Reversible Cerebral Vasoconstriction Syndrome in Association with Fingolimod Use (P3.118)

2014 
OBJECTIVE: Case report of reversible cerebral vasoconstriction syndrome (RCVS) with fingolimod use, and compare this case with two other cases; one with hemorrhagic encephalitis, and the other with peripheral vasoconstriction, both associated with fingolimod use. BACKGROUND: Fingolimod is the first oral disease-modifying drug approved by the FDA for relapsing forms of multiple sclerosis (MS). Fingolimod acts as an antagonist on the sphingosine-1phosphate (S1P) receptor family, specifically at 4 of the 5 G protein-coupled S1P receptors. S1P receptors regulate many physiologic processes in different tissues. Fingolimod’s pharmacological effects therefore have the capacity to be very broad and diverse. Its mechanism of action for MS is reversible sequestration of lymphocytes in the lymph nodes, thus inhibiting migration to sites of inflammation. DESIGN/METHODS: A single observational study compared to two other single observational studies in the literature. Our case is a 39-year-old female with relapsing remitting MS on fingolimod for five months when she was admitted to neurology service for treatment of a MS exacerbation, confirmed by MRI, with IV steroids. On Day 6 of admission she had a non-contrast CT head for thunderclap headache and decreased level of alertness which showed a large frontal hemorrhage. Four-vessel angiogram showed diffuse beading and stenotic appearance. Complete infectious and inflammatory workup was negative for vasculitis including leptomeningeal biopsy. Repeat angiogram at 10 weeks showed resolution of vascular abnormalities. Workup was consistent with RCVS. RESULTS: To date no case of RCVS has been reported with patients on fingolimod. There has been one case of peripheral vasoconstriction and one case of focal hemorrhagic encephalitis. We suspect the case of focal hemorrhagic encephalitis could have been RCVS because no infectious cause was found. CONCLUSIONS: Clinicians should be aware of possible RCVS in patients taking fingolimod. Disclosure: Dr. Belliston has nothing to disclose. Dr. Hammond has received research support from Sunovion Pharmaceuticals. Dr. Sundararajan has nothing to disclose.
    • Correction
    • Source
    • Cite
    • Save
    • Machine Reading By IdeaReader
    0
    References
    0
    Citations
    NaN
    KQI
    []