Anti-myelin-associated-glycoprotein neuropathy successfully treated with tirabrutinib
Background: Anti-myelin-associated-glycoprotein (MAG) neuropathy is a distal, predominantly demyelinating, sensory or sensory-motor polyneuropathy most often developing in the context of an IgM-type monoclonal gammopathy due to monoclonal gammopathy of undetermined significance or lymphoplasmacytic...
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| Format: | Article |
| Language: | English |
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Elsevier
2022-10-01
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| Series: | Heliyon |
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| Online Access: | http://www.sciencedirect.com/science/article/pii/S2405844022022162 |
| _version_ | 1811257272447270912 |
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| author | Hajime Yasuda Yuji Tomizawa Sakiko Harada Makoto Sasaki Norio Komatsu Jun Ando Nobutaka Hattori Miki Ando |
| author_facet | Hajime Yasuda Yuji Tomizawa Sakiko Harada Makoto Sasaki Norio Komatsu Jun Ando Nobutaka Hattori Miki Ando |
| author_sort | Hajime Yasuda |
| collection | DOAJ |
| description | Background: Anti-myelin-associated-glycoprotein (MAG) neuropathy is a distal, predominantly demyelinating, sensory or sensory-motor polyneuropathy most often developing in the context of an IgM-type monoclonal gammopathy due to monoclonal gammopathy of undetermined significance or lymphoplasmacytic lymphoma. Rituximab is considered standard therapy for treatment naïve patients, but optimal treatment methods for relapsed/refractory patients have not been established. Case presentation: We demonstrate that tirabrutinib, a second-generation Burton kinase inhibitor, led to drastic improvements of polyneuropathy that were affirmed by nerve conduction studies in a rituximab-refractory anti-MAG neuropathy patient. Tirabrutinib continues to give excellent disease control with no apparent adverse events at 11 months since initiation, and the patient remains free of plasmapheresis sessions which were originally mandatory. Conclusion: Tirabrutinib is an extremely promising treatment option for anti-MAG neuropathy. |
| first_indexed | 2024-04-12T17:53:53Z |
| format | Article |
| id | doaj.art-7c758f1c874f4665a838ed740e1ad7aa |
| institution | Directory Open Access Journal |
| issn | 2405-8440 |
| language | English |
| last_indexed | 2024-04-12T17:53:53Z |
| publishDate | 2022-10-01 |
| publisher | Elsevier |
| record_format | Article |
| series | Heliyon |
| spelling | doaj.art-7c758f1c874f4665a838ed740e1ad7aa2022-12-22T03:22:25ZengElsevierHeliyon2405-84402022-10-01810e10928Anti-myelin-associated-glycoprotein neuropathy successfully treated with tirabrutinibHajime Yasuda0Yuji Tomizawa1Sakiko Harada2Makoto Sasaki3Norio Komatsu4Jun Ando5Nobutaka Hattori6Miki Ando7Department of Hematology, Juntendo University School of Medicine, Tokyo, Japan; Corresponding author.Department of Neurology, Juntendo University School of Medicine, Tokyo, JapanDepartment of Hematology, Juntendo University School of Medicine, Tokyo, JapanDepartment of Hematology, Juntendo University School of Medicine, Tokyo, JapanDepartment of Hematology, Juntendo University School of Medicine, Tokyo, Japan; Laboratory for the Development of Therapies Against MPN, Juntendo University School of Medicine, Japan; Department of Advanced Hematology, Juntendo University School of Medicine, JapanDepartment of Hematology, Juntendo University School of Medicine, Tokyo, Japan; Department of Cell Therapy and Transfusion Medicine, Juntendo University School of Medicine, JapanDepartment of Neurology, Juntendo University School of Medicine, Tokyo, JapanDepartment of Hematology, Juntendo University School of Medicine, Tokyo, JapanBackground: Anti-myelin-associated-glycoprotein (MAG) neuropathy is a distal, predominantly demyelinating, sensory or sensory-motor polyneuropathy most often developing in the context of an IgM-type monoclonal gammopathy due to monoclonal gammopathy of undetermined significance or lymphoplasmacytic lymphoma. Rituximab is considered standard therapy for treatment naïve patients, but optimal treatment methods for relapsed/refractory patients have not been established. Case presentation: We demonstrate that tirabrutinib, a second-generation Burton kinase inhibitor, led to drastic improvements of polyneuropathy that were affirmed by nerve conduction studies in a rituximab-refractory anti-MAG neuropathy patient. Tirabrutinib continues to give excellent disease control with no apparent adverse events at 11 months since initiation, and the patient remains free of plasmapheresis sessions which were originally mandatory. Conclusion: Tirabrutinib is an extremely promising treatment option for anti-MAG neuropathy.http://www.sciencedirect.com/science/article/pii/S2405844022022162Anti-MAG antibodyIbrutinibBTK inhibitorLymphoplasmacytic lymphomaIgM MGUSRituximab |
| spellingShingle | Hajime Yasuda Yuji Tomizawa Sakiko Harada Makoto Sasaki Norio Komatsu Jun Ando Nobutaka Hattori Miki Ando Anti-myelin-associated-glycoprotein neuropathy successfully treated with tirabrutinib Heliyon Anti-MAG antibody Ibrutinib BTK inhibitor Lymphoplasmacytic lymphoma IgM MGUS Rituximab |
| title | Anti-myelin-associated-glycoprotein neuropathy successfully treated with tirabrutinib |
| title_full | Anti-myelin-associated-glycoprotein neuropathy successfully treated with tirabrutinib |
| title_fullStr | Anti-myelin-associated-glycoprotein neuropathy successfully treated with tirabrutinib |
| title_full_unstemmed | Anti-myelin-associated-glycoprotein neuropathy successfully treated with tirabrutinib |
| title_short | Anti-myelin-associated-glycoprotein neuropathy successfully treated with tirabrutinib |
| title_sort | anti myelin associated glycoprotein neuropathy successfully treated with tirabrutinib |
| topic | Anti-MAG antibody Ibrutinib BTK inhibitor Lymphoplasmacytic lymphoma IgM MGUS Rituximab |
| url | http://www.sciencedirect.com/science/article/pii/S2405844022022162 |
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