First-line ibrutinib for Bing-Neel syndrome

Tallant, Aaron; Selig, Daniel J.; Wanko, Sam O.; Roswarski, Joseph

doi:10.1136/bcr-2018-226102

Cited by 5 publications

(4 citation statements)

References 12 publications

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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%

How we manage Bing–Neel syndrome

Castillo

Treon

2019

Br J Haematol

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Summary Bing–Neel syndrome (BNS) is an uncommon presentation of Waldenström macroglobulinaemia (WM), seen during the course of the disease in about 1% of patients. BNS occurs when WM cells gain access to the central nervous system (CNS) causing neurological deficits. The diagnosis of BNS is suggested by the presence of radiological abnormalities, such as leptomeningeal enhancement on magnetic resonance imaging and confirmed by the presence of clonal lymphoplasmacytic cells and MYD88 L265P in the cerebrospinal fluid. The treatment of BNS requires agents with good penetration into the CNS, such as fludarabine, methotrexate and cytarabine. The novel Bruton Tyrosine Kinase inhibitor ibrutinib has shown CNS‐penetrating properties, and recent data suggest a therapeutic role in BNS. In this review, we will discuss the clinical and pathological features, diagnostic criteria, treatment options and outcomes of patients with BNS.

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Section: How Do We Treat Bns?mentioning

confidence: 99%

How we manage Bing–Neel syndrome

Castillo

Treon

2019

Br J Haematol

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“…[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%

Treatment of Bing–Neel syndrome with first line sequential chemoimmunotherapy

Gavriatopoulou

Ntanasis‐Stathopoulos

Moulopoulos

et al. 2019

Medicine

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Rationale: Bing-Neel syndrome (BNS) is a rare manifestation of Waldenström macroglobulinemia (WM) with <200 cases reported in the literature. Herein, we describe a case of newly diagnosed BNS treated with a novel therapeutic strategy.Patient concerns: A 67-year-old woman diagnosed with asymptomatic WM 3 years ago presented with gradual vision deterioration the past 3 months. Ophthalmologic examination revealed bilateral reduction in visual acuity (7/10) and bilateral optic disc swelling which was more prominent in the left eye.Diagnoses: Brain imaging revealed bilateral swelling of optic nerves extending from the retina to the optic chiasm and swelling of the left optic tract. Patchy enhancement of optic nerves was also shown upon intravenous contrast administration. Flow cytometry of the cerebrospinal fluid (CSF) revealed the presence of k-light chain restricted, monoclonal B-lymphocytes. CSF protein electrophoresis showed a monoclonal band in the gamma region and immunofixation was positive for immunoglobulin M and kappa light chain. Thus, the diagnosis of BNS was established. Interventions:The patient was initially treated with intrathecal methotrexate and systemic chemotherapy. Following 2 intrathecal methotrexate infusions, CSF flow cytometry did not detect any cells, whereas the patient reported improvement in visual acuity. Therefore, we opted to start maintenance treatment with IV rituximab and per os ibrutinib.Outcomes: Following 1 year posttreatment initiation, visual problems have resolved completely and the patient remains on hematologic and imaging complete response.Lessons: We propose a novel sequential chemoimmunotherapy approach for BNS treatment aiming both at rapid disease control and deep and durable remission with minimization of induced toxicity.

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“…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%

Waldenstrom Macroglobulinemia: Tailoring Therapy for the Individual

Gertz

2022

JCO

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With the introduction of multiple new effective therapeutic options for the treatment of macroglobulinemia, a structured approach to management of this rare lymphoma is currently needed. A review of phase II and III treatment trials over the past 20 years was performed, and high-quality trials are summarized in this manuscript. Because of the lack of large prospective trials comparing different classes of therapy, a uniform recommendation applicable to all patients cannot be made, and the approach must be individualized incorporating patient preferences, comorbidities, and the range of therapeutic toxicities. Therapeutic options for patients with newly diagnosed and previously treated macroglobulinemia are presented on the basis of the best available evidence in the literature.

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First-line ibrutinib for Bing-Neel syndrome

Cited by 5 publications

References 12 publications

How we manage Bing–Neel syndrome

How we manage Bing–Neel syndrome

Treatment of Bing–Neel syndrome with first line sequential chemoimmunotherapy

Waldenstrom Macroglobulinemia: Tailoring Therapy for the Individual

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