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 CASE REPORT
Year : 2015  |  Volume : 63  |  Issue : 1  |  Page : 72--76

An unusual case of inflammatory necrotizing myopathy and neuropathy with pipestem capillaries


Department of Neurological Sciences, Christian Medical College, Vellore, Tamil Nadu, India

Correspondence Address:
Dr. Mathew Alexander
Prof and Head, Neurology, Department of Neurological Sciences, Christian Medical College, Vellore, Tamil Nadu - 632 004
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.152642

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Necrotizing myopathy with pipestem capillaries is a form of chronic inflammatory myopathy, with histopathology showing necrotizing myopathy, minimal cellular infiltration, and microangiopathy. A 30-year-old female presented with progressive limb weakness of 6 months, with skin pigmentation and Raynaud's phenomenon. Serum creatine phosphokinase was 3990 u/L. Muscle biopsy showed necrotic fibers, focal sparse perivascular inflammation/perifascicular atrophy, endomysial/epimysial vessel wall thickening with luminal narrowing. The features were of inflammatory necrotizing myopathy and neuropathy with pipestem capillaries/microangiopathy. She was pulsed with intravenous immunoglobulin, methylprednisolone, and cyclophosphamide and showed a good improvement. In the absence of widespread inflammatory response and classical histopathology findings, it is important to diagnose this condition as it shows a good response to aggressive and prolonged immunotherapy.






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Online since 20th March '04
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