Autoimmune cerebellar hypermetabolism: Report of three cases and literature overview

Courssou, J.-B. Brunet de; Castilla-Lievre, M.A.; Maillot, Jean; Bréchemier, Marie-Laure; Ohlmann, C.; Sallansonnet-Froment, M.; Tafani, C.; Psimaras, Dimitri; Ricard, Damien; Bompaire, Flavie; Taifas, Irina

doi:10.1016/j.neurol.2021.07.018

Cited by 3 publications

(4 citation statements)

References 42 publications

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“…tients. Cerebellar MRI showed T2/fluid attenuated inversion recovery (FLAIR) hyperintensities (n = 5), enhancement (n = 2), edema (n = 2) or atrophy (n = 2), whilst it was unremarkable in the other patients.Brain MRI frequently demonstrated extracerebellar abnormalities usually consistent with the reported clinical symptoms, such as mesial temporal lobe involvement (n = 3), cranial nerve involvement (n = 3), cortical ribbon interruption (n = 1), small inflammatory cortical lesions (n = 1), medulla/pulvinar T2-weighted hyperintensity (n = 1), leptomeningeal enhancement (n = 1), generalized atrophy (n = 1) and unspecific white matter T2-weighted hyperintensities (n = 1) 18. F-fluorodeoxyglucose positron emission tomography and [ 123 I]N-isopropylp-iodoamphetamine single photon emission computed tomography scans were performed in a minority of patients and demonstrated hypermetabolism/hyperperfusion of the cerebellar vermis (n = 2), reduced perfusion of occipital and cerebellar parenchyma (n = 1), diffuse hypometabolism with frontotemporal atrophy (n = 1) and hypometabolism of temporal poles (n = 1).…”

supporting

confidence: 82%

“…In this setting, inflammatory CSF findings are of utmost importance to confirm the diagnosis and to rule out meningeal carcinomatosis [2, 23]. Finally, in very few cases, nuclear medicine imaging supported the diagnosis by demonstrating cerebellar hypermetabolism [12, 18].…”

Section: Discussionmentioning

confidence: 99%

“…Of these, 61 full-text articles were assessed and 27 of them fulfilled inclusion criteria. Five more records were included from other sources, and thus 32 articles reporting 46 patients were included in the qualitative synthesis ( [10][11][12][13][14][15][16][17][18][19][20][21][22] and Data S1). The detailed PRISMA flow chart of the systematic review is reported in Figure 1, whilst relevant data of the included patients are summarized in Table 1.…”

Section: Re Sultsmentioning

confidence: 99%

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Cerebellar involvement associated with immune checkpoint inhibitors: A systematic review

Dinoto

Mantovani

Ferrari

et al. 2022

Euro J of Neurology

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Background and purpose Immune checkpoint inhibitors (ICIs) targeting programmed death receptor 1 (PD‐1), cytotoxic T‐lymphocyte‐associated‐4 (CTLA‐4) and programmed cell death ligand 1 can be associated with immune‐related adverse events (iRAEs). Amongst neurological iRAEs, cerebellar involvement seems to be rare and currently lacks a proper characterization. The aim of this study was to phenotype cerebellar iRAEs. Methods A systematic review was performed according to PRISMA guidelines including reported patients with cerebellar involvement related to ICIs and with available individual data. Results After screening 2765 records, 32 studies with 46 patients were included. Median age was 63 years (20–82), and most patients were male (63.0%). Isolated cerebellitis was observed in 32.6% of cases, whilst the remaining cases had “cerebellitis plus”, mostly associated with encephalitis/encephalopathy. Associated tumors included most frequently lung cancer, melanoma and Merkel cell carcinoma. PD‐1 inhibitor was the most administered treatment (n = 29, 64.4%), whilst exposure to CTLA‐4 inhibitor was rare (n = 2, 4.5%). Magnetic resonance imaging was abnormal in 43.2% of patients and inflammatory cerebrospinal fluid findings were frequently observed. Autoantibodies were detected in 61.9% of patients and included novel reactivities. Amongst treatment strategies, the most common were steroids (n = 36) and ICI discontinuation (n = 28, 90.3%). Relapses were reported in 10% of patients. Most patients showed improvement/remission (n = 31) but, at last follow‐up, 12 had died. Isolated cerebellitis versus cerebellitis‐plus differed in terms of outcomes, whilst seropositive versus seronegative patients had distinct tumor associations. Discussion Cerebellar iRAEs are usually multifocal, have heterogeneous tumor associations, are most associated with PD‐1 inhibitor exposure and are related to autoantibodies, including novel reactivities.

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supporting

confidence: 82%

Section: Discussionmentioning

confidence: 99%

Section: Re Sultsmentioning

confidence: 99%

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Cerebellar involvement associated with immune checkpoint inhibitors: A systematic review

Dinoto

Mantovani

Ferrari

et al. 2022

Euro J of Neurology

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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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Immune-globulin/nivolumab

2022

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Autoimmune cerebellar hypermetabolism: Report of three cases and literature overview

Cited by 3 publications

References 42 publications

Cerebellar involvement associated with immune checkpoint inhibitors: A systematic review

Cerebellar involvement associated with immune checkpoint inhibitors: A systematic review

Autoimmune Movement Disorders Complicating Treatment with Immune Checkpoint Inhibitors

Immune-globulin/nivolumab

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