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PubMed1985

Diagnosis and management of gastrointestinal perforations in childhood dermatomyositis with particular reference to perforations of the duodenum.

Four children with dermatomyositis were recently seen with gastrointestinal perforations. The sites of perforation in the four cases were: (1) the duodenum, esophagus, and colon; (2) the duodenum; (3) the distal stomach; and (4) the traverse colon. The gastric and transverse colon perforations were intraperitoneal and easily diagnosed. The gastric perforation was treated successfully by partial gastrectomy. The patient with the colon perforation underwent exteriorization; death occurred from cerebral complications possibly related to vasculitis. Both duodenal perforations were posterior in the distal descending portion. Enzymatic dissection into the right lower quadrant produced confusing clinical and radiographic signs and extensive retroperitoneal necrosis. Successful treatment was obtained by partial gastrectomy, sump drainage of the perforation, and parenteral nutrition. Gastrointestinal perforation is a well-recognized complication of vasculitis in childhood dermatomyositis. In particular, perforations of the distal duodenum, as reported by others, are associated with delay in diagnosis and high mortality.

Child↗
PubMed1980

Juvenile dermatomyositis: a clinical and immunologic study.

Twenty-one children were diagnosed as having juvenile dermatomyositis on the basis of the strict criteria of Bohan and Peter. In addition to the typical skin and muscle changes, abnormalities of esophageal motility (eight of 19), pulmonary function (14 of 17), ECG (10 of 20), and gastrointestinal absorption of D-xylose (two of eight) with active disease were observed. Clinical signs of other collagen vascular disease appeared in five children. Serologic evaluation demonstrated that ANA and rheumatoid factor were transiently positive in six; one child developed a persistently positive rheumatoid factor after four years of disease inactivity. Antibody to ENA was negative in all, but antibody to PM-1 antigen was present in four of 18. Six had a low C3 or C4; evidence of immune complexes was demonstrated by Clq or Raji binding in eight with active disease. One child was IgA deficient. The HLA-B8 antigen was present in 72% of the Caucasian children as compared with the expected incidence of 21%. Therefore, classical dermatomyositis in children has more systemic involvement then previously appreciated, may be related to the presence of circulating immune complexes, and appears to be under immunogenetic control.

Adolescent↗
PubMed1999

Novel gastrointestinal tract manifestations in juvenile dermatomyositis.

We report a case of juvenile dermatomyositis in which a dilated atonic esophagus was associated with delayed gastric emptying and intestinal mucosal thickening, resulting in a radiographic "stacked coin" appearance. These findings, which can also occur in infectious, neoplastic, or other immune-mediated diseases, broaden the spectrum of gastrointestinal tract manifestations in juvenile dermatomyositis. Physicians should be alert for these treatable manifestations in children with myositis who present with unexplained gastrointestinal symptoms, which are reversible with immunosuppressive therapy.

Anti-Inflammatory Agents↗
PubMed1988

Ureteral necrosis in dermatomyositis.

We describe a child with dermatomyositis and calcified necrosis in the middle third of both ureters. Histopathological examination showed vasculitis associated with ureteral necrosis and calcification. These findings together with a similar previous report in the literature have prompted us to correlate dermatomyositis in childhood with ureteral necrosis and to anticipate specifically a lesion in the middle third of the ureter because of the relative lack of blood flow in that segment.

Child↗
PubMed1998

The dermatomyositis-specific autoantigen Mi2 is a component of a complex containing histone deacetylase and nucleosome remodeling activities.

Histone acetylation and deacetylation were found to be catalyzed by structurally distinct, multisubunit complexes that mediate, respectively, activation and repression of transcription. ATP-dependent nucleosome remodeling, mediated by different multisubunit complexes, was thought to be involved only in transcription activation. Here we report the isolation of a protein complex that contains both histone deacetylation and ATP-dependent nucleosome remodeling activities. The complex contains the histone deacetylases HDAC1/2, histone-binding proteins, the dermatomyositis-specific autoantigen Mi2beta, a polypeptide related to the metastasis-associated protein 1, and a novel polypeptide of 32 kDa. Patients with dermatomyositis have a high rate of malignancy. The finding that Mi2beta exists in a complex containing histone deacetylase and nucleosome remodeling activities suggests a role for chromatin reorganization in cancer metastasis.

Adenosine Triphosphatases↗
PubMed1998

Dermatomyositis.

Dermatomyositis is a rare inflammatory myopathy with characteristic skin manifestations and muscular weakness. The disease can be categorized as adult idiopathic, juvenile, or amyopathic dermatomyositis as well as that associated with a connective tissue disease or a malignancy. Immunologic factors are most likely involved in the pathogenesis of the disease; however, genetic and environmental issues may also play important roles. Treatment with immunosuppressive agents has proved successful in the majority of patients, although significant morbidity still occurs.

Autoantibodies↗
PubMed2001

[Macro-CK disclosing disseminated epidermoid carcinoma of the pyriform sinus in a patient with dermatomyositis].

INTRODUCTION: The association between dermatomyositis and cancer is clearly established, but its frequency remains difficult to define. EXEGESIS: We report the case of an association between a dermatomyositis and a cancer of the piriform antrum. Four months after surgical treatment and radiotherapy, the increased macro-CK level gave us reason to suspect a cancer relapse with pulmonary, hepatic, splenic and renal metastases without progressive clinical signs. CONCLUSION: Mitochondrial macro-CK detection must evoke the presence of neoplasia with or without metastasis, which would be a poor prognosis, as has been shown in our patient.

Carcinoma, Squamous Cell↗
PubMed1998

Germ cell cancer and dermatomyositis.

Although the association of dermatomyositis and malignancy is well recognized, there have been few previous reports of germ cell tumours occurring in this context. Two cases of teratoma with dermatomyositis have presented at the Wessex Medical Oncology Unit in Southampton, which prompted a review of the literature.

Adult↗
PubMed2000

Dermatomyositis and Whipple's disease.

A 14-year-old boy presented with a 3-year history of a skin rash typical of juvenile dermatomyositis, and a 2-month history of mild proximal weakness, myalgia, and weight loss. A quadriceps biopsy showed perifascicular fibre atrophy, focal necrosis and regeneration, immunohistochemical labelling for HLA-1 on the surface of the fibres, and focal C5-9 deposition in capillaries. Macrophages with diastase-resistant, PAS-positive cytoplasm were present. Ultrastructural studies showed electron dense and membranous debris. The patient's symptoms responded to intravenous immunoglobulin and oral prednisolone. Four months after discontinuing prednisolone, the patient developed cardiac failure, ventricular tachycardia, and a recurrence of his rash. The 16S ribosomal RNA specific for Tropheryma whippelii was identified by polymerase chain reaction (PCR) analysis in skeletal and cardiac muscle. The myalgia and skin rash responded to prednisolone and oral co-trimoxazole, and the tachycardia is controlled by oral verapamil. This patient appears to have a novel association of juvenile dermatomyositis and Whipple's disease.

Actinobacteria↗
PubMed1999

Bilateral condylar resorption in dermatomyositis: a case report.

Polymyositis is an inflammatory disease commonly affecting the striated muscle. When it is accompanied by characteristic skin lesions, the condition is called dermatomyositis. Bilateral condylar resorption has been reported with autoimmune conditions and chronic systemic steroids. We report the first documented case of bilateral condylar resorption in a patient with dermatomyositis. Possible etiologic factors and treatment outcomes are discussed.

Adult↗
PubMed1998

Childhood dermatomyositis: clinical course of 36 patients treated with low doses of corticosteroids.

Thirty-six patients with juvenile dermatomyositis, seen consecutively between 1983 and 1996 and treated initially with low doses of corticosteroids (prednisolone 1 mg/kg/day), were studied retrospectively to evaluate their long-term evolution and to identify factors predictive of the functional outcome. After a mean follow-up of 4.9 years, 28 (78%) of the patients were well without functional impairment; five patients had inactive disease but with persisting disabilities; and three patients had active disease despite several years of treatment. Fifteen children (42%) developed dystrophic calcifications which, in five of these patients, interfered with functions. These patients treated for juvenile dermatomyositis with a low dose corticosteroid regimen had an evolution identical to that of the published series of patients treated with higher doses and probably had a better quality of life. The best predictors of good functional recovery and minimal calcinosis were early treatment after the onset of symptoms and low creatine kinase serum level at the time of diagnosis.

Adolescent↗
PubMed2001

An extremely severe case of cutaneous calcinosis with juvenile dermatomyositis, and successful treatment with diltiazem.

A case of cutaneous calcinosis associated with juvenile dermatomyositis is described. The patient was a 3-year-old girl who had been diagnosed as having dermatomyositis at age 1 year. She was treated with prednisolone, but developed multiple calcified nodules in the subcutaneous tissues and intermuscular fascia. These nodules gradually increased in size despite continual therapy with steroids and aluminium hydroxide. Treatment with diltiazem completely suppressed the development of calcinosis.

Calcinosis↗
PubMed1999

Dermatomyositis-like eruption and leg ulceration caused by hydroxyurea in a patient with psoriasis.

We report the case of an elderly woman who had been on hydroxyurea for long-standing widespread psoriasis. After approximately 5 years's treatment with hydroxyurea, she developed a symmetrical dermatomyositis-like eruption on her hands, together with bilateral leg ulceration. Although similar skin eruptions have been reported after long-term hydroxyurea treatment, all of the previous patients were being treated for myeloproliferative disorders. A dermatomyositis-like eruption has not previously been reported to occur as a consequence of hydroxyurea treatment for psoriasis. Its recognition is important to prevent unnecessary investigation or treatment withdrawal.

Aged↗
PubMed1998

Amyopathic dermatomyositis in Hong Kong -- association with nasopharyngeal carcinoma.

BACKGROUND: Amyopathic dermatomyositis (ADM) is a rare variant of dermatomyositis (DM) in which there is no apparent muscle involvement at initial presentation, but clinical and laboratory evidence of myopathy may appear later. Like DM, it has a known association with malignancy. OBJECTIVE: To record the occurrence of ADM in Hong Kong during the period from January 1988 to June 1996, and to establish the types of carcinoma with which it is associated. METHODS: The records of all patients diagnosed with ADM in three major dermatology clinics in Hong Kong were retrospectively studied to confirm the diagnosis and to look for any associated malignancy at any stage of the disease. RESULTS: Between 1988 and 1996, six patients in Hong Kong (five men and one woman; mean age, 50 years) fulfilled the criteria of ADM. During a follow-up period ranging from 20 to 90 months (average, 35.6 months), none developed any evidence of myositis. Associated malignancy was found in the five male patients. There were three cases of nasopharyngeal carcinoma (NPC), one case of non-small cell carcinoma of the lung, and one case of metastatic carcinoma of the cervical lymph node of unknown primary. No carcinoma had developed in the female patient during a follow-up period of 90 months. CONCLUSIONS: There is a strong association between ADM and NPC in Hong Kong.

Adult↗
PubMed2001

Dermatomyositis.

Dermatomyositis is a disease that has a characteristic skin eruption that may occur with or without a proximal myopathy. The disease with cutaneous features only is classified as amyopathic dermatomyositis. The origin is unknown, but autoimmune factors are believed to play an important role. Autoantibodies are found in most patients and some have myositis-specific antibodies. Systemic changes may occur and there appears to be a relationship to internal malignancy, particularly in older patients. Juvenile disease has an associated vasculopathy. Treatment includes systemic corticosteroids and other immunosuppressive agents. The cutaneous changes may be difficult to treat.

Dermatomyositis↗
PubMedDate not supplied

Juvenile dermatomyositis with respiratory failure and peripheral nerve paralyses. A case report.

A 9.5 years old girl suffering from an acute dermatomyositis, developed a respiratory failure, which was successfully managed with mechanical ventilation for 6 days. Paralysis of the right radial and the left peroneus nerves were recognized early in the disease course and were confirmed later by electrophysiologic examinations. A complete remission of the dermatomyositis and the paralysis of the radial nerve were achieved within months with prednisolone medication and physiotherapy; the peroneus nerve paralysis improved only slightly. Further uncommon manifestations were thrombocytopenia, retinitis and cerebral convulsions.

Child↗
PubMed2003

Multicentric reticulohistiocytosis presenting with clinical features of dermatomyositis.

Multicentric reticulohistiocytosis (MRH) is a rare disorder with proliferating histiocytes that develop into multinucleated giant cells with "ground-glass" cytoplasm. The disease presents with a rapidly destructive, sometimes permanently debilitating, polyarthritis and a papulonodular eruption, generally of the face and hands. We present 3 cases of MRH in which the initial clinical diagnosis was thought to be dermatomyositis (DM). The cutaneous findings in these cases included an erythematous, predominantly photodistributed macular and papular eruption clinically consistent with DM. However, skin biopsy specimens revealed the diagnosis of MRH. This previously unreported clinical similarity between MRH and DM is significant because the treatment options and future complications vary greatly between the 2 diseases. Thus, it is important to consider the possibility of multicentric reticulohistiocytosis when evaluating a patient with the clinical diagnosis of dermatomyositis.

Adult↗