Successful treatment with tirabrutinib for relapsed Bing-Neel                    syndrome following high-dose methotrexate and craniospinal                    irradiation

Saburi, Masuho; Sakata, Masanori; Okuhiro, Kazuki; Kawano, Katsuya; Uesugi, Souhei; Wada, Junpei; Urabe, Shogo; Saburi, Yoshio; Ohtsuka, Eiichi

doi:10.3960/jslrt.22018

Cited by 5 publications

(4 citation statements)

References 21 publications

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“…In this case, preventing the relapse of BNS was possible due to the poor age-related prognosis and treatment for WM prior to BNS diagnosis [20]. A search for "Bing-Neel syndrome" and "tirabrutinib" in PubMed on December 3, 2023, identified five studies on tirabrutinib for treating BNS (Table 2) [6][7][8][9][10]. Tirabrutinib demonstrated therapeutic effects in all cases, despite variations in BNS lesions.…”

Section: Discussionmentioning

confidence: 99%

“…Determining the occurrence of BNS is important for treatment selection because BNS is treated with drugs that need to penetrate the CNS. Case reports have suggested that tirabrutinib is effective against BNS [6][7][8][9][10].…”

Section: Introductionmentioning

confidence: 99%

“…Following rituximab, high-dose methotrexate, procarbazine, and vincristine (R-MPV) therapy, the patient's symptoms were resolved with tirabrutinib treatment. We also reviewed five previously reported cases of BNS treated with tirabrutinib [6][7][8][9][10].…”

Section: Introductionmentioning

confidence: 99%

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Waldenström Macroglobulinemia-Associated Peripheral Neuropathy in the Brachial Plexus With Bing-Neel Syndrome Diagnosed Through Repeated Cerebrospinal Tests

Hatasa,

Imoto,

Komori

et al. 2024

Cureus

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In Waldenström macroglobulinemia (WM), confirming the presence of Bing-Neel syndrome (BNS) is important because drugs that penetrate the central nervous system (CNS) must be selected. We report the case of a 75-year-old man for whom tirabrutinib, a second-generation Bruton's tyrosine kinase inhibitor (BTKi), was useful in treating WM-associated peripheral neuropathy (PN) with BNS. Numbness and muscle weakness in the fingers occurred three years after the initial treatment of WM. WM-associated PN due to demyelinating disease was diagnosed based on the results of a nerve conduction study and magnetic resonance imaging showing bilateral symmetric swelling of the brachial plexus. The cerebrospinal fluid (CSF) cytology results were initially negative; however, the CSF test was repeated because of extremely high protein levels (984 mg/dL) and slightly elevated leukocyte counts (14/µL). The second test revealed abnormal lymphoplasmacytic cells (189/µL), indicating BNS. Rituximab and high-dose methotrexatecontaining chemotherapy were administered. Despite the subsequent negative CSF cytology results, his neurological symptoms persisted but subsided soon after the initiation of tirabrutinib. The therapeutic effects of tirabrutinib persisted for 25 months. This case suggested that a careful search for concurrent BNS is important when lesions are close to the CNS or when atypical CSF findings are obtained in patients with WM-associated PN, especially when BTKi options are available.

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Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Waldenström Macroglobulinemia-Associated Peripheral Neuropathy in the Brachial Plexus With Bing-Neel Syndrome Diagnosed Through Repeated Cerebrospinal Tests

Hatasa,

Imoto,

Komori

et al. 2024

Cureus

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“…A 24‐month follow‐up analysis of the data revealed durable response rates [33], and tirabrutinib is now approved in Japan for the treatment of WM. Multiple case reports have also demonstrated the efficacy of this agent in patients with BNS, often after the relapse of WM, subsequent to traditional chemotherapy regimens [34‒38].…”

Section: Discussionmentioning

confidence: 99%

Mixed Diffuse and Tumoral Form of Bing-Neel Syndrome Successfully Treated with Ibrutinib

Naveed Ahmad,

Schroeder,

Yun

et al. 2023

Case Rep Oncol

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Introduction: Bing-Neel syndrome (BNS) is a rare and heterogenous manifestation of Waldenström macroglobulinemia (WM) involving central nervous system (CNS) infiltration by malignant lymphoplasmacytic cells. Efforts to standardize diagnostic criteria have improved in recent years, as have treatment options including the use of the Bruton tyrosine kinase inhibitor (BTKI) ibrutinib. Case Presentation: Here, we present the case of a 70-year-old male with a remote history of WM previously treated with bendamustine and rituximab, who presented to medical attention with several months of left-sided weakness, headache, and ataxia. Brain magnetic resonance imaging revealed numerous enhancing masses in the bilateral cerebral hemispheres, inferior medulla, and upper cervical spine. Laboratory studies showed serum IgM lambda monoclonal gammopathy and elevated free serum kappa and lambda light chains, while cerebrospinal fluid flow cytometry revealed CD19+ B cells. Stereotactic brain biopsy of a right frontal brain lesion was consistent with lymphoplasmacytic lymphoma, confirmed by a positive MYD88 L265P mutation. He received ibrutinib 420 mg orally daily, and this resulted in appreciable clinical and radiologic responses, which have persisted over a 31-month period. Conclusion: The advent of molecularly targeted agents and novel therapies for WM has provided patients and clinicians with additional therapeutic options. The use of BTK inhibitors with their high-level CNS penetrance, in particular, offers a novel way to treat BNS and improve patient overall survival while maintaining a high level of quality of life. We discuss the importance of MYD88 L265P testing in the context of BNS as well as the expanding role of BTKIs in treating this disease.

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Successful treatment with tirabrutinib for relapsed Bing-Neel syndrome following high-dose methotrexate and craniospinal irradiation

Cited by 5 publications

References 21 publications

Waldenström Macroglobulinemia-Associated Peripheral Neuropathy in the Brachial Plexus With Bing-Neel Syndrome Diagnosed Through Repeated Cerebrospinal Tests

Waldenström Macroglobulinemia-Associated Peripheral Neuropathy in the Brachial Plexus With Bing-Neel Syndrome Diagnosed Through Repeated Cerebrospinal Tests

Mixed Diffuse and Tumoral Form of Bing-Neel Syndrome Successfully Treated with Ibrutinib

Multiple drugs

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