The authors report the first case in which the spherules and arthroconidia of Coccidioides immitis were identified from an indwelling cerebrospinal fluid reservoir. The organism was recovered in culture. Present as isolated meningitis, the patient's infection disseminated over a 12-year period despite intrathecal and intravenous administration of amphotericin B. The unusual findings in this patient are compared with findings in 95 cases of infections containing arthroconidia reported since 1940. Arthroconidia is most likely to be found in the tissues of white male patients who are in the third or fourth decade of life and have pulmonary lesions.