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Childhood-type myositis and linear scleroderma.

T Miike, Y Ohtani, S Hattori

    Neurology
    |July 1, 1983
    PubMed
    Summary

    Linear scleroderma in a child presented with muscle wasting and skin inflammation. Biopsied tissue revealed autoimmune collagen vascular disorder, suggesting a link between scleroderma and dermatomyositis-like changes.

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    Area of Science:

    • Pediatric Rheumatology
    • Dermatology
    • Neuromuscular Pathology

    Background:

    • Linear scleroderma is a localized connective tissue disease.
    • Childhood dermatomyositis is an autoimmune inflammatory myopathy.
    • Understanding the overlap between these conditions is crucial for diagnosis.

    Observation:

    • A 5-year-old girl presented with linear scleroderma and shoulder girdle muscle wasting.
    • Direct immunofluorescence of skin biopsy showed IgM deposits in blood vessels and dermal-epidermal junction.
    • Muscle biopsy revealed fascicular atrophy, perifascicular atrophy, and perivascular cellular infiltration.

    Findings:

    • Muscle pathology findings were highly similar to childhood-type dermatomyositis.
    • Abnormalities in muscle blood vessels included basal lamina edema and thickening.

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  • Immunofluorescence and histopathology suggested an autoimmune etiology.

    • Implications:

    • This case highlights a potential autoimmune collagen vascular disorder underlying linear scleroderma with myopathic features.
    • The findings suggest a possible overlap or shared pathogenic mechanism between scleroderma and dermatomyositis in children.
    • Further research is warranted to explore the autoimmune basis of scleroderma presenting with muscle involvement.