A case of inclusion‐body myositis appearing clinically as an atypical distal atrophy lasting for more than 14 years was diagnosed by visualization of characteristic filamentous inclusions in subsarcolemmal nuclei and in the sarcoplasm near vacuoles. A strain of adenovirus type 2 was isolated from two successive muscle biopsy specimens, and the serum neutralizing antibody titer to this strain was 1:64. The data provide objective support for the assumption that inclusion‐body myositis is caused by a virus infection of muscle, although the filamentous inclusions differ from all known viral structures.

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