Case Report: Severe rebound after withdrawal of fingolimod in a patient with neuromyelitis optica spectrum disorder
PurposeFingolimod, an oral treatment for relapsing-remitting multiple sclerosis (RRMS), has been associated with a significant rebound in disease activity after therapy cessation. We described a patient with neuromyelitis optica spectrum disorder (NMOSD) who was previously diagnosed with RRMS and ex...
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| Format: | Article |
| Language: | English |
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Frontiers Media S.A.
2023-04-01
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| Series: | Frontiers in Immunology |
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| Online Access: | https://www.frontiersin.org/articles/10.3389/fimmu.2023.1115120/full |
| _version_ | 1797847230071177216 |
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| author | Wei Lin Chung-Hsing Chou Fu-Chi Yang Chia-Kuang Tsai Yu-Kai Lin Yueh-Feng Sung |
| author_facet | Wei Lin Chung-Hsing Chou Fu-Chi Yang Chia-Kuang Tsai Yu-Kai Lin Yueh-Feng Sung |
| author_sort | Wei Lin |
| collection | DOAJ |
| description | PurposeFingolimod, an oral treatment for relapsing-remitting multiple sclerosis (RRMS), has been associated with a significant rebound in disease activity after therapy cessation. We described a patient with neuromyelitis optica spectrum disorder (NMOSD) who was previously diagnosed with RRMS and experienced fatal rebound syndrome after cessation of fingolimod.Case reportA 54-year-old woman, previously diagnosed with RRMS, experienced relapse after orthopedic surgery. The diagnosis was later revised to NMOSD based on a positive aquaporin-4 antibody. Three weeks after converting the immunomodulator from fingolimod to azathioprine, severe disease reactivation was observed. Considering the multiple new and enlarging magnetic resonance imaging lesions, the temporal relationship between fingolimod cessation and symptom onset, and the relatively low possibility of disease reactivation within a short time, the diagnosis of fingolimod withdrawal syndrome was proposed. Although immediate steroid pulse therapy and plasma exchange were performed, the patient eventually died owing to a fulminant clinical course.ConclusionFingolimod withdrawal syndrome is well known in patients with multiple sclerosis (MS). It can also occur in patients with NMOSD. Recognizing patients with NMOSD who present with MS-like manifestations, and avoiding drugs that may be harmful to patients with NMOSD, are important. |
| first_indexed | 2024-04-09T18:09:03Z |
| format | Article |
| id | doaj.art-77bc941446854da3aeed0cfb8b950060 |
| institution | Directory Open Access Journal |
| issn | 1664-3224 |
| language | English |
| last_indexed | 2024-04-09T18:09:03Z |
| publishDate | 2023-04-01 |
| publisher | Frontiers Media S.A. |
| record_format | Article |
| series | Frontiers in Immunology |
| spelling | doaj.art-77bc941446854da3aeed0cfb8b9500602023-04-14T04:29:43ZengFrontiers Media S.A.Frontiers in Immunology1664-32242023-04-011410.3389/fimmu.2023.11151201115120Case Report: Severe rebound after withdrawal of fingolimod in a patient with neuromyelitis optica spectrum disorderWei LinChung-Hsing ChouFu-Chi YangChia-Kuang TsaiYu-Kai LinYueh-Feng SungPurposeFingolimod, an oral treatment for relapsing-remitting multiple sclerosis (RRMS), has been associated with a significant rebound in disease activity after therapy cessation. We described a patient with neuromyelitis optica spectrum disorder (NMOSD) who was previously diagnosed with RRMS and experienced fatal rebound syndrome after cessation of fingolimod.Case reportA 54-year-old woman, previously diagnosed with RRMS, experienced relapse after orthopedic surgery. The diagnosis was later revised to NMOSD based on a positive aquaporin-4 antibody. Three weeks after converting the immunomodulator from fingolimod to azathioprine, severe disease reactivation was observed. Considering the multiple new and enlarging magnetic resonance imaging lesions, the temporal relationship between fingolimod cessation and symptom onset, and the relatively low possibility of disease reactivation within a short time, the diagnosis of fingolimod withdrawal syndrome was proposed. Although immediate steroid pulse therapy and plasma exchange were performed, the patient eventually died owing to a fulminant clinical course.ConclusionFingolimod withdrawal syndrome is well known in patients with multiple sclerosis (MS). It can also occur in patients with NMOSD. Recognizing patients with NMOSD who present with MS-like manifestations, and avoiding drugs that may be harmful to patients with NMOSD, are important.https://www.frontiersin.org/articles/10.3389/fimmu.2023.1115120/fullfingolimod withdrawal syndromerelapse-remitting multiple sclerosisneuromyelitis optica spectrum disorderfingolimod rebound syndromeaquaporin-4 antibody |
| spellingShingle | Wei Lin Chung-Hsing Chou Fu-Chi Yang Chia-Kuang Tsai Yu-Kai Lin Yueh-Feng Sung Case Report: Severe rebound after withdrawal of fingolimod in a patient with neuromyelitis optica spectrum disorder Frontiers in Immunology fingolimod withdrawal syndrome relapse-remitting multiple sclerosis neuromyelitis optica spectrum disorder fingolimod rebound syndrome aquaporin-4 antibody |
| title | Case Report: Severe rebound after withdrawal of fingolimod in a patient with neuromyelitis optica spectrum disorder |
| title_full | Case Report: Severe rebound after withdrawal of fingolimod in a patient with neuromyelitis optica spectrum disorder |
| title_fullStr | Case Report: Severe rebound after withdrawal of fingolimod in a patient with neuromyelitis optica spectrum disorder |
| title_full_unstemmed | Case Report: Severe rebound after withdrawal of fingolimod in a patient with neuromyelitis optica spectrum disorder |
| title_short | Case Report: Severe rebound after withdrawal of fingolimod in a patient with neuromyelitis optica spectrum disorder |
| title_sort | case report severe rebound after withdrawal of fingolimod in a patient with neuromyelitis optica spectrum disorder |
| topic | fingolimod withdrawal syndrome relapse-remitting multiple sclerosis neuromyelitis optica spectrum disorder fingolimod rebound syndrome aquaporin-4 antibody |
| url | https://www.frontiersin.org/articles/10.3389/fimmu.2023.1115120/full |
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