Complete response with tirabrutinib for relapsed and refractory Bing-Neel syndrome
A 62-year-old female patient was diagnosed with Waldenstrom's macroglobulinemia/lymphoplasmacytic lymphoma (WM/LPL) 8 years ago, which was resolved with rituximab (R) monotherapy. Five years ago, she experienced numbness of the lower limbs, followed by diminished lower limb muscle strength and hearing disturbance. PET-CT scans showed accumulations along the peripheral nerves of the upper and lower limbs together with clonal B lymphocytes in the cerebrospinal fluid, thus a diagnosis of relapse with Bing-Neel syndrome (BNS). After a temporal remission by high-dose cytarabine or bendamustine plus R regimens as salvage treatments, WM/LPL recurred for the third time accompanied by gait disturbances due to muscle weakness and urinary retention. Thus, tirabrultinib was started as a subsequent therapy, which significantly improved the neurological condition together with abnormal findings of magnetic resonance imaging or cerebrospinal fluids. This case is valuable since few relapsed BNS was reported in the literature with successful tirabrutinib treatment.
| Medienart: |
E-Artikel |
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| Erscheinungsjahr: |
2022 |
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| Erschienen: |
2022 |
| Enthalten in: |
Zur Gesamtaufnahme - volume:63 |
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| Enthalten in: |
Rinsho ketsueki] The Japanese journal of clinical hematology - 63(2022), 7 vom: 25., Seite 770-775 |
| Sprache: |
Japanisch |
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| Beteiligte Personen: |
Hagihara, Masao [VerfasserIn] |
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| Links: |
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| Themen: |
Bing-Neel syndrome |
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| Anmerkungen: |
Date Completed 05.08.2022 Date Revised 05.08.2022 published: Print Citation Status MEDLINE |
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| doi: |
10.11406/rinketsu.63.770 |
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| funding: |
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| Förderinstitution / Projekttitel: |
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| PPN (Katalog-ID): |
NLM344425134 |
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| 500 | |a Citation Status MEDLINE | ||
| 520 | |a A 62-year-old female patient was diagnosed with Waldenstrom's macroglobulinemia/lymphoplasmacytic lymphoma (WM/LPL) 8 years ago, which was resolved with rituximab (R) monotherapy. Five years ago, she experienced numbness of the lower limbs, followed by diminished lower limb muscle strength and hearing disturbance. PET-CT scans showed accumulations along the peripheral nerves of the upper and lower limbs together with clonal B lymphocytes in the cerebrospinal fluid, thus a diagnosis of relapse with Bing-Neel syndrome (BNS). After a temporal remission by high-dose cytarabine or bendamustine plus R regimens as salvage treatments, WM/LPL recurred for the third time accompanied by gait disturbances due to muscle weakness and urinary retention. Thus, tirabrultinib was started as a subsequent therapy, which significantly improved the neurological condition together with abnormal findings of magnetic resonance imaging or cerebrospinal fluids. This case is valuable since few relapsed BNS was reported in the literature with successful tirabrutinib treatment | ||
| 650 | 4 | |a Case Reports | |
| 650 | 4 | |a Journal Article | |
| 650 | 4 | |a Bing-Neel syndrome | |
| 650 | 4 | |a Relapse | |
| 650 | 4 | |a Tirabrutinib | |
| 650 | 4 | |a Waldenstrom’s macroglobulinemia | |
| 650 | 7 | |a Imidazoles |2 NLM | |
| 650 | 7 | |a Pyrimidines |2 NLM | |
| 650 | 7 | |a tirabrutinib |2 NLM | |
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| 700 | 1 | |a Ohara, Shin |e verfasserin |4 aut | |
| 700 | 1 | |a Imai, Yui |e verfasserin |4 aut | |
| 700 | 1 | |a Uchida, Tomoyuki |e verfasserin |4 aut | |
| 700 | 1 | |a Inoue, Morihiro |e verfasserin |4 aut | |
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