A 37-year-old man with the characteristic clinical signs of Kartagener's syndrome, including bronchiectasia, chronic paranasal sinusitis, and situs inversus, was found to have immotile cilia and sperm. Electron microscopy demonstrated that the majority of cilia lacked dynein arms and radial spokes and that various defects of microorgans existed in the sperm. The most frequent defect was total defect of axoneme followed by defect of inner dynein arms in the sperm. These findings suggest an association between the structural abnormality of absent inner dynein arms and immotility of cilia and sperm in Kartagener's syndrome.