anti-inflammatory therapy.

Antigens, CD↗
PubMed2004

Acute thrombotic thrombocytopenic purpura following doxycycline treatment of Chlamydia pneumoniae infection in a patient with dermatomyositis.

A 48-year-old white female who was suffering from dermatomyositis and Chlamydia pneumoniae infection, developed acute rapidly fatal thrombotic thrombocytopenic purpura (TTP) following treatment with steroids and doxycycline. As a relationship between TTP and the inflammatory myopathies is now probably well established, it is very likely that our patient's TTP became manifest in association with dermatomyositis. Nevertheless, C. pneumoniae infection and doxycycline therapy cannot be excluded entirely as cofactors responsible for triggering her thrombotic microangiopathy.

Administration, Oral↗
PubMedDate not supplied

Dermatomyositis.

Dermatomyositis and polymyositis are diseases which must be carefully defined in each patient. Criteria such as those suggested by Bohan and Peter will help define the disease and exclude other possible causes of myositis. Immunological factors are probably involved in the pathogenesis of the disease, although varied types of injury may manifest in clinically similar disease. Dermatomyositis, not polymyositis, is related to internal malignancy in adults; however, an extensive malignancy evaluation is not indicated. Therapy with corticosteroids will improve the quality of life, will prevent contractures and calcinosis and may improve the chance of survival. In cases where steroids are ineffective, immunosuppressives may be helpful.

Dermatomyositis↗
PubMed1985

Dermatomyositis and cancer in Singapore.

Of 12 patients with adult dermatomyositis studied, 5 (41%) had associated malignancies, of which 3 were nasopharyngeal carcinomas. The youngest was a 23-year-old woman. In view of the high incidence of cancer and the predominance of nasopharyngeal carcinoma, malignancy evaluation in Chinese patients from the Far East, 20 years of age or older with dermatomyositis should have extended workups for postnasal cancer. Two patients who had positive antinuclear not have associated cancers. Three patients died of acute myocardial infarction.

Adult↗
PubMed1985

Discordance of skin and muscle involvement in dermatomyositis.

The skin manifestations of dermatomyositis infrequently occur without the myositis. A 24-year-old woman presented with concordance of the skin and muscle components of dermatomyositis, followed by a remission of the myositis with a persistence of significant rash for 17 years, finally presenting with a flare of both skin and muscle components together. Clinicians should be alert to the recurrence of an underlying myositis at any time.

Adult↗
PubMed1992

Severe acute form of adult dermatomyositis treated with cyclosporine.

The classical treatment of severe forms of dermatomyositis includes high doses of steroids and/or cytotoxic agents. Acute forms are frequently life threatening. Because cyclosporine is a fast-acting immunosuppressive drug, it appears to be a good candidate for the treatment of refractory forms of acute dermatomyositis. We report a dramatic improvement of a severe, acute, steroid-resistant adult form after cyclosporine administration. A rapid clinical and biochemical improvement is reported, and the reversibility of immunologic abnormalities is emphasized.

Acute Disease↗
PubMedDate not supplied

Combined hepatocellular-cholangiocarcinoma associated with dermatomyositis.

A 56 year old female developed combined hepatocellular cholangiocarcinoma associated with dermatomyositis. Serum tumour markers except for carbohydrate antigen (CA 19-9; 6400 ng/ml) were within normal range. Despite extensive chemotherapy, no clinical response was obtained and the patient's course deteriorated after 4 months. Macroscopically, the liver was mainly occupied by hepatocellular carcinoma but cholangiocarcinoma was found in the hilum. This is the first case of a rare association of combined hepatocellular-cholangiocarcinoma and dermatomyositis.

Adenoma, Bile Duct↗
PubMed1985

Recurrent photosensitive dermatitis preceding juvenile dermatomyositis.

The classic features of childhood dermatomyositis include muscle weakness, elevated muscle enzymes, and characteristic abnormal muscle biopsy and electromyography. Paramount to the diagnosis are cutaneous dermatoses that include a heliotrope rash and Gottron's papules. Rarely, a photo-sensitive dermatosis may occur. A recurrent photoexacerbated dermatoses can be an initial sign of occult childhood dermatomyositis.

Adrenal Cortex Hormones↗
PubMedDate not supplied

Partial lipodystrophy associated with juvenile dermatomyositis: report of two cases.

A 27-year-old woman and a 13-year-old girl diagnosed with juvenile dermatomyositis in childhood developed clinical findings of partial lipodystrophy 10 years after diagnosis. Exhaustive clinical and laboratory examinations showed an association with other abnormalities: hypertrichosis, steatohepatitis, and an abnormal insulin response to the glucose loading test in the first patient. Hypertrichosis, steatohepatitis, insulin-resistant diabetes mellitus, and acanthosis nigricans were observed in the second patient. Renal function was normal in both patients. Although a localized form of lipodystrophy has been reported associated with connective tissue disease (connective tissue lipoatrophy), the partial form has been infrequently described in association with juvenile dermatomyositis.

Adolescent↗
PubMed2005

[The clinical spectrum of dermatomyositis].

Dermatomyositis is characterized by marked clinical heterogeneity. Many classification schemes are available, based on internal organ involvement, presence of circulating autoantibodies and additional symptoms characteristic for other connective tissue diseases. Viral infections and underlying malignancies are possible mechanisms to trigger this autoimmune disease. Intensive clinical investigations to detect such malignancies are therefore required. The prognosis of the individual patient depends on the involvement of internal organs and the characterization of circulating autoantibodies. There are patients with rapidly developing subsets of dermatomyositis and those with slowly progressive disease. Therefore treatment has to be adjusted for the disease severity. Sometimes high dosages of corticosteroids in combination with immunosuppressive agents or immunoglobulins are required.

Adult↗
PubMed1982

C-reactive protein in childhood dermatomyositis.

Serum levels of C-reactive protein (CRP) were determined in 9 patients with childhood dermatomyositis. Four children were seen during clinical relapse and all had serum CRP levels less than 1 mg/l. In addition direct immunofluorescent staining of muscle biopsies from 4 patients showed no evidence of CRP deposition in muscle tissue. Such patients appear to be able to produce CRP in response to acute infections, and it is suggested therefore that the pathological process in childhood dermatomyositis may not induce a significant CRP response.

Adolescent↗
PubMed1990

Evans' syndrome associated with dermatomyositis.

Autoimmune haematological complications in dermatomyositis are very uncommon. This case report describes autoimmune haemolytic anaemia and thrombocytopenia in a patient with dermatomyositis and pulmonary fibrosis.

Aged↗
PubMed1992

Staphylococcal infections in childhood dermatomyositis--association with the development of calcinosis, raised IgE concentrations and granulocyte chemotactic defect.

There is a high incidence of staphylococcal infection in children with dermatomyositis, which is limited to those children who either already have or subsequently develop calcinosis. Of 15 children followed up for 3-10 years after diagnosis, all nine who developed calcinosis had infections with Staphylococcus aureus compared with none of six without calcinosis. Of these nine, the occurrence of staphylococcal infections before calcinosis was observed in four, suggested by history in two, and unclear in three children. Granulocyte chemotaxis to Staphylococcus aureus was more severely depressed in those children with calcinosis, whereas those without calcinosis did not differ significantly from controls. The chemotactic defect was due to a serum factor (patients' serum depressed control chemotaxis and control serum corrected the patients' chemotaxis). The nine children with calcinosis also had significantly higher serum IgE concentrations than non-atopic age matched controls; the six without calcinosis did not differ from controls. The increased IgE concentrations appeared to develop after staphylococcal infection and before calcinosis. Two of five patients with calcinosis had increased antistaphylococcal IgE antibodies; neither of the two patients without calcinosis had such increased antibodies. This suggests preceding immunological differences in patients with dermatomyositis who do and do not subsequently develop calcinosis, either increasing susceptibility to Staphylococcus aureus infection or potentially resulting from such infections.

Antibodies, Bacterial↗
PubMed1999

Detection of anti-ADAM 10 antibody in serum of a patient with pulmonary fibrosis associated with dermatomyositis.

OBJECTIVES: It has been suggested that the humoral immune system plays a part in the pathogenesis of pulmonary fibrosis. Although circulating autoantibodies to lung protein(s) have been suggested, few lung proteins have been characterised. The purpose of this study is to determine the antigen recognised by serum of a patient with pulmonary fibrosis associated with dermatomyositis. METHODS: To accomplish this, anti-small airway epithelial cell (SAEC) antibody in a patient's serum was evaluated using a western immunoblot. RESULTS: An autoantibody against SAEC was found, and the antigen had a molecular weight of 62 kDa. Using the patient's serum, clones from the normal lung cDNA library were screened and demonstrated that anti-SAEC antibody in the patient's serum was against ADAM (A disintegrin and metalloprotease) 10. CONCLUSION: This is the first report that demonstrates the existence of anti-ADAM 10 antibody in a patient with pulmonary fibrosis associated with dermatomyositis.

ADAM Proteins↗
PubMed2001

Difference in B cell activation between dermatomyositis and polymyositis: analysis of the expression of RP105 on peripheral blood B cells.

BACKGROUND: It has previously been shown that RP105, a new B cell surface protein, is lost in activated human B cells. OBJECTIVE: To investigate whether there is a difference in B cell activation between patients with dermatomyositis (DM) and those with polymyositis (PM) using RP105 as a marker. METHODS: The population of RP105 negative B cells (activated B cells) in the peripheral blood mononuclear cells of seven patients with dermatomyositis (DM) and 11 with polymyositis (PM) was analysed by flow cytometry. RESULTS: The percentage of RP105 negative B cells in the peripheral blood of patients with PM was low (5.8 (SD 2.4)%), similar to that of normal subjects. In contrast, all patients with DM showed increased RP105 negative B cell populations (33.0 (6.9)%). Bronchoalveolar lavage fluid from a patient with DM and active interstitial pneumonitis contained a large number of RP105 negative B cells. CONCLUSION: These findings suggest that the expansion of RP105 negative B cells is a hallmark of DM, and that B cell activation in DM may be pathogenetically different from that in PM.

Adult↗
PubMed1986

Pathogenesis of heart block in a fatal case of dermatomyositis.

Approximately 70% of patients with dermatomyositis have evidence of cardiac damage; in one third of cases this affects principally or solely the conduction tissue. In infants similar histological lesions have been associated with a maternal autoantibody, anti-Ro, that crosses the placenta and produces congenital heart block. Anti-Ro antibody was detected in a fatal case of dermatomyositis with lesions of the conduction system.

Atrioventricular Node↗
PubMed1990

Metastatic non-seminomatous germ cell tumour and dermatomyositis.

Twenty to thirty percent of cases of dermatomyositis in adults are associated with malignancy. We report a case in which dermatomyositis heralded the development of a testicular germ cell tumour. Germ cell testicular tumours are treatable and our case emphasizes the importance of measuring blood levels of beta-human chorionic gonadotrophin and alpha-fetoprotein and examination of the testes in young males with indications of possible malignancy.

Adult↗