Ipilimumab/Nivolumab-related Opsoclonus-Myoclonus-Ataxia Syndrome Variant in a Patient with Malignant Pleural Mesothelioma

Maller, Bradley; Peguero, Edwin; Tanvetyanon, Tawee

doi:10.1097/cji.0000000000000228

Cited by 18 publications

(7 citation statements)

References 7 publications

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“…For these patients, diplopia or eye movement abnormalities, hyperphagia, or impaired consciousness were present. Less frequently, patients presented with extralimbic encephalitis involving the cerebellum, basal ganglia, or encephalomyelitis, in whom cerebellar ataxia, chorea, or weakness in the extremities, respectively, was present. Signs such as fever and language impairment were less frequently reported in this group.…”

Section: Resultsmentioning

confidence: 99%

Encephalitis Induced by Immune Checkpoint Inhibitors

et al. 2021

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IMPORTANCEEncephalitis is a severe immune-related adverse event secondary to treatment with immune checkpoint inhibitors (ICIs). The spectrum of ICI-induced encephalitis (ICI-iE) ranges from disease that resolves fully to lethal forms. Moreover, ICIs may unmask a paraneoplastic encephalitis. To our knowledge, the factors associated with ICI-iE prognosis are unknown.OBJECTIVES To evaluate the presentation of ICI-iE and to identify features helpful in assessing outcomes.EVIDENCE REVIEW This systematic review pooled case series from the published literature (n = 77) and medical records from 1 center (n = 5) to assess the association between the form of ICI-iE presentation and its prognosis. Eligibility criteria included references identified by searches of PubMed and Web of Knowledge databases in the English literature from June 2000 (first patient dose of ipilimumab) to April 17, 2020, that examined patients with encephalitis with presumed autoimmune etiologic features induced by ICIs. Information regarding clinical, cerebrospinal fluid, and neuroimaging (magnetic resonance imaging) features, as well as treatment given, were extracted. FINDINGS A total of 82 patients (52 men [63%]; median age, 61.0 years [interquartile range, 52.5-70.0 years]) were included. Most patients presented with focal syndromes (39 [48%])or meningoencephalitis (36 [44%]). Seven patients (9%) had nonclassifiable ICI-iE. Neuronal autoantibodies were detected in 23 patients with focal syndromes and 1 patient with nonclassifiable ICI-iE. Most autoantibodies were onconeuronal (17 of 24 [71%]), targeting intracellular antigens. Patients without a focal syndrome or with a negative-antibody focal syndrome had a good prognosis (49 of 55 [89%]). Among patients with autoantibodies, those with anti-glutamic acid decarboxylase or anticell surface responded to treatment and had a favorable prognosis (100%). However, patients with other autoantibodies had poor outcomes (17 of 24 [71%]). Antineuronal autoantibodies (13 of 24 [54%] vs 5 of 41 [12%]; P < .001), focal syndrome (16 of 39 [41%] vs 4 of 43 [9%]; P = .001), and abnormal magnetic resonance imaging findings (14 of 39 [36%] vs 4 of 32 [13%]; P = .02) were associated with poor outcomes. Conversely, fever (21 of 23 [91%] vs 41 of 59 [70%]; P = .04) and more inflammatory changes in cerebrospinal fluid (30 of 31 [97%] vs 21 of 33 [64%]; P = .001) were associated with a better prognosis.CONCLUSIONS AND RELEVANCE Immune checkpoint inhibitors may induce mainly 2 different encephalitic syndromes: a focal limbic or extralimbic encephalitis and a meningoencephalitis. Immune checkpoint inhibitor-induced encephalitis is associated with an overall favorable outcome, with a low rate of fatal events. An undetected preexisting paraneoplastic encephalitic syndrome may be triggered by ICIs, and this type of syndrome has the worst outcome among all the different types of ICI-induced encephalitis syndromes. Clinical presentation and systematic measurement of autoantibodies will be a helpful guide for the therapeutic st...

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Section: Resultsmentioning

confidence: 99%

Encephalitis Induced by Immune Checkpoint Inhibitors

et al. 2021

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“…Of 5682 eligible papers, 26 articles 8–33 reporting 28 patients were included. A detailed PRISMA flowchart, the assessment of the risk of bias for each study, and the estimation of frequency in larger cohorts of patients with neurological irAEs are provided in Supporting Information.…”

Section: Resultsmentioning

confidence: 99%

Autoimmune Movement Disorders Complicating Treatment with Immune Checkpoint Inhibitors

Dinoto,

Trentinaglia,

Carta

et al. 2024

Movement Disord Clin Pract

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BackgroundImmune checkpoint inhibitors (ICI) may trigger autoimmune neurological conditions, including movement disorders (MD).ObjectivesThe aim of this study was to characterize MDs occurring as immune‐related adverse events (irAEs) of ICIs.MethodsA systematic literature review of case reports/series of MDs as irAEs of ICIs was performed.ResultsOf 5682 eligible papers, 26 articles with 28 patients were included. MDs occur as a rare complication of cancer immunotherapy with heterogeneous clinical presentations and in most cases in association with other irAEs. Inflammatory basal ganglia T2/fluid attenuated inversion recovery abnormalities are rarely observed, but brain imaging is frequently unrevealing. Cerebrospinal fluid findings are frequently suggestive of inflammation. Half of cases are associated with a wide range of autoantibodies. Steroids and ICI withdrawal usually lead to improvement, even though some patients experienced relapses or a severe clinical course.ConclusionMDs are a rare complication of ICIs that should be promptly recognized to offer patients a correct diagnosis and treatment.

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“… 38 One case of opsoclonus-myoclonus-ataxia syndrome was reported following ipilimumab and nivolumab therapy, which resolved with systemic corticosteroids and IVIg. 134 …”

Section: Resultsmentioning

confidence: 99%

<p>Neuro-ophthalmic Complications of Immune Checkpoint Inhibitors: A Systematic Review</p>

Yau

Mezey

et al. 2020

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Objective Immune checkpoint inhibitors (ICIs) are novel cancer therapies that may be associated with immune-related adverse events (IRAEs) and come to the attention of neuro-ophthalmologists. This systematic review aims to synthesize the reported ICI-associated IRAEs relevant to neuro-ophthalmologists to help in the diagnosis and management of these conditions. Methods A systematic review of the literature indexed by MEDLINE, Embase, CENTRAL, and Web of Science databases was searched from inception to May 2020. Reporting followed the Preferred Reporting Items for Systematic Review and Meta-analysis (PRISMA) guidelines. Primary studies on ICIs and neuro-ophthalmic complications were included. Outcomes included number of cases and incidence of neuro-ophthalmic IRAEs. Results Neuro-ophthalmic complications of ICIs occurred in 0.46% of patients undergoing ICI and may affect the afferent and efferent visual systems. Afferent complications include optic neuritis (12.8%), neuroretinitis (0.9%), and giant cell arteritis (3.7%). Efferent complications include myasthenia gravis (MG) (45.0%), thyroid-like eye disease (11.9%), orbital myositis (13.8%), general myositis with ptosis (7.3%), internuclear ophthalmoplegia (0.9%), opsoclonus-myoclonus-ataxia syndrome (0.9%), and oculomotor nerve palsy (0.9%). Pembrolizumab was the most common causative agent for neuro-ophthalmic complications (32.1%). Mortality was highest for MG (19.8%). Most patients (79.8%) experienced improvement or complete resolution of neuro-ophthalmic symptoms due to cessation of ICI and immunosuppression with systemic corticosteroids. Conclusion While incidence of neuro-ophthalmic IRAEs is low, clinicians involved in the care of cancer patients must be aware of their presentation to facilitate prompt recognition and management. Collaboration between oncology and neuro-ophthalmology teams is required to effectively manage patients and reduce morbidity and mortality.

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Ipilimumab/Nivolumab-related Opsoclonus-Myoclonus-Ataxia Syndrome Variant in a Patient with Malignant Pleural Mesothelioma

Abstract: A variant of opsoclonus-myoclonus-ataxia syndrome may occur following treatment with ipilimumab and nivolumab.

Cited by 18 publications

References 7 publications

Encephalitis Induced by Immune Checkpoint Inhibitors

Encephalitis Induced by Immune Checkpoint Inhibitors

Autoimmune Movement Disorders Complicating Treatment with Immune Checkpoint Inhibitors

<p>Neuro-ophthalmic Complications of Immune Checkpoint Inhibitors: A Systematic Review</p>

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