Myopathy with anti-Jo-1 antibodies: pathology in perimysium and neighbouring muscle fibres
Tahseen Mozaffar, Alan Pestronk
Department of
Neurology, Washington University School of Medicine, St Louis, MO
63110, USA
Correspondence to: Dr Alan Pestronk, Department of Neurology, Box 8111, 660 South Euclid Avenue, St Louis, MO 63110, USA email pestronk{at}kids.wustl.edu
Received 22 July 1999 and in revised form 1 November 1999;
Accepted 23 November
1999
OBJECTIVE
To
evaluate muscle pathology and clinical characteristics in patients with
a myopathy and serum antibodies to the Jo-1 antigen (histidyl t-RNA synthetase).
BACKGROUNDAnti-Jo-1
antibodies occur in syndromes that may include muscle weakness and
pain, Raynaud's phenomenon, interstitial lung disease, arthritis, and
a skin rash different from that seen in dermatomyositis. The muscle
pathology is not well defined.
METHODSCase series.
Review of charts, muscle biopsies, and laboratory records. Features of
myopathology in 11 patients with anti-Jo-1 antibody associated
myopathies were compared with other types of inflammatory myopathies.
RESULTSMyopathology
in patients with anti-Jo-1 antibodies consistently included
fragmentation of, and macrophage predominant inflammation in,
perimysial connective tissue. Perifascicular myopathic changes, including atrophy, regenerating muscle fibres, and some muscle fibre
necrosis, were most common in regions near the connective tissue
pathology and were most prominent in patients with more severe
weakness. Unlike many other inflammatory myopathies, inflammation in
endomysial and perivascular regions was uncommon. By contrast with
dermatomyositis, capillary density was normal.
CONCLUSIONSMyopathological
changes in the anti-Jo-1 antibody syndrome include perimysial
connective tissue fragmentation and inflammation, with muscle fibre
pathology in neighbouring perifascicular regions. Myositis with
anti-Jo-1 antibodies may result from an immune mediated disorder of
connective tissue.
Keywords: myositis; fasciitis; Jo-1 antibody; myopathy; immune
© 2000 by Journal of Neurology, Neurosurgery, and Psychiatry
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