Cryptococcus neoformans is an important fungal pathogen causing pneumonia and central nervous system infections mainly in immunocompromised hosts. Fingolimod is an immunomodulatory drug approved in the US and Europe for the treatment of multiple sclerosis.

We herein report a case of cryptococcal meningoencephalitis in a 46-year-old male with a history of fingolimod for five years. He suffered from a progressive headache and visual impairment. These symptoms led to a suspicion of a central nervous system infection and C. neoformans was identified with nucleic acid-based PCR method. Subsequently, appropriate treatment was initiated, and the patient recovered.

Our case underlines the importance of active diagnostic measures such as cerebrospinal fluid analysis in patients under fingolimod treatment with central nervous symptoms. While multiple sclerosis may cause headache and vision impairment, similar symptoms may be caused by central nervous system infections. It has been suggested that fingolimod may subject one to infections and this may occur even years after initiation of the treatment. For our case patient Cerebrospinal fluid sample combined with PCR-based identification provided a rapid diagnosis.