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PubMed1999

Dermatomyositis with elevated serum hyaluronate.

We report two Japanese patients with dermatomyositis whose serum hyaluronate levels were correlated with their disease activities. The diagnosis of one patient was primary idiopathic dermatomyositis, and that of the second patient was dermatomyositis associated with neoplasia. The serum hyaluronate level of the former patient was decreased by systemic corticosteroid therapy, and that of the latter patient decreased following the surgical resection of mammary carcinoma and subsequent chemotherapy. The disease activity of each patient was greatly improved after these therapies. The longitudinal measurement of serum hyaluronate concentration may be useful to estimate the disease activity of patients with dermatomyositis whose initial serum levels of hyaluronate are elevated.

Adult↗

PubMed1998

Crusted scabies of the scalp in dermatomyositis patients: three cases treated with oral ivermectin.

BACKGROUND: Cutaneous features in the scalp area among adult patients are rarely considered as a manifestation of scabies. METHODS: Three patients with clinical and laboratory data of dermatomyositis with scalp involvement (fulfill three or four of Bohan and Peter's criteria), of 4 years, 8 months, and 3 years duration, were seen at our department between 1995 and 1996. For relapses of ordinary scabies, they were treated repeatedly with local scabicide with temporary effect. After a symptom-free period during the treatment of dermatomyositis with corticosteroids and azathioprine, they developed diffuse redness with scales and crusts on the scalp areas. Light microscopy examination of material taken from these crusts showed an abundance of live mites. RESULTS: All patients were successfully cured of scabies with a twice oral dose of 200 microg/kg ivermectin within 8 days. CONCLUSIONS: Our patients with crusted scabies of the scalp and dermatomyositis prompted us to change our standard diagnostic and therapeutic regimens. Fascinating features included mimicry of scabies in patients with dermatomyositis, location of parasites on the scalp, suppressed cell-mediated immunity and successful cure of mange by ivermectin.

Adult↗

PubMed2002

Cutaneous involvement of dermatomyositis can respond to Dapsone therapy.

BACKGROUND: Dapsone (4,4-diaminodiphenylsulfone) is a sulfone antibiotic used in the treatment of leprosy, but dermatologists more commonly utilize its anti-inflammatory properties particularly directed against leukocytes to treat various bullous disorders, erythema nodosum, pyoderma gangrenosum, Sweet's syndrome, cutaneous vasculitis, and cutaneous forms of lupus erythematosus. The cutaneous manifestations of dermatomyositis are often resistant to antimalarial and immunosuppressive therapies. METHODS: Two patients with cutaneous dermatomyositis unresponsive to combination therapy with prednisone, hydroxychloroquine, quinacrine, and immunosuppressive medications had Dapsone added to their therapy. RESULTS: Both patients showed rapid improvement with the addition of Dapsone. Each had an exacerbation of their cutaneous dermatomyositis on Dapsone with withdrawal and subsequent improvement when the Dapsone was reinstituted. CONCLUSIONS: Dapsone therapy for cutaneous dermatomyositis may have a wider role in treatment for these patients refractory to prednisone and antimalarial therapy.

Adult↗

PubMed2003

Periorbital edema as the presenting sign of dermatomyositis.

BACKGROUND: Periorbital edema can occur in dermatomyositis, which is characterized by symmetric macular erythema, Gottron's papules, Gottron's sign, periungual telangiectasia, heliotrope rash, and poikilodermatous macules on the shoulders, arms, or upper back (shawl sign). CASE REPORT: We report the case of an 81-year-old man with dramatic periorbital edema. It was not until he was hospitalized with dysphagia 6 months after developing the edema that the diagnosis of dermatomyositis was considered. RESULTS: Laboratory tests, skin biopsy, and electromyography resulted in a diagnosis of dermatomyositis. CONCLUSIONS: Periorbital edema may appear as the presenting cutaneous manifestation of dermatomyositis.

Aged↗

PubMed2000

An unusual presentation of dermatomyositis: the type Wong variant revisited.

We describe a 53-year-old white woman with dermatomyositis (DM) who had additional clinical findings of pityriasis rubra pilaris (type Wong dermatomyositis) with histopathologic features of both pityriasis rubra pilaris (PRP) and porokeratosis. Type Wong dermatomyositis was originally described in 11 patients by Wong in 1969 and has been reported in 5 additional patients. This is a rarely described phenomenon in which patients with DM develop cutaneous hyperkeratotic lesions that resemble PRP and histologically show follicular hyperkeratosis and hair follicle destruction. Arrector pilorum muscles also show degenerative findings and myositis. We believe that this is the first reported case of a patient with type Wong DM who also has clinical and histologic features suggestive of porokeratosis. This is important because of the association of adult-onset dermatomyositis with internal malignancy and the well-documented association of porokeratosis with immunosuppression. These clinical and histologic findings serve as markers for malignancy in patients with DM. These patients warrant a complete review of systems and investigation for age-appropriate neoplasms as well as close long-term follow-up by dermatologists to ensure that these cutaneous eruptions are not overlooked.

Dermatomyositis↗

PubMed2005

The effects of meditation and visual imagery on an immune system disorder: dermatomyositis.

OBJECTIVE: To analyze the relationship between a patient's "spontaneous recovery" from dermatomyositis and her practice of transcendental meditation and visual imagery without confounding effects of conventional therapies. DESIGN: Study of time-varying relationships between (1) measures of arm strength and skin condition (rash and pain) and (2) mind-body interventions-controlling for psychologic stress-in a patient with dermatomyositis, using regression analysis to determine half-lives of treatments and stress. SETTING: Institutional referral center. INTERVENTION: Transcendental meditation and visual imagery (no drugs). OUTCOME MEASURES: Daily measurements of arm strength and skin condition over 294 days. Events producing psychologic stress were also rated using a numerical scale. RESULTS: The patient recovered, which is a low-probability event without conventional therapy. Regression analysis of time dependence between measures of arm strength, rash, and pain and application of mind-body treatments revealed statistically significant relationships for both meditation (p values 0.02 to 0.001) and visual imagery (p values 0.02 to 0.002). Stress had a significant negative impact on skin symptoms but not arm strength. Beneficial effects of meditation had half-lives of 48-59 days for skin condition and no detectable decay for arm strength. Benefits of visual imagery were more transient (half-lives 4-18 days). The effects of stress had half-lives of only 1-3 days. CONCLUSIONS: The results demonstrate a statistically significant relationship between mind-body therapies and the patient's recovery from dermatomyositis, possibly mediated by influences on the humoral immune system. A key factor in the recovery was the slower decay rate of meditation and visual imagery compared to stress. As dermatomyositis is a humorally mediated immune microvasculopathy, the benefits of meditation and imagery in our patient comport with a growing body of evidence showing that these techniques influence immune system function.

Attitude to Health↗

PubMed2005

Rituximab in the treatment of refractory dermatomyositis.

Dermatomyositis is an inflammatory myopathy characterized by muscle weakness and inflammation. In contrast to polymyositis and inclusion body myositis, humoral immune mechanisms appear to contribute to the pathogenesis of dermatomyositis. A 56-year-old man with dermatomyositis resistant to conventional therapies was treated with 6 weekly infusions of the anti-CD-20 monoclonal antibody, rituximab, at a dosage of 100 mg/m in addition to other agents. The patient demonstrated a remarkable clinical response as indicated by an increase in muscle strength and a decline in creatine kinase enzymes. B-cell depletion therapy with rituximab used alone or in combination with other immunosuppressive therapies may be a viable option in patients with dermatomyositis as well as other autoimmune diseases refractory to current therapies.

Antibodies, Monoclonal↗

PubMed1993

Dermatomyositis, carcinoma of colon and meningioma in the same patient.

Dermatomyositis and carcinoma of colon were diagnosed in a 66-year-old woman. Meticulous physical examination excluded further systemic or cutaneous involvement. The musculocutaneous disorders responded well to daily oral corticosteroid, and the malignant tumor was totally removed surgically. After a seven-year follow-up of actual dermatomyositis controlled by maintenance doses of prednisone ranging from 5 to 15 mg daily, the patient developed a meningioma. Current concepts and data regarding various aspects of the combination between dermatomyositis and tumors are discussed. To our knowledge, this is the first reported case of meningioma associated with dermatomyositis.

Adenocarcinoma↗

PubMed2005

Relapse of dermatomyositis after 10 years in remission following curative surgical treatment of lung cancer.

A 55-year-old woman with dermatomyositis and small cell lung cancer was successfully treated with surgery followed by combination chemotherapy in 1987. She had been in remission without further immunosuppressive therapy for 10 years. However, myositis with cutaneous manifestations specific for dermatomyositis relapsed when the patient was 69 years old. Intensive examinations revealed no neoplasm, and she responded to a moderate dose of systemic corticosteroids. This case suggests a long-lasting autoimmune abnormality in dermatomyositis and that a neoplasm is an important factor in eliciting an occult dermatomyositis.

Administration, Oral↗

PubMed1995

Dermatomyositis treated with high-dose intravenous immunoglobulins and associated with panniculitis.

Polymyositis and dermatomyositis are idiopathic inflammatory myopathies characterized by subacute symmetrical weakness of proximal limb and trunk muscles. Dermatomyositis is distinguished from polymyositis by the presence of rash. We describe an adult patient with treatment-resistant childhood-type dermatomyositis who made a good response to high dose intravenous immunoglobulins. Additionally, there was evidence of panniculitis which is an unusual histopathological finding in dermatomyositis.

Adult↗

PubMed1980

Cutaneous immunofluorescence in dermatomyositis.

Seven patients with dermatomyositis who displayed severe skin and muscle disease, and in whom coexistent systemic lupus erythematosus (LE) was excluded, were evaluated by direct immunofluorescent biopsies of skin lesions. Specimens from six showed deposits of immunoglobulins and complement in small to moderate amounts at the dermal-epidermal junction zone. These deposits were usually in the form of focal granular accumulations which lacked continuity and were not well developed as are those seen in LE, or fluorescent subepidermal hyaline bodies. One patient, however, had a more well developed band of immunoglobulins at the dermal-epidermal junction. All normal skin specimens in these patients were negative by immunofluorescence. These findings were helpful in clarifying differences in cutaneous immunofluorescence between dermatomyositis and LE, indicating that dermatomyositis specimens can usually be differentiated from those of LE patients, but demonstrating the possibility that confusion might rarely occur in interpreting a lesional immunofluorescent biopsy in dermatomyositis.

Adult↗

PubMed1997

Calcinosis cutis presenting years before other clinical manifestations of juvenile dermatomyositis: report of two cases.

Calcinosis cutis in dermatomyositis is dystrophic calcification appearing late in the course of the disease. Two cases are reported here of calcinosis cutis that presented years before other clinical manifestations of juvenile dermatomyositis. The first case was a 14-year-old Thai girl who had asymptomatic subcutaneous nodules that spontaneously ruptured, exuding a chalky discharge and healing with an atrophic scar 8 years before the onset of other clinical manifestations of juvenile dermatomyositis; that is, Gottron's papules, proximal muscle weakness grade IV/V with atrophy, slightly elevated serum creatinine phosphokinase level and an abnormal electromyogram compatible with myopathy. The second case was a 15-year-old Thai boy who had calcinosis cutis 3 years before the onset of other clinical manifestations of juvenile dermatomyositis, and the calcinosis cutis was so severe that it interfered with the movement of his extremities. Both cases responded well to aluminium hydroxide therapy.

Adolescent↗

PubMed2004

Interstitial lung disease, a common manifestation of newly diagnosed polymyositis and dermatomyositis.

OBJECTIVES: To estimate the prevalence and predictors of interstitial lung disease in newly diagnosed polymyositis and dermatomyositis. METHODS: A prospective study in which consecutive patients with newly diagnosed poly- and dermatomyositis, regardless of clinical symptoms of pulmonary disease, were investigated with chest x ray, high resolution computed tomography (HRCT), pulmonary function tests, and biochemical and autoantibody analysis. Patients with inclusion body myositis, malignancy, other defined inflammatory connective tissue diseases (CTDs), or antibody profile indicating other CTDs were excluded. RESULTS: Between March 1998 and September 2000, 26 new cases of poly- or dermatomyositis were diagnosed; 17 of those patients were included in the study. Interstitial lung disease (ILD), defined as radiological signs on chest x ray examination/HRCT or restrictive ventilatory defect, were found in 11 (65%) patients and were more common in men than in women. Arthritis and occurrence of anti-Jo-1 antibodies were found more often in patients with ILD than in those without. There was no statistically significant association between respiratory symptoms, other serological or laboratory variables and ILD. CONCLUSIONS: ILD is a common early manifestation in patients with poly- and dermatomyositis and is not always related to clinical symptoms. Chest x ray examination, HRCT, pulmonary function tests, and analysis of anti-Jo-1 antibodies should be included in the initial investigation of patients with myositis regardless of respiratory symptoms.

Adult↗

PubMed2006

Possible role for tumour necrosis factor inhibitors in the treatment of resistant dermatomyositis and polymyositis: a retrospective study of eight patients.

OBJECTIVE: To understand the use of tumour necrosis factor (TNF)alpha inhibitors in refractory dermatomyositis and polymyositis in an academic centre. METHODS: A retrospective study of eight patients with dermatomyositis or polymyositis refractory to corticosteroids and immunosuppressives who were treated with TNF inhibitors between 1998 and 2004. RESULTS: 8 patients with dermatomyositis or polymyositis who were treated with TNF inhibitors as adjunct treatment were identified. The mean (SD) duration of disease before initiation of TNF inhibitors was 8.5 (4.4) years. The patients failed to respond to treatment with corticosteroids (oral and intravenous); intravenous immunoglobulin and immunosuppressants (methotrexate, azathioprine, mycophenolate mofetil and leflunomide); 4.5 (1.4) immunosuppressants had been used before TNF treatment. Six patients were treated with etanercept alone, one with infliximab and one sequentially with both agents. Of the eight patients, six showed a favourable response with improved motor strength and decreased fatigue after 15.2 (6.5) months. Two of the patients did not respond after 4 (1.4) months and TNF inhibitors were discontinued. Responders showed a 54.4% (27.7%) decrease in serum concentration of creatine kinase, which was grossly abnormal (4463.5 (4036.4) U/l). Non-responders had similar reductions in creatine kinase concentration (56.1% (20.4%)), but their pre-treatment concentrations were in the normal range (118.5 (19.1) U/l). CONCLUSION: Anti-TNF agents may be useful in some patients with refractory dermatomyositis or polymyositis.

Adult↗

PubMed1993

Polymyositis and dermatomyositis: no persistence of enterovirus or encephalomyocarditis virus RNA in muscle.

OBJECTIVES: A persistent infection of enteroviruses and cardioviruses has been implicated in polymyositis and dermatomyositis, but conventional hybridisation studies of the presence of enterovirus RNA and encephalomyocarditis (EMC) virus RNA in affected muscle have yielded conflicting results. To investigate further the possibility of viral persistence, the presence of viral RNA in muscle from patients with adult onset polymyositis and dermatomyositis was investigated using a polymerase chain reaction (PCR) technique. METHODS: Muscle tissue was obtained from 10 patients with polymyositis and five patients with dermatomyositis, all with adult onset active disease. A PCR was performed using primers with high specificity for enterovirus and EMC virus RNA, followed by Southern blot hybridisation with an oligonucleotide probe directed against the internal portion of the amplified product. A PCR directed against the Abelson tyrosine kinase mRNA served as an internal control for the presence and quality of RNA. RESULTS: A specific amplification for enterovirus or for EMC virus could not be seen in any of the muscle biopsy samples, despite a sensitivity of about 30 plaque forming units for enterovirus and of 100 plaque forming units for EMC virus. Southern blot hybridisation confirmed these results in that positive controls hybridised with the oligonucleotide probe, but no signal was obtained with the muscle specimens. CONCLUSION: A sensitive and specific PCR technique showed no evidence of the presence of enterovirus or EMC virus RNA in muscle samples from patients with polymyositis or dermatomyositis. These data do not support the proposal that viral RNA persistence plays a part in these idiopathic inflammatory myopathies.

Adult↗

PubMed1995

Chronic pneumomediastinum and subcutaneous emphysema: association with dermatomyositis.

We describe a 41-year-old patient with adult-onset dermatomyositis who developed persistent pneumomediastinum and severe subcutaneous emphysema due to end-stage interstitial lung disease. The diagnosis of dermatomyositis was based on proximal muscle weakness, electromyographic findings of inflammatory myopathy, and positive findings on muscle biopsy. Low levels of creatine kinase elevation were found at the time of diagnosis (a form of dermatomyositis which has been associated with a poor prognosis). The patient had no signs of cutaneous vasculitis. Despite treatment with prednisone and azathioprine, she died of intercurrent gram-negative sepsis 15 months after the diagnosis of dermatomyositis.

Adult↗

PubMed1997

A case of dermatomyositis complicated by thrombotic thrombocytopenic purpura.

A 60-year-old man with dermatomyositis was admitted to our hospital because of dyspnea and hypertension. He had high fever and convulsive seizures after admission. Laboratory examinations showed hemolytic anemia, thrombocytopenia, and renal failure. A clinical diagnosis of thrombotic thrombocytopenic purpura (TTP) was made. He failed to respond to plasma exchange therapy, pulse therapy with methylprednisolone, high-dose gamma-globulin therapy, and antiplatelet therapies with ticlopidine, dipyridamole and a prostacyclin analog of beraprost sodium. He died on his 17th day in hospital. Autopsy examination revealed widespread microthrombi in his kidneys, lungs, spleen, and intestine. Only seven cases of dermatomyositis or polymyositis complicated by TTP have been cited in the literature. TTP was fatal in 6 of these 7 cases. Early diagnosis and prompt treatment may improve the outcome of TTP patients with dermatomyositis. Dermatologists should keep in mind that TTP occasionally arises as a serious complication of dermatomyositis.

Anti-Inflammatory Agents↗

PubMed1995

Dermatomyositis with follicular hyperkeratosis.

We report 2 cases of dermatomyositis with follicular hyperkeratosis (FHK) in children. They occurred in a 10-year-old Vietnamese girl and a 9-year-old Caucasian boy. The girl's FHK disappeared after 2 months of treatment. The boy presented, 15 months after the onset of his dermatomyositis, with a generalised FHK which lasted for 6 months. FHK can appear before, during or after dermatomyositis. It is more often generalized but can be localised. Erector pili myositis and ostial hyperkeratosis may be the explanation. The prognostic value of FHK in dermatomyositis is unknown. This manifestation, initially considered to be more frequent in the Far East, is not as rare in Western countries as the few reported cases suggest.

Child↗