Contributions
Abstract: EP1725
Type: Poster Sessions
Abstract Category: Therapy - Others
Introduction: Unexpected magnetic resonance imaging (MRI) activity, with an unusually high number of gadolinium-enhancing lesions, and severe clinical rebound of disease is observed in multiple sclerosis (MS) patients, 8-24 weeks after fingolimod withdrawal. Here we describe a case of a patient who, after several years under fingolimod, presented MRI characteristics of a severe « rebound syndrome », while being adherent to treatment.
Case summary: We report the case of a 41 year-old female with relapsing-remitting MS (disease duration = 20 years), initially treated with subcutaneous interferon beta-1a during 13 years, who was subsequently switched to fingolimod due to its oral intake and the lack of control of disease activity (five relapses between 1997 and 2008, EDSS score = 1). Both clinical and radiological stability was achieved under fingolimod during six years. However, a routine MRI depicted more than 30 gadolinium enhancing lesions together with an abnormal neurological exam (EDSS = 3; back to 1 after intravenous methyprednisolone). Patient was paucisymptomatic : chief complaints included dizziness and instability. Adherence to recommended treatment was irreproachable, supported by a grade III lymphopenia. Serologic and cerebral spinal fluid (CSF) testing for infectious diseases were negative, as well as bacterial and fungus cultures. AQP4 and MOG antibodies were unremarkable. No tumoral cells were found in the CSF. Given the unusual radiological presentation, and to exclude lymphoma or infections, a brain biopsy of the right frontal lesion was performed, showing evidence of active inflammation and demyelination as seen in MS.
Conclusion: although a large body of evidence confirms important inflammatory activity occuring after fingolimod withdrawal, only few publications reported this kind of radiological progression during adequate treatment intake, generally ressembling more to pseudotumoral relapses than multifocal widespread relapse as seen here. We hypothesize that, in certain patients, fingolimod might entail SP1Rs downregulation, thus increasing CNS inflammation risk. Further studies must be carried out to confirm this hypothesis.
Disclosure: G. Breville have nothing to disclose. A.M. Lascano received travelling expenses from Roche. P. H. Lalive received honoraria for speaking from Biogen-Idec, CSL Bering, Merck Serono, Novartis, Sanofi-Aventis, Teva; consulting fees from Biogen-Idec, Geneuro, Genzyme, Merck Serono, Novartis, Roche, Sanofi-Aventis, Teva; research grants from Biogen-Idec, Merck Serono, Novartis, Swiss MS society, Swiss National Foundation. S. Roth received honoraria from Biogen, Merck, Novartis, Roche, Sanofi-Genzyme, Teva.
Abstract: EP1725
Type: Poster Sessions
Abstract Category: Therapy - Others
Introduction: Unexpected magnetic resonance imaging (MRI) activity, with an unusually high number of gadolinium-enhancing lesions, and severe clinical rebound of disease is observed in multiple sclerosis (MS) patients, 8-24 weeks after fingolimod withdrawal. Here we describe a case of a patient who, after several years under fingolimod, presented MRI characteristics of a severe « rebound syndrome », while being adherent to treatment.
Case summary: We report the case of a 41 year-old female with relapsing-remitting MS (disease duration = 20 years), initially treated with subcutaneous interferon beta-1a during 13 years, who was subsequently switched to fingolimod due to its oral intake and the lack of control of disease activity (five relapses between 1997 and 2008, EDSS score = 1). Both clinical and radiological stability was achieved under fingolimod during six years. However, a routine MRI depicted more than 30 gadolinium enhancing lesions together with an abnormal neurological exam (EDSS = 3; back to 1 after intravenous methyprednisolone). Patient was paucisymptomatic : chief complaints included dizziness and instability. Adherence to recommended treatment was irreproachable, supported by a grade III lymphopenia. Serologic and cerebral spinal fluid (CSF) testing for infectious diseases were negative, as well as bacterial and fungus cultures. AQP4 and MOG antibodies were unremarkable. No tumoral cells were found in the CSF. Given the unusual radiological presentation, and to exclude lymphoma or infections, a brain biopsy of the right frontal lesion was performed, showing evidence of active inflammation and demyelination as seen in MS.
Conclusion: although a large body of evidence confirms important inflammatory activity occuring after fingolimod withdrawal, only few publications reported this kind of radiological progression during adequate treatment intake, generally ressembling more to pseudotumoral relapses than multifocal widespread relapse as seen here. We hypothesize that, in certain patients, fingolimod might entail SP1Rs downregulation, thus increasing CNS inflammation risk. Further studies must be carried out to confirm this hypothesis.
Disclosure: G. Breville have nothing to disclose. A.M. Lascano received travelling expenses from Roche. P. H. Lalive received honoraria for speaking from Biogen-Idec, CSL Bering, Merck Serono, Novartis, Sanofi-Aventis, Teva; consulting fees from Biogen-Idec, Geneuro, Genzyme, Merck Serono, Novartis, Roche, Sanofi-Aventis, Teva; research grants from Biogen-Idec, Merck Serono, Novartis, Swiss MS society, Swiss National Foundation. S. Roth received honoraria from Biogen, Merck, Novartis, Roche, Sanofi-Genzyme, Teva.