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Dermatomyositis and Whipple's disease.

Authors :
Helliwell TR
Appleton RE
Mapstone NC
Davidson J
Walsh KP
Source :
Neuromuscular disorders : NMD [Neuromuscul Disord] 2000 Jan; Vol. 10 (1), pp. 46-51.
Publication Year :
2000

Abstract

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.

Details

Language :
English
ISSN :
0960-8966
Volume :
10
Issue :
1
Database :
MEDLINE
Journal :
Neuromuscular disorders : NMD
Publication Type :
Academic Journal
Accession number :
10677863
Full Text :
https://doi.org/10.1016/s0960-8966(99)00054-1