Anti-GAD65 Antibody-Associated Autoimmune Encephalitis With Predominant Cerebellar Involvement Following Toripalimab Treatment: A Case Report of a Novel irAE of Toripalimab

Zhou, Huanyu; Xie, Xiaoxi; Zhang, Tianyu; Yang, Menghan; Zhou, Dong; Yang, Tianhua

doi:10.3389/fimmu.2022.850540

Cited by 8 publications

(3 citation statements)

References 38 publications

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“…Therefore, she was diagnosed with toripalimab-related n-irAE accompanied with multiple demyelinating imaging changes, in combination with asymptomatic myocarditis and hypothyroidism. This n-irAE displayed a different timing of onset as well as the symptoms and we found that intracranial imaging changed markedly compared to the case of early-onset cognitive dysfunction along with overt signs and symptoms of cerebellar involvement as reported earlier by Zhou et al [ 22 ]. In an analysis of 347 patients treated with pembrolizumab or nivolumab, the median time to onset of n-irAEs was 5.5 cycles.…”

Section: Discussionsupporting

confidence: 68%

Stuttering as a signal of encephalopathy associated with toripalimab in a pancreatic ductal adenocarcinoma patient: a case report

Chen²,

Cheng³

et al. 2023

BMC Neurol

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Background Immune checkpoint inhibitor (ICI) combined with chemotherapy has exhibited promising results in small sample studies of pancreatic cancer patients. The efficacy of toripalimab, a programmed cell death protein 1 (PD-1) monoclonal antibody has been explored in the previous studies and it was established that immune-related adverse events (irAEs) associated with administration of this drug deserve proper attention and adequate management. Case presentation A 43-year-old female patient with advanced pancreatic ductal adenocarcinoma (PDAC) was treated with toripalimab in combination with gemcitabine and nab-paclitaxel (T-GA) as the first-line treatment. She developed immune-related encephalopathy with stuttering as the main clinical symptom and Magnetic resonance imaging (MRI) showed multiple cerebral white matter demyelination changes, concomitant with asymptomatic cardiac enzyme elevation and hypothyroidism. The symptoms resolved after the discontinuation of toripalimab and corticosteroid treatment. Conclusions Stuttering might be an early sign of neurotoxicity which can be easily neglected during the treatment. These findings provide guidance for the identification of these rare and occult neurological irAEs (n-irAEs) in the clinical practice.

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Section: Discussionsupporting

confidence: 68%

Stuttering as a signal of encephalopathy associated with toripalimab in a pancreatic ductal adenocarcinoma patient: a case report

Chen²,

Cheng³

et al. 2023

BMC Neurol

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“…Encephalitis is a common adverse reaction of the nervous system and has been reported to be associated with PD-1 inhibitor immunotherapy in the clinic ( Williams et al, 2016 ). Patients often have elevated levels of autoantibodies, but lack specificity, such as Anti-Hu ( Papadopoulos et al, 2018 ), Anti-PCA-2 ( Segal et al, 2022 ), Anti-GAD65 ( Chung et al, 2020 ; Zhou et al, 2022 ) and Anti-Glial fibrillary acidic protein (GFAP; Kapadia et al, 2020 ). When diagnosing CNS encephalitis, it is necessary to differentiate it from paraneoplastic neurological diseases (PNS) by excluding infectious diseases, tumor metastasis, etc., and distinguishing it based on the time of onset of encephalitis symptoms, whether they appear after PD-1 treatment, and the effectiveness of immunosuppressive therapy.…”

Section: Common Symptoms Of Naementioning

confidence: 99%

Neurological adverse events associated with PD-1/PD-L1 immune checkpoint inhibitors

Zhou

2023

Front. Neurosci.

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Immunotherapy is a promising method for cancer treatment. Among them, immune checkpoint inhibitors targeting PD-1/PD-L1 are increasingly used for certain cancers. However, with the widespread use of such drugs, reports of immune-related adverse events (irAEs) are also increasing. Neurological adverse events (nAEs) are one of the irAEs that affect the peripheral and central nervous systems. They are characterized by low incidence, hard to diagnose, and life-threatening risks, which have a significant impact on the prognosis of patients. Biomarker-based early diagnosis and subsequent treatment strategies are worthy of attention, and comprehensive management of irAEs is important for optimizing patients’ quality of life and long-term outcomes. In this review, we summarized the mechanisms, common symptoms, early biomarkers, treatments, and future research directions of nAEs, in order to provide a comprehensive overview of immune checkpoint inhibitor-related nAEs targeting PD-1/PD-L1.

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“…Ocular motor abnormalities have also been rarely described during treatment with ICIs [9][10][11], including vertical or horizontal nystagmus associated with acute cerebellar ataxia or brainstem encephalitis.…”

Section: Introductionmentioning

confidence: 99%

A Case of Anti-Ma2 Encephalitis Presenting with Pendular Torsional Nystagmus

Vaghi,

Vegezzi,

Bini

et al. 2023

Cerebellum

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Antibodies against the neuronal protein Ma2 have been reported in a peculiar form of paraneoplastic encephalitis with prominent involvement of the limbic, brainstem, and diencephalic structures and usually associated with germ cell testicular, lung, or breast cancer. The diagnosis is frequently challenged by atypical clinical manifestations including parkinsonism, sleep disturbances, hypothalamic-pituitary dysfunctions, and motor neuron-like syndrome. In recent years, the advent of monoclonal antibodies targeting immune checkpoints has deeply changed the treatment of different tumors, especially melanoma and lung cancer. However, given their nature, an increasing number of neurological immune-related adverse events, including ocular motor abnormalities, have been described. Here, we report a woman with advanced non-small cell lung cancer treated with anti-PD-L1 durvalumab, presenting with an isolated pendular torsional nystagmus, in association with anti-Ma2 antibodies. This peculiar case widens our knowledge on the clinical presentation of anti-Ma2 encephalitis associated with checkpoint inhibitors.

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Anti-GAD65 Antibody-Associated Autoimmune Encephalitis With Predominant Cerebellar Involvement Following Toripalimab Treatment: A Case Report of a Novel irAE of Toripalimab

Cited by 8 publications

References 38 publications

Stuttering as a signal of encephalopathy associated with toripalimab in a pancreatic ductal adenocarcinoma patient: a case report

Stuttering as a signal of encephalopathy associated with toripalimab in a pancreatic ductal adenocarcinoma patient: a case report

Neurological adverse events associated with PD-1/PD-L1 immune checkpoint inhibitors

A Case of Anti-Ma2 Encephalitis Presenting with Pendular Torsional Nystagmus

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