Background

While cytomegalovirus is known to be a highly prevalent virus in humans with seroprevalence for CMV worldwide believed to range from ~60%–100%, it is a relatively rare cause of meningoencephalitis, with most described cases reported in severely immunocompromised patients. While the precise mechanism of immunosuppressive action of hydroxychloroquine is not definitively known, hydroxychloroquine has been hypothesized to interfere with the processing and presentation of antigens and the production of cytokines. There are only a few reports in the literature regarding meningitis in patients undergoing hydroxychloroquine therapy, and no diagnosed CMV meningitis cases have been described in stable patients on hydroxychloroquine. With the resurgence of hydroxychloroquine use in light of the COVID 19 pandemic, we believe this case would cast further light on possible risks associated with the medication.

Case presentation

We report a case of a 17-year old with anti-phospholipid syndrome (on Warfarin) on chronic Hydroxychloroquine therapy, who presented with intractable new onset headaches for several days, that was resistant to high intensity pain medication. The headaches were unremitting, pressure-like, with mild nuchal rigidity, worse on laying flat, and were not associated with fever or visual changes. Neurological examination was non focal, and fundoscopy revealed no papilledema. MRI Brain and C-spine, MRA Brain, MRV brain were unremarkable. LP opening pressure was 27 cm. Meningoencephalitis PCR panel was positive for CMV. Serum CMV IgG was positive, IgM equivocal.

Conclusion

CMV Meningitis has been described in severely immunocompromised patients, however, is considered extremely rare in immunocompetent patients. Elevated CSF pressure is also not frequently associated with viral meningitis. Hydroxychloroquine, while used as an immunomodulator for certain autoimmune conditions, has not been commonly implicated in severe CMV infections like meningitis. Further evaluation and recognition of risks associated with hydroxychloroquine is important in the context of increased use of medication.