Rituximab treatment for autoimmune limbic encephalitis in an institutional cohort

Lee, Woojin; Lee, Sang Kun; Byun, Jung Ick; Sunwoo, Jun Sang; Kim, Tae Joon; Lim, Joo Hyun; Moon, Jangsup; Lee, Han Sang; Shin, Yeong-Gil; Lee, Keon Joo; Kim, Soyun; Jung, Keun Hwa; Jung, Ki Young; Chu, Kon

doi:10.1212/wnl.0000000000002635

Cited by 160 publications

(170 citation statements)

References 32 publications

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“…The outcomes following immunotherapies are generally good, regardless of the identification of autoantibodies [6,7]. For those nonresponsive to first-line immunotherapy, second-line immunotherapies such as rituximab, which induces CD20 + Bcell destruction, yield substantial benefit [8]. However, a considerable proportion of AE might still exhibit inadequate clinical responses to rituximab and consequent unfavorable clinical outcomes [6,8,9].…”

Section: Introductionmentioning

confidence: 99%

“…For those nonresponsive to first-line immunotherapy, second-line immunotherapies such as rituximab, which induces CD20 + Bcell destruction, yield substantial benefit [8]. However, a considerable proportion of AE might still exhibit inadequate clinical responses to rituximab and consequent unfavorable clinical outcomes [6,8,9]. Therefore, clinicians should consider various immune-modulation strategies for AE refractory to rituximab, including further maintenance of rituximab or the Woo-Jin Lee and Soon-Tae Lee contributed equally to this work.…”

Section: Introductionmentioning

confidence: 99%

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Tocilizumab in Autoimmune Encephalitis Refractory to Rituximab: An Institutional Cohort Study

et al. 2016

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A considerable portion of autoimmune encephalitis (AE) does not respond to conventional immunotherapies and subsequently has poor outcomes. We aimed to determine the efficacy of tocilizumab, an anti-interleukin-6 antibody, in rituximab-refractory AE compared with other treatment options. From an institutional cohort of AE, 91 patients with inadequate clinical response to first-line immunotherapy and following rituximab were retrospectively reviewed. Patients were grouped according to their further immunotherapy strategies. Thirty (33.0 %) patients were included in the tocilizumab group, 31 (34.0 %) in the additional rituximab group, and 30 (33.0 %) in the observation group. Outcomes were defined as the favorable modified Rankin Scale scores (≤2) at 1 and 2 months from the initiation of each treatment strategy and at the last follow-up. Favorable clinical response (improvement of the modified Rankin Scale scores by ≥ 2 points or achievement of the mRS scores ≤ 2) at the last follow-up was also analyzed. The tocilizumab group showed more frequent favorable mRS scores at 2 months from treatment initiation and at the last follow-up compared with those at the relevant time points of the remaining groups. The majority (89.5 %) of the patients with clinical improvement at 1 month from tocilizumab treatment maintained a long-term favorable clinical response. No serious adverse effects of rituximab or tocilizumab were reported. Therefore, we suggest that tocilizumab might be a good treatment strategy for treating AE refractory to conventional immunotherapies and rituximab. The tocilizumab-mediated clinical improvement manifests as early at 1 month after treatment initiation.

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Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Tocilizumab in Autoimmune Encephalitis Refractory to Rituximab: An Institutional Cohort Study

et al. 2016

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“…The patients with anti‐NMDAR encephalitis were included who were treated with BTXA injection due to disabling hypersalivation from Sep 30, 2016, when the first injection of BTXA was performed, until Mar 1, 2017. Anti‐NMDAR encephalitis was diagnosed according to clinical presentation and the detection of NMDAR autoantibodies in the serum or cerebrospinal fluid using rat brain section and cell‐based immunocytochemistry kit (Euroimmune Ag, Germany), as described previously 6, 14. The study protocol was approved by the Seoul National University Hospital Institutional Review Board and followed to the principles of the Declaration of Helsinki.…”

Section: Methodsmentioning

confidence: 99%

“…By the first‐line immunotherapy (steroids, immunoglobulins, or plasma exchange) and second‐line immunotherapy (rituximab or cyclophosphamide), about 80% of patients with anti‐NMDAR encephalitis recovers favorably to be able to look after own affairs without assistance 5. However, 7% of the patients still die due to complications such as autonomic dysfunction, pneumonia, hypoventilation, rhabdomyolysis, or status epilepticus 5, 6, 7…”

Section: Introductionmentioning

confidence: 99%

Botulinum toxin treatment for hypersalivation in anti‐NMDA receptor encephalitis

Jun

Seo

Lee

et al. 2017

Ann Clin Transl Neurol

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Hypersalivation is one of the intractable symptoms of anti‐N‐methyl‐d‐aspartate receptor (NMDAR) encephalitis. While anticholinergic medications partially improve the hypersalivation, they can aggravate the autonomic dysfunctions associated with anti‐NMDAR encephalitis. Thus, we investigated the efficacy and safety of botulinum toxin type A injection on hypersalivation refractory to anticholinergics in six patients with anti‐NMDAR encephalitis. Hypersalivation was well‐controlled without remarkable adverse reaction over 16 weeks after botulinum toxin type A, although two patients were reinjected at 12 weeks due to reaggravation of hypersalivation. Our findings suggest that botulinum toxin type A might be a better choice than anticholinergics for management of hypersalivation in patients with anti‐NMDAR encephalitis.

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“…Although most patients with autoimmune encephalitis respond to immunotherapy,1, 2, 3 a small, but significant, number of patients show an insufficient response even after aggressive immunotherapy; therefore, several treatment options are being explored 4, 5, 6. Long‐lived plasma cells that can survive without cell division and continuously secrete autoantibodies are considered to contribute to the poor clinical course in the patients, given their known resistance to B‐cell depleting agents, conventional immunosuppressants, and antiproliferative agents 7…”

Section: Introductionmentioning

confidence: 99%

Bortezomib treatment for severe refractory anti‐NMDA receptor encephalitis

Shin

Lee

Kim

et al. 2018

Ann Clin Transl Neurol

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ObjectiveTo evaluate the therapeutic potential of bortezomib, a proteasome inhibitor that target plasma cells, in order to revive stalled recovery in patients with anti‐N‐methyl‐d‐aspartate (NMDA) receptor encephalitis who remain bedridden even after aggressive immunotherapy.MethodsWe consecutively enrolled patients with anti‐NMDA receptor encephalitis who remained bedridden after first‐line immunotherapy (steroids and intravenous immunoglobulin), second‐line immunotherapy (rituximab), and tocilizumab treatment, and treated them with subcutaneous bortezomib. Clinical response, functional recovery, and changes in antibody titer in the serum and cerebrospinal fluid were measured.ResultsBefore the bortezomib treatment, the five patients with severe refractory anti‐NMDA receptor encephalitis were in a vegetative state. During the 8 months of follow‐up period, three patients improved to minimally conscious states within 2 months of bortezomib treatment, one failed to improve from a vegetative state. However, no patient achieved functional recovery as measured by the modified Rankin Scale score (mRS). Three patients advanced to a cyclophosphamide with bortezomib and dexamethasone regimen, which only resulted in additional adverse events, without mRS improvement. Among the four patients whose antibody titer was followed, two demonstrated a twofold decrease in the antibody titer in serum and/or cerebrospinal fluid after 2 cycles of bortezomib.InterpretationAlthough there were some improvements in severe refractory patients, clinical response to bortezomib was limited and not clearly distinguishable from the natural course of the disease. The clinical benefit of bortezomib in recent studies requires further validation in different clinical settings.

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Rituximab treatment for autoimmune limbic encephalitis in an institutional cohort

Abstract: This study provides Class IV evidence that rituximab improves mRS scores for patients with autoimmune limbic encephalitis who fail first-line therapy.

Cited by 160 publications

References 32 publications

Tocilizumab in Autoimmune Encephalitis Refractory to Rituximab: An Institutional Cohort Study

Tocilizumab in Autoimmune Encephalitis Refractory to Rituximab: An Institutional Cohort Study

Botulinum toxin treatment for hypersalivation in anti‐NMDA receptor encephalitis

Bortezomib treatment for severe refractory anti‐NMDA receptor encephalitis

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