2018
DOI: 10.1136/bcr-2018-226102
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First-line ibrutinib for Bing-Neel syndrome

Abstract: The authors present a case of an elderly man with a history of Waldenstrom macroglobulinaemia in remission who presented with progressively worsening gait abnormalities and falls for several months. His examination was notable for bilateral lower extremity weakness and an unsteady gait. Brain and spinal MRI showed focal leptomeningeal enhancement in the brain and spinal column. Lumbar puncture was performed and cerebrospinal fluid flow cytometry demonstrated a monoclonal CD5/CD10-negative, CD20-positive B-cell… Show more

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Cited by 5 publications
(4 citation statements)
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“…The level of ibrutinib in CSF was above the 50% inhibitory concentration (IC50) for BTK inhibition. Additional reports have further reported the efficacy of ibrutinib on BNS patients with tumoural forms, as well as in the frontline setting (Boudin et al, 2018;O'Neil et al, 2018;Plander et al, 2018;Tallant et al, 2018).…”
Section: How Do We Treat Bns?mentioning
confidence: 99%
“…The level of ibrutinib in CSF was above the 50% inhibitory concentration (IC50) for BTK inhibition. Additional reports have further reported the efficacy of ibrutinib on BNS patients with tumoural forms, as well as in the frontline setting (Boudin et al, 2018;O'Neil et al, 2018;Plander et al, 2018;Tallant et al, 2018).…”
Section: How Do We Treat Bns?mentioning
confidence: 99%
“…[4] Ibrutinib has been shown to penetrate the blood-brain barrier [9] and has shown significant clinical efficacy as monotherapy among 28 BNS cases in a multicenter case series study [6] and 9 case reports. [9][10][11][12][13][14][15] In our case, we initially chose to administrate high-dose intrathecal methotrexate along with systemic chemotherapy that resulted in rapid improvement in symptoms and disappearance of malignant clonal cells in the CSF. Subsequently, based on the reported efficacy results of both rituximab and ibrutinib we decided to switch therapy to a less toxic chemofree regimen.…”
Section: Discussionmentioning
confidence: 99%
“…Nonetheless, sustained clinical improvement and improvement in imaging of the leptomeninges have been reported with ibrutinib. 56 In a case series of 28 patients, ibrutinib at doses of 420 mg and 560 mg resulted in clinical and imaging improvement in 85% and 60% of patients, respectively, at 3 months. Ibrutinib should be considered the first choice of therapy when the central nervous system is involved with lymphoplasmacytic lymphoma.…”
Section: Ibrutinibmentioning
confidence: 98%