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PubMed2002

[Case of dermatomyositis complicated with massive pleural effusion that preceded the myopathy].

A 36-year-old man was admitted to a hospital with complaints of fever, polyarthralgia and dyspnea. Erythema was observed on his face, extensor surface of the fingers and extremities, and a chest X-ray revealed massive bilateral pleural effusion. He had no sign of myopathy at this point. Pleural fluid was proved to be exudative and contained extremely high levels of hyaluronic acid. He was also complicated with interstitial pneumonitis and was given a pulse therapy with methyl prednisolone followed by daily administration of 55 mg prednisolone (PSL). Twenty days after the commencement of the therapy, pleural effusion decreased but muscle weakness gradually appeared, accompanied by elevation of myogenic enzymes. Myogenic changes on electromyogram, and irregularity of the muscle fibers with slight inflammatory cell infiltrates in a biopsy specimen were demonstrated. He was transferred to our hospital, and a diagnosis of dermatomyositis was made. Later, pleural effusion waxed and waned depending on the dosage of PSL, but no other causative disorder was demonstrated by extensive examinations. This case indicates that the pleuritis could be one of the vasculitic manifestations of dermatomyositis.

Adult↗

PubMed2004

[A case of amyopathic dermatomyositis with acute interstitial pneumonia (DAD pattern)].

A 61-year-old man was admitted to our hospital because of edematous erythema on his upper eyelids and dry cough. No subjective nor objective findings suggestive of skeletal muscle involvement, such as muscle weakness and elevated levels of aldolase and creatine phosphokinase were noted. Chest high-resolution computed tomography revealed a ground glass opacity and consolidation of his lower lung. Skin biopsy findings were compatible with dermatomyositis. Therefore, he was diagnosed as amyopathic dermatomyositis (ADM) with acute interstitial pneumonia and treatment with steroid pulse therapy was started. Since histological evaluation showed diffuse alveolar damage during the initial treatment, the treatment was changed into the combination therapy of prednisolone and cyclosporine. However, his acute interstitial pneumonia did not respond to this treatment and passed away by aggravation of a breathing state and concurrence of disseminated intravascular coagulation. Japanese patients with ADM have been shown to be more frequently associated with intractable acute interstitial pneumonia than Caucasian patients, suggesting that the racial difference influences the occurrence of acute interstitial pneumonia in ADM. Since autoantibodies specific for ADM have not been detected, we performed immunoprecipitation analysis using 35S methionine-labeled K562 cells to identify them. His sera immunoprecipitated a polypeptide of 140 kDa. The 140 kDa polypeptide might be one of autoantibodies specific for ADM with acute interstitial pneumonia, although future analysis using a larger number of patients with ADM will be required to confirm this result.

Dermatomyositis↗

PubMedDate not supplied

[Rare forms of panniculitis: subcutaneous sarcoidosis and panniculitis associated with dermatomyositis].

Panniculitides represent heterogenous group of disorders involving subcutaneous fat tissue, and are etiologically related to different causes, including systemic diseases. Two female patients having subcutaneous sarcoidosis and panniculitis associated with dermatomyositis are presented in this paper. The first patient, (38 years of age), was with the nodes on forearms and lower legs, which occurred one month after hypophysectomy for adrenocorticoscopic (ACTH)-secreting pituitary adenoma, and showed histopathologically confirmed epithelioid, noncaseating granulomas in the subcutaneous fat tissue. Laboratory and radiographic findings were normal, with the exception of positive rheumatoid factor and incompletely developed cysts in proximal phalanges of the hand. The second patient (56 years of age), was with subcutaneous nodes on the upper arm and the lateral chest wall, which were noticed several months after the diagnosis of dermatomyositis. Histopathological examination showed lobular pannicilitis with hyaline fibrosis and plasmocytic-lymphocytic infiltrate.

Adult↗

PubMed2006

Successful treatment with regimen of intravenous gamma globulin and cyclophosphamide for dermatomyositis accompanied by interstitial pneumonia, opportunistic infection and steroid psychosis.

BACKGROUND: A 47-year-old Japanese woman was suffering from dermatomyositis with progressive interstitial pneumonia, which was resistant to treatment with prednisolone (Pred) and cyclosporine A (CsA). Unfortunately, the opportunistic infection and steroid psychosis made therapeutic intervention using additional immunosuppressive drugs problematic. To overcome these difficulties, we created a regimen of intravenous gammaglobulin (IVIG) and cyclophosphamide (CPM) for the treatment of this patient. METHODS: For the simultaneous treatment, IVIG-CPM was added to Pred/CsA by means of infusion of IVIG on five consecutive days per month, followed by CPM infusion on the ninth day after the last IVIG administration. The treatment was repeated for four months. RESULTS: This regimen induced almost full remission without exacerbation of the opportunistic infection or mental disturbance. CONCLUSIONS: The outcome reported here suggests that the combination therapy of Pred/CsA and IVIG-CPM appears to be useful for the treatment of dermatomyositis with pulmonary, infectious and mental complications.

Cyclophosphamide↗

PubMed2007

Predicting factors of interstitial lung disease in dermatomyositis and polymyositis.

The aim of this study was to define the predicting factors and evaluate the prognosis of interstitial lung disease in dermatomyositis/polymyositis. For the period 1995-2005, we retrospectively reviewed the clinical information and laboratory data of 56 patients who were diagnosed as definite and probable dermatomyositis and polymyositis. Interstitial lung disease is common (41.9%) in these patients. Dyspnoea and cough were the two most common initial presentations. Anti-Jo1 antibody was more common in those with interstitial lung disease. Univariate and multivariate analyses identified primary idiopathic dermatomyositis subtype, cough and dyspnoea at onset to be the three independent clinical predicting factors of interstitial lung disease. High serum lactate dehydrogenase level (>400 U/l) was inversely associated with development of interstitial lung disease (OR 0.088, p=0.031). Serum lactate dehydrogenase level and presence of anti-Jo1 antibody can serve as laboratory indicators of lung complications. Patients with malignancy and older age at onset (more than 60 years) had poorer prognosis for dermatomyositis/polymyositis (p=0.047 and p=0.035, respectively). Interstitial lung disease did not affect the survival of dermatomyositis/polymyositis patients.

Adult↗

PubMed1979

Dermatomyositis associated with BCG vaccination.

Two young boys developed serious forms of dermatomyositis following BCG vaccination. Possibilities of a causal relationship between the disease and the vaccination are discussed. Extensive immunological tests, however, including in vitro stimulation of lymphocytes with PPD, gave no decisive evidence of abnormalities. It is concluded that in cases of dermatomyositis there is an absolute indication for a full anamnesis with regard to previous vaccination, to obtain clarification of the practical and theoretically important questions of a possible connection in this respect.

Adolescent↗

PubMed1999

Hyperimmunoglobulin E-recurrent infection syndrome in a patient with juvenile dermatomyositis.

A 13-year-old girl presented with multiple skin abscesses. She was diagnosed as having juvenile dermatomyositis (DM) at the age of 7 years. She had suffered from recurrent skin infections, atypical pruritic dermatitis and pneumonia since the age of 8 years. Bacteriologic and fungal cultures for skin abscesses and oral mucosa were positive S. aureus and C. albicans, respectively. Chemotactic defect in peripheral blood neutrophils was observed. The level of serum IgE was markedly elevated, and anti-S.aureus specific IgE was found. A diagnosis of hyperimmunoglobulin E-recurrent infection syndrome (HIE) was made and she was successfully treated with surgical drainage and antibiotics. To our knowledge, this is the first case report of HIE in a patient with juvenile dermatomyositis.

Adolescent↗

PubMed2002

Prevention of progression of interstitial lung lesions by early combination therapy with corticosteroids and cyclosporine/cyclophosphamide in two patients with amyopathic dermatomyositis.

Two patients with amyopathic dermatomyositis complicated by interstitial lung lesions were effectively treated with a combination of corticosteroids and cyclosporine and/or cyclophosphamide. A 48-year-old female patient was treated with pulse methylprednisolone and cyclosporine 2 months after onset of dermal symptoms. A 45-year-old male patient was treated with oral prednisolone and pulse cyclophosphamide 2 1/2 months after onset of dermal symptoms. Early evaluation of interstitial lung lesions and early extensive therapy may improve prognosis of interstitial lung lesions in patients with amyopathic dermatomyositis.

Adrenal Cortex Hormones↗

PubMed1976

Dermatomyositis and malignancy. A review of the literature.

Although there appears to be an increased incidence of malignancy among patients with dermatomyositis, demonstration of definitive statistical significance is precluded by the lack of large, controlled series. Patients with the two diseases tend to be older than the general dermatomyositis population and younger than those with cancer alone; and there is a preponderance of female patients. Tumors of the ovary and stomach are more frequently observed than in the general population, while colorectal malignancies are underrepresented. Most reported cases show development of the diseases within a year of one another, and, in some patients, the course of the myopathy follows that of the tumor. No definitive cause for the myopathy in these patients has been established.

Adolescent↗

PubMed1990

Dermatomyositis-like syndrome in X-linked hypogammaglobulinemia. Case-report and review of the literature.

A case of dermatomyositis-like syndrome is described in a 19-year-old man with a history of Bruton's hypogammaglobulinemia. Although the patient had central-nervous-system manifestations (seizures), no echovirus was isolated in the cerebrospinal fluid, in contrast to previously reported cases. Data for our case and the 15 cases previously reported in the literature are reviewed. HLA typing of our patient revealed the presence of HLA B8 and DR3, which seems to play a major role in juvenile dermatomyositis.

Adult↗

PubMed1998

[Clinical analysis of 11 cases of juvenile dermatomyositis and polymyositis].

Juvenile dermatomyositis (JDM) is characterized by microvasculopathy of the striated muscle, which indicates different etiology, clinical manifestation and prognosis from the adult-onset dermatomyositis. We experienced 10 cases of JDM and 1 case of juvenile polymyositis (JPM) in the recent 14 years, and analyzed clinical manifestation, laboratory findings, treatment anrognosis. The cases were 9 girls and 2 boys. The onset of the disease was 2 years of age in 2 patients, and 9 to 13 years of age in 9 patients. During the follow-up courses, no cases were dead or complicated with neoplasm. Skin rash was the most frequent manifestation at the onset, and facial erythema was common. Muscle weakness was observed only in 4 cases at the onset, and in all cases muscle enzymes including creatine kinase and aldolase were elevated. The clinical course was classified into three groups; monocyclic (5 cases), chronic and recurrent (4 cases), and fulminant (2 cases). Prognosis depended not on the degree of the elevated serum muscle enzymes, but on the initial therapy employed at the onset of the disease. Five cases including 2 cases of fulminant type were initially treated with methylprednisolon pulse therapy, and all of these had no recurrence. On the other hand, 6 cases were started the therapy with p.o. prednisolone. Four of them had frequent recurrences in accordance with tapering of prednisolone. These cases were effectively treated with the combination with immunosuppressants. In previous reports, JDM and JPM were reported to be a disorder which had relatively favorable prognosis. But we found that one third of the cases had chronic and recurrent courses. Methylprednisolone pulses as initial therapy may be effective in preventing the chronicity and recurrence of the disease.

Adolescent↗

PubMed1999

Epidermal nuclear CIq deposits in a patient with amyopathic dermatomyositis.

We report a case of amyopathic dermatomyositis, in which C1q deposits on the epidermal nucleus were immunohistologically found as well as Ig deposits at the dermoepidermal junction (DEJ). Direct immunofluorescence (IF) examination of infiltrated erythematous lesions revealed C1q deposits on the epidermal nucleus and fibrinogen at the DEJ, and examination of hyperkeratotic erythematous lesions showed linear deposits of Ig G and Ig A at the DEJ but not in the nuclei of epidermal cells. The authors discuss the direct in vivo interaction between nucleus and immunoreactants in dermatomyositis.

Aged↗

PubMed1999

Amyopathic dermatomyositis associated with transitional cell carcinoma of the bladder.

Transitional cell carcinoma of the bladder is the most common tumor of the urinary tract. However, it has only been reported twice in the literature to be associated with the paraneoplastic syndrome dermatomyositis. We report a case of amyopathic dermatomyositis in a patient whose painless gross hematuria was due to transitional cell carcinoma of the bladder as well as review this association.

Aged↗

PubMed1999

"Centripetal flagellate erythema": a cutaneous manifestation associated with dermatomyositis.

We describe 3 patients with dermatomyositis who presented with flagellate erythema. This cutaneous eruption is characterized by erythematous linear lesions on the trunk and proximal extremities. Histologic examination of this eruption in one of our cases revealed an interface dermatitis. Review of the literature and records of 183 patients with connective tissue diseases from our institution has shown that this peculiar eruption has been reported only in dermatomyositis. Because of the location of this eruption, we encourage the use of the term "centripetal flagellate erythema" to distinguish this entity from other linear eruptions seen in patients with connective tissue diseases.

Adult↗

PubMedDate not supplied

Autoimmune thrombocytopenic purpura and dermatomyositis associated with chronic hepatitis C. A therapeutic dilemma.

We recently observed a patient with chronic C hepatitis who developed first autoimmune thrombocytopenic purpura and subsequently dermatomyositis. While the association could be coincidental, it is possible that hepatitis C virus could have induced autoantibodies or circulating immune complexes which contributed to both. Although concerns are sometimes raised about the use of corticosteroids and immunosuppressive therapy to control symptoms in hepatitis C virus infected patients with rheumatic or autoimmune diseases, corticosteroid and immunosuppressive therapies are considered appropriate in cases of chronic C hepatitis with rapidly progressive autoimmune diseases. Our case illustrates the paradox that, despite the continuing presence of a viremic form of chronic C hepatitis, clinical symptoms improved with combined immunosuppressive therapy, without deterioration of the hepatitis. Our results could be the consequence of the association of immunosuppressive therapy intensive enough to control the autoimmune thrombocytopenic purpura and dermatomyositis with alpha-interferon whose antiviral capacity may have been able to prevent re-exacerbation of the hepatitis.

Aged↗

PubMed1999

[Successful combination chemotherapy with low-dose methotrexate and steroids for dermatomyositis complicated by interstitial pneumonitis].

A 44-year-old woman visited our hospital in August 1997 with complaints of general fatigue and low-grade fever. Initial examination disclosed slightly elevated muscle enzymes and inflammatory reactions. A diagnosis of dermatomyositis was made on the basis of findings of skin symptoms consistent with Gottron's signs and a slight decrease in muscular strength. Reticular shadows were observed in both lower lung fields on chest X-ray films, and examination of transbronchial lung biopsy specimens revealed interstitial pneumonitis. After 1 course of steroid pulse therapy, the patient was placed on a regimen of prednisolone (PSL) at 60 mg/day and cyclosporin (CsA) at 200 mg/day. Although her skin symptoms and interstitial pneumonitis temporarily subsided, aggravated interstitial pneumonitis and elevated blood LDH levels were observed as the steroid dose (PSL) was being tapered. After a second course of steroid pulse therapy, the PSL dose was increased to 50 mg/day and CsA was replaced by methotrexate (MTX), at 7.5 mg/week. This brought the patient's interstitial pneumonitis under control. Although our patient had dermatomyositis complicated by interstitial pneumonitis which belongs to the group of poor prognosis, she responded favorably to combination chemotherapy with low-dose MTX.

Adult↗

PubMedDate not supplied

[Lipedema of the leg associated with dermatomyositis].

Lipedema represents a form of lipodistrophy, which consists of abnormal accumulation of fat in subcutaneous tissue of the lower limbs with consecutive development of lymphostasis and lymphedema. The aim of this article was to review one clear case of lower limbs lipedema, of unusual occurrence and appearance, which was associated with dermatomyositis. A moderately manifested lipedema in 8 years old little girl was reported with its expressive segmental distribution to upper and lower legs, without significant increase in its size during last 10 years and without signs of lymphostasis. The hereditary influence was not confirmed. Histological examination of lipedematous tissue revealed significant presentation of immune component of the disease. According to the available literature, association between lipedema and dermatomyositis, lower limbs lipedema with segmental distribution as noticed above and its appearance as a consequence of corticosteroid therapy have not yet been published.

Adult↗

PubMed1999

Familial dermatomyositis.

Susceptibility, response to therapy, prognosis, and natural history of connective tissue diseases are variably linked to histocompatibility antigens (HLA), although the precise role that such genes play is still not clear. In juvenile dermatomyositis (JDMS) there are few reports of both concordance and discordance in identical twins and occurrence in more than one family member. To further document the familial occurrence of JDMS, we describe 2 sisters, one with classical JDMS and the other with amyopathic dermatomyositis. The 2 patients were shown to share the HLA-DQA1*0501 allele, known to be associated with susceptibility to JDMS.

Child↗