Cogan's syndrome (CS) is a rare autoimmune disease in which approximately 10%-13% of people with the condition develop neurological symptoms. While glucocorticoids are the standard of care for patients with CS, disease-modifying anti-rheumatic drugs (DMARDs) and biologics agents are more widely used to treat the systemic and vestibular auditory manifestations of CS. Herein, we report a rare case of CS with central nervous system damage who failed to respond to systemic use of glucocorticoids and DMARDs. However, his symptoms were successfully improved by intrathecal injection of methotrexate (MTX) and dexamethasone. To our knowledge, the use of intrathecal injections of MTX and dexamethasone to treat CS has not been reported in any literature. Therefore, the present case may provide a new idea for clinicians to treat central nervous system symptoms in patients with CS.
Keywords: Cogan's syndrome; central nervous system damage; intracranial hypertension; intrathecal injection.
© 2023 The Authors. International Journal of Rheumatic Diseases published by Asia Pacific League of Associations for Rheumatology and John Wiley & Sons Australia, Ltd.