Reversible cerebral vasoconstriction syndrome in association with fingolimod use.
BACKGROUND
Reversible cerebral vasoconstriction syndrome (RCVS), also known as Call-Fleming syndrome, is characterized by thunderclap headaches, non-aneurysmal segmental cerebral vasoconstriction seen on arteriogram, and spontaneously resolves within 12 weeks. Fingolimod has been reported to cause posterior reversible encephalopathy syndrome (PRES) and one case of RCVS.


OBJECTIVE
We report a case of RCVS possibly related to fingolimod use, and compare to cases of adverse outcomes in fingolimod use.


METHODS
This is a single observational study without controls; therefore, level of evidence is IV. A literature review in pubmed with keywords, fingolimod, vasospasm, RCVS, Call-Fleming, stroke, PRES and hemorrhage.


RESULTS
One case of RCVS on fingolimod in the post-partum period. Two other cases in the literature were found one with hemorrhagic encephalitis and the other critical vasospasm in the upper extremity associated with fingolimod 1.25 mg daily in the FREEDOMS II trial and TRANSFORMS study, respectively. Additionally, Novartis reports nine cases of PRES related to fingolimod use.


CONCLUSIONS
Fingolimod has the potential to cause vasoconstriction however appears to be rare and more likely on doses higher than 0.5 mg daily. Fingolimod may be associated in RCVS and should be considered in patients with severe headache on fingolimod.
a Department of Neurology.
Scott
S
Belliston
a Department of Neurology.
Jayshree
J
Sundararajan
a Department of Neurology.
Nancy
N
Hammond
b Department of Pathology , University of Kansas Medical Center , Kansas City , KS , United States.
Kathy
K
Newell
a Department of Neurology.
Sharon
S
Lynch
D000328:Adult; D002533:Cerebral Angiography; D005260:Female; D000068876:Fingolimod Hydrochloride / Q000009:adverse effects*; D006801:Humans; D007166:Immunosuppressive Agents / Q000009:adverse effects*; D008279:Magnetic Resonance Imaging; D020529:Multiple Sclerosis, Relapsing-Remitting / Q000000981:diagnostic imaging / Q000188:drug therapy; D014661:Vasoconstriction / Q000187:drug effects; D020301:Vasospasm, Intracranial / Q000139:chemically induced* / Q000000981:diagnostic imaging
D007166:Immunosuppressive Agents; D000068876:Fingolimod Hydrochloride
Multiple sclerosis, fingolimod, reversible cerebral vasoconstriction syndrome, Gilenya; cerebral hemorrhage