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PubMed2000

[Dermatomyositis: clinical study of 34 patients].

Clinical data of 34 patients with DM, who have been treated during the years 1971 and 1998 were evaluated. 79% of the patients (27 patients) were female, 21% of them (7 patients) were male. 59% of the patients (20 pts) were between the ages of 41 and 50 years. The characteristic heliotrop rash were observed in 26 patients, Gottron's papules in 20 patients, poikiloderma in 2 patients, calcification, ulcers, Raynaud syndrome in 1 patient. 3 of the 34 patients presented with strongly itching erythematopapulosus symptoms, most prominently on the scalp. Cardiac involvement were present in 10 patients (29%), lung involvement in 8 patients (23%), gastrointestinal complaints in 11 patients, dysphagia, dysphonia in 4 patients, joint pain in 5 patients. Overlap syndrome--scleroderma-dermatomyositis, SLE-dermatomyositis--was present in 2 patients. 9 of the 34 patients (26%) suffered from malignant tumours: gastric, breast, lung, epipharynx carcinoma, malignant melanoma. 13 of the 34 patients have been treated with corticosteroids together with immunosuppressor agents, in most cases azathioprin was administered. Cyclosporin was given in 7 cases, chloroquine in 2 cases.

Adult↗
PubMed2000

Intravenous immunoglobulin therapy for juvenile dermatomyositis: efficacy and safety.

OBJECTIVE: To assess the efficacy of intravenous immunoglobulin (IVIG) for the treatment of juvenile dermatomyositis (JDM) in patients who were unresponsive to corticosteroids (steroid resistant or steroid dependent) or showed unacceptable toxicity. METHODS: A retrospective chart review of the course of all patients with JDM treated with IVIG who attended the Dermatomyositis Clinic at The Hospital for Sick Children, Toronto, Canada, from August 1986 to December 1996. RESULTS: Eighteen patients with JDM were treated with IVIG. Ten patients were taking additional 2nd line treatments, methotrexate, azathioprine, cyclosporine, and cyclophosphamide. The main indication for starting IVIG was the failure of steroid therapy to induce remission of JDM. Twelve patients showed clinical improvement with IVIG. In these patients, the corticosteroid dose was reduced by > 50% for > 3 months without clinical or biochemical flare. Nine of these 12 patients had IVIG alone as a 2nd line agent, whereas 3 patients were treated with additional agents. Six patients remained steroid dependent; they subsequently required multiple agents to induce remission of JDM. CONCLUSION: Most steroid dependent or steroid resistant patients in our clinic were able to markedly reduce their dose of corticosteroid with the addition of IVIG. Given the retrospective nature of our data and the fact that multiple agents were sometimes used together, it will be important to confirm these findings in a controlled trial.

Antirheumatic Agents↗
PubMed2000

[Therapeutic efficacy of cyclosporin A in four cases of amyopathic dermatomyositis with rapidly progressive interstitial pneumonia].

OBJECTIVES: We investigated the clinical features of amyopathic dermatomyositis (ADM) with rapidly progressive interstitial pneumonia (IP), and the effectiveness of cyclosporin A (CsA) for IP. METHODS: In our four patients who were accorded with the notion of amyopathic dermatomyositis proposed by Euwer and treated with CsA for IP, and eight reported patients for whom we could obtain details of the clinical course, we investigated the relationship between the clinical features of them at the start of CsA administration and the clinical effectiveness of CsA. RESULTS: When we investigated the effectiveness of CsA in our four patients and eight reported patients, we could judge that effective cases were four and ineffective cases were eight. We compared the PaO2 at the start of CsA administration between effective and ineffective cases, the average PaO2 at the start of CsA administration was 65.7 mmHg in four effective cases, while it was 51.0 mmHg in eight ineffective cases. There was a statistically significant difference between the groups. CONCLUSIONS: Because our results suggested that many ineffective cases were treated with CsA after they had developed severe respiratory failure, we think that CsA should be given to ADM patients with rapidly progressive IP before the progression of respiratory failure.

Adult↗
PubMedDate not supplied

[Classic/amyopathic dermatomyositis].

Dermatomyositis (DM) is an inflammatory myopathy characterized by a typical cutaneous rash and proximal weakness. DM results from endothelium deposition of the complement membranolytic attack complex C5b-9, followed by inflammation. It is thus possible to have cases of DM with the typical rash, but with no associated myositis--amiopathic DM. DM represents a higher risk of association with malignancy and sometimes behaves as a paraneoplastic syndrome. As DM can be the alarm sign, once diagnosed, exclusion of malignancy should be done. In this article we also discuss the treatment and prognosis of dermatomyositis, according to the literature.

Adolescent↗
PubMedDate not supplied

Skin metastasis of breast cancer clinically undistinguished from amyopathic dermatomyositis.

We report a 65-year-old woman who consulted us on May 25, 1998, showing pruritic, partially flagellate erythema on the back and upper extremities, livedo lesions with erythema on the loins, and erythematous papules on the dorsal finger joints for 2 months. Histopathological findings of erythema on the back showed mononuclear cell infiltration around capillaries and marked edema in the dermis. Laboratory data were within normal range except for positive anti-nuclear antibody. She had undergone total left mastectomy on June 2, 1997 for breast cancer. Supraclavicular lymph node metastasis was found at the beginning of May, 1998. A diagnosis of amyopathic dermatomyositis associated with breast cancer was made. Erythema with itching gradually subsided from the end of August, 1998. Treatment with radiation and chemotherapy reduced lymph node swelling, but complete remission was not obtained. Erythema similar to the previous lesion but without itching re-appeared on the back from January, 2000. Histological findings of erythema showed many carcinoma cells similar to the primary lesion of left breast cancer in the whole dermis. A diagnosis of skin metastasis of breast cancer was made. These findings suggest that skin metastasis should be taken into account for patients with erythema on the trunk similar to dermatomyositis.

Adenocarcinoma↗
PubMed2001

[A case of Sjögren's syndrome with dermatomyositis who died of rapidly progressive interstitial pneumonia].

We report a case of 55 year-old woman with six year history of Sjögren's syndrome developed fatal rapidly progressive interstitial pneumonia. She had been well until February 1999. She developed swelling and erythematous lesions in the cheek and hands in spring 1999. She was admitted to our hospital for investigations of skin lesions in May 1999. Physical examination on admission revealed small hemorrhagic lesions in the nailfold. Serum CK level was slightly elevated. Electromyogram and MRI suggested mild myositis in the proximal upper extremities. She was suspected to have dermatomyositis along with Sjögren's syndrome. Prednisolone 10 mg/day had been given for her skin problems since March 1999. Suddenly, dyspnea on exertion was appeared on 34th day of admission. Chest X-ray film showed an acute worsening of interstitial pneumonia. Methylprednisolone pulse therapy (1000 mg for 3 days) and cyclophosphamide pulse therapy (500 mg for a day) were started, and she was subsequently treated with 60 mg/day of prednisolone and 250 mg/day of Cyclosporin A. However, interstitial pneumonia did not respond to the treatment, and pneumomediastinum and pneumothorax have developed. She died of respiratory failure on 55th day. We consider that most likely explanation for fatal interstitial pneumonia is concomitantly occurred dermatomyositis.

Cyclophosphamide↗
PubMed1975

[Cardiovascular pathology in dermatomyositis].

Among 110 patients with dermatomyositis cardiovascular pathology was found in 77, and only in 37 of them it was attributable to the underlying pathology. In all of these patients myocardial lesions were found, that manifested themselves most frequently by non-specific ECG changes. Cardiac insufficiency and arrhythmias were revealed less frequently, mainly during exacerbations of the pathological process. Only in one patient endocarditis was diagnosed, and in two others elements of chronic cor pulmonale due to pneumonitis. In 18 patients signs of moderate myocardial dystrophy developed after a long-term therapy with corticosteroid hormones. A study of the central haemodynamics in 20 patients with dermatomyositis revealed a tendency towards a hyperkinetic state of the circulation in those patients who had the most sereve muscle syndrome. Angioscopy of the bulbar conjunctiva revealed the changes that persisted even during the remission period, achieved by means of active steroid therapy.

Adolescent↗
PubMed2001

Pulmonary nocardial infection and pseudomonas infection of the tongue in a patient with dermatomyositis.

Opportunistic infections in immunocompromised patients are common. We report the case of a 63 year old female patient with dermatomyositis who while on oral steroids developed nocardial infection of the lung and pseudomonas infection of the tongue simultaneously. Nocardial infections are not very commonly seen in patients with dermatomyositis. Pseudomonas infection of the tongue is a rarity. We report this case for its rarity as regards the type and site of infections and review the relevant literature.

Dermatomyositis↗
PubMed2001

[A patient who developed dermatomyositis during the 1st trimester of gestation and improved after abortion].

This patient was a 30-year-old woman who was in the 8th week of her first pregnancy with three embryos. She developed fever, myalgia and weakness of the proximal muscles, and erythema of the face, dorsal aspects of elbows, and knees. Routine blood examinations showed elevated serum CK. Immunologically, an anti-Jo-1 antibody was positive. Skin biopsy revealed mucinosis and edema in the superficial layer of the corium, and liquid alternation in the basal layer of the epidermis. From these findings, this patient was diagnosed as having dermatomyositis. She was placed on oral prednisolone (80 mg daily), but her clinical symptoms did not improve and all fetuses died by the 11th week of gestation. Then she underwent dilation and curettage and after this operation her disease rapidly subsided. It seemed that fetuses were causatively related to the development of dermatomyositis possibly by changing maternal immune condition. There were six reported cases with dermatomyositis/polymyositis who developed during the first trimester of gestation. Four of these 6 patients were treated with oral steroid; however, only one patient ended in normal delivery. More aggressive therapy, other than corticosteroid, may be required to improve fetal prognosis.

Abortion, Spontaneous↗
PubMed2002

[Bronchiolitis obliterans organizing pneumonia associated with dermatomyositis and antiphospholipid syndrome].

Dermatomyositis and antiphospholipid syndrome are rare diseases that can be associated pulmonary damage. We report a case of anti-phospholipid syndrome that presented with bilateral pulmonary embolism and subsequently developed pulmonary infiltrates in association with the characteristic muscular and cutaneous features of dermatomyositis. Histological examination of trans-bronchial biopsies revealed bronchiolitis obliterans organizing pneumonia.

Antiphospholipid Syndrome↗
PubMed2002

[A case of dermatomyositis with significantly high level of KL-6 associated with ovarian cancer].

The patient was a 58 year-old woman. Starting about seven months before the initial examination, the patient began to experience a general malaise, coupled with reddish purple scaling eruption on the MP joints, knuckles, elbows, and knees, and dark purplish erythema on both upper eyelids. About two months before the initial examination, the patient visited a local medical doctor because she began to experience arthralgia and muscular weakness. Based on the condition of her weakening proximal muscles and increased levels of myogenic enzymes, the patient was diagnosed as having Dermatomyositis. Oral administration of PSL (25 mg a day) was initiated, and the patient was referred to our department to undergo thorough examination. Although deterioration in muscular strength and elevation of myogenic enzyme levels were mild during the initial examination, the level of KL-6 was significantly elevated to 2600 U/ml. Numerous blood gas analyses and chest CT did not reveal any exacerbation of interstitial pneumonia. Various tests were performed to determine whether or not there was any malignancy, and although a small amount of ascites fluid was detected, diagnostic imaging revealed no clear abnormalities. Since cytological diagnosis of the ascites fluid was class V, an exploratory laparotomy was performed, and ovarian cancer complicated by peritoneal metastasis was discovered. As a result, the patient was diagnosed as having dermatomyositis accompanied by ovarian cancer. The level of KL-6 in the ascites fluid was significantly high at 10,900 U/ml, and immunohistological staining using anti-KL-6 antibodies confirmed the presence of KL-6 in the ovarian tumor, thus suggesting that KL-6 was produced by the ovarian cancer.

Antigens↗
PubMed2002

Outcome in juvenile dermatomyositis.

The clinical features, outcome and complications of juvenile dermatomyositis were studied in a tertiary care hospital by retrospective analysis of case records. Nineteen patients were treated over an 11-year period. Median age at diagnosis was 12 years (2.5-16 years). Median duration of disease prior to diagnosis was 12 months (2-96 months). Proximal muscle weakness was seen in all 19 cases, neck muscle weakness in 14, pharyngeal muscle involvement in 5 and respiratory muscle involvement in 3 cases. Heliotrope rash was seen in 9 and Gottrons rash in 8 patients. Myocarditis and GI bleed were seen in 1 each while interstitial lung disease was seen in 2 patients. All except one patient received prednisolone. Methotrexate was used in 13 and azathioprine in 3 patients. Eight patients are in complete remission (CR), 8 partial remission and 2 patients had no response. Complications were calcinosis in 5, contractures in 2, TB in 4 and pyogenic infections in 4 patients. Juvenile dermatomyositis needs to be recognised early and treated aggressively to improve outcome.

Adolescent↗
PubMedDate not supplied

Porphyria cutanea tarda, dermatomyositis and non-Hodgkin lymphoma in virus C infection.

Virus C infection has been associated with a broad spectrum of extrahepatic diseases such as essential mixed cryoglobulinemia, membranous glomerulonephritis, vasculitis, rheumatoid arthritis and lupus erythematosus. The etiologic role of virus C has also been observed in some neoplasms such as non-Hodgkin's lymphoma and the monoclonal gammapathies. Many studies also support the link between this virus and porphyria cutanea tarda (PCT). Isolated cases suggest a relationship with dermatomyositis. Herein, we report the coexistence of PCT, non-Hodgkin's lymphoma and dermatomyositis in the same patient affected with virus C infection which has never previously been described.

Antineoplastic Combined Chemotherapy Protocols↗
PubMed2003

[Multi-center analysis of calcinosis in children with juvenile dermatomyositis].

OBJECTIVES: To reveal the frequency and the clinical characteristics of dystrophic calcification that occurs in children with juvenile dermatomyositis, multi-center analysis was constructed. METHOD: Fifty children with JDM were enrolled, and 14 of them (28.0%) were complicated with calcinosis. Clinical symptoms and laboratory tests at onset, initial therapy and disease course were compared in children with and without calcinosis. RESULTS: The mean age of the onset of calcinosis was 4.78 +/- 3.33 years, and it was younger than those of children without calcinosis (8.66 +/- 3.85 years) (P = 0.0017). No differences of clinical manifestation except Gower's sign were observed. The frequency of positive anti-nuclear antibody was 7.1% in children with calcinosis and 52.9% without calcinosis (P = 0.0112). The initial therapy of methylprednisolon pulses gave no effects on prognosis of calcium deposition. The calcinosis appeared in 1.56 +/- 1.91 year after the onset of the disease. The various types of calcium deposition including large tumorous clumps, subcutaneous plaques or nodules, sheet-type calcification were deserved. They appeared over knee joints (64.3%), elbow joint (64.3%), and hip processes (50.0%). Calcinosis affecting the subcutaneous tissues frequently resulted in painful superficial ulceration of the overlying skin (42.9%), local infection (50.0%), and limitation of joint movement (14.3%). Although aluminum phosphate was effective in 2 children among 7, no other effective treatment was recommended. In 5 cases, surgical removal of tumorous clumps was operated. Thus, juvenile dermatomyositis is frequently complicated with calcinosis. This type of calcinosis was found to be unlikely to resolve completely, and resulted in severe disability in children.

Age of Onset↗
PubMed2003

[Appearance of numerous lobulated fibers after a protracted course of 18 years in a case of dermatomyositis].

A woman aged 47 first noticed weakness in her proximal muscles of all four limbs at the age of 29. After the initial investigation at our hospital, she was diagnosed as having dermatomyositis associated with primary biliary cirrhosis. In the following 18 years, she was kept on steroids. Although she responded to this treatment, weakness progressed slowly necessitating re-investigation including a second muscle biopsy. It was remarkable that in her first muscle biopsy, no lobulated fiber had been, while they were numerous in the second one. As a result of these studies, possibilities of limb-girdle muscle dystrophy (LGMD) and other myopathies remained but were ultimately ruled out after immunohistochemical/and genetic studies, and the original diagnosis was confirmed. Her weakness responded to an increased dose of steroids. Lobulated fibers were originally described in LGMD, and were reported also in the facioscapulohumeral muscular dytrophy and other hereditary myopathies. They have been rarely described in the inflammatory myopathies. Based on the observation of the present case, we concluded that lobulated fibers have little disease specificity and can newly appear in the course of any chronic myopathies including acquired myopathies like dermatomyositis.

Anti-Inflammatory Agents↗
PubMed1992

[Dermatomyositis associated with two neoplasms].

A case of dermatomyositis in a patient with two different malignancies is reported. Skin lesions cleared only following exeresis of the second tumor. This case provides the opportunity for a discussion of the main theories put forward to explain relationships between dermatomyositis and malignant disease.

Aged↗
PubMed2003

Dermatomyositis: evolution of a diagnosis.

BACKGROUND AND PURPOSE: As direct access evolves, physical therapists will increasingly encounter patients with pathology that might have an underlying systemic origin. The purpose of this case report is to describe the diagnostic process that led a patient's physical therapist to recognize signs and symptoms of dermatomyositis. CASE DESCRIPTION: The patient was an 18-year-old woman who was referred for physical therapy by her primary care physician on 3 occasions with 3 separate musculoskeletal diagnoses. During the third episode, the physical therapist recognized signs and symptoms that could be indicative of dermatologic disease and referred the patient to a dermatologist. OUTCOMES: A rheumatologist diagnosed the patient's condition as dermatomyositis and referred her for physical therapy. The physical therapy plan of care focused on strengthening and stretching, with an emphasis on a home exercise program. The Medical Outcomes Study 36-Item Short-Form Health Survey (SF-36) indicated that the patient continually made functional improvements over an 18-month period. DISCUSSION: Although diagnosis of diseases such as inflammatory myopathies is not within a physical therapist's scope of practice, this case demonstrates the role a physical therapist can play in recognition of underlying systemic pathology by using the diagnostic process.

Adolescent↗
PubMed2003

[Cardiac effects of dermatomyositis. A case report].

The cardiac effects of dermatomyositis and polymyositis are found frequently at autopsy, even though the clinical manifestations are rare. We report the observation of a patient with dermatomyositis, in whom ventricular tachycardia, and dilated hypokinetic cardiomyopathy were in the foreground of the cardiac anomalies. This rhythm disturbance responds to treatment, as opposed to the associated dilated cardiomyopathy. Electrocardiographic anomalies are frequent, notably conduction disturbances. Ventricular rhythm disturbances are of poorly defined frequency. Cardiac insufficiency is often stabilised with treatment. The other effects are much rare.

Aged↗