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Pembrolizumab-Induced Isolated Cranial Neuropathy: A Rare Case Report and Review of Literature

Introduction: Anti-PD1 agents are widely used in the treatment of solid tumors. This has prompted the recognition of a class of immune-related adverse events (irAEs), due to the activation of autoimmune T-cells. Pembrolizumab is an anti-PD1 agent, which has been related to an increased risk of vario...

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Autores principales: Bruno, Francesco, Palmiero, Rosa Antonietta, Ferrero, Bruno, Franchino, Federica, Pellerino, Alessia, Milanesi, Enrica, Soffietti, Riccardo, Rudà, Roberta
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8144636/
https://www.ncbi.nlm.nih.gov/pubmed/34046006
http://dx.doi.org/10.3389/fneur.2021.669493
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author Bruno, Francesco
Palmiero, Rosa Antonietta
Ferrero, Bruno
Franchino, Federica
Pellerino, Alessia
Milanesi, Enrica
Soffietti, Riccardo
Rudà, Roberta
author_facet Bruno, Francesco
Palmiero, Rosa Antonietta
Ferrero, Bruno
Franchino, Federica
Pellerino, Alessia
Milanesi, Enrica
Soffietti, Riccardo
Rudà, Roberta
author_sort Bruno, Francesco
collection PubMed
description Introduction: Anti-PD1 agents are widely used in the treatment of solid tumors. This has prompted the recognition of a class of immune-related adverse events (irAEs), due to the activation of autoimmune T-cells. Pembrolizumab is an anti-PD1 agent, which has been related to an increased risk of various neurological irAE (n-irAEs). Here, we present a rare case of pembrolizumab-induced neuropathy of cranial nerves. Case Report: A 72-year-old patient was diagnosed with a lung adenocarcinoma in February 2018 (EGFR–, ALK–, and PDL1 90%). According to the molecular profile, pembrolizumab was started. After three administrations, the patient developed facial paresis, ptosis, ophthalmoplegia, and dysphonia. As brain metastases and paraneoplastic markers were excluded, a drug-related disorder was suspected and pembrolizumab was discontinued. A nerve conduction study and electromyography excluded signs of neuropathy and myopathy at four limbs, and repetitive nerve stimulation was negative. However, altered blink reflex and nerve facial conduction were consistent with an acute neuropathy of the cranial district. Thus, the patient was treated with two cycles of intravenous immunoglobulins (IVIg), which rapidly allowed improvement of both symptoms and neurophysiological parameters. However, the patient died in October 2018 for a progression of lung tumor. Discussion: Only 16 cases of pembrolizumab-related neuropathies have been described so far. Our case is of particular interest for the isolated involvement of cranial nerves and the prompt response to IVIg. Conclusion: N-irAEs are insidious conditions that require solid knowledge of onco-immunotherapy complications: it is mandatory not to delay any treatment that would potentially modify the course of a neurological complication.
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spelling pubmed-81446362021-05-26 Pembrolizumab-Induced Isolated Cranial Neuropathy: A Rare Case Report and Review of Literature Bruno, Francesco Palmiero, Rosa Antonietta Ferrero, Bruno Franchino, Federica Pellerino, Alessia Milanesi, Enrica Soffietti, Riccardo Rudà, Roberta Front Neurol Neurology Introduction: Anti-PD1 agents are widely used in the treatment of solid tumors. This has prompted the recognition of a class of immune-related adverse events (irAEs), due to the activation of autoimmune T-cells. Pembrolizumab is an anti-PD1 agent, which has been related to an increased risk of various neurological irAE (n-irAEs). Here, we present a rare case of pembrolizumab-induced neuropathy of cranial nerves. Case Report: A 72-year-old patient was diagnosed with a lung adenocarcinoma in February 2018 (EGFR–, ALK–, and PDL1 90%). According to the molecular profile, pembrolizumab was started. After three administrations, the patient developed facial paresis, ptosis, ophthalmoplegia, and dysphonia. As brain metastases and paraneoplastic markers were excluded, a drug-related disorder was suspected and pembrolizumab was discontinued. A nerve conduction study and electromyography excluded signs of neuropathy and myopathy at four limbs, and repetitive nerve stimulation was negative. However, altered blink reflex and nerve facial conduction were consistent with an acute neuropathy of the cranial district. Thus, the patient was treated with two cycles of intravenous immunoglobulins (IVIg), which rapidly allowed improvement of both symptoms and neurophysiological parameters. However, the patient died in October 2018 for a progression of lung tumor. Discussion: Only 16 cases of pembrolizumab-related neuropathies have been described so far. Our case is of particular interest for the isolated involvement of cranial nerves and the prompt response to IVIg. Conclusion: N-irAEs are insidious conditions that require solid knowledge of onco-immunotherapy complications: it is mandatory not to delay any treatment that would potentially modify the course of a neurological complication. Frontiers Media S.A. 2021-05-11 /pmc/articles/PMC8144636/ /pubmed/34046006 http://dx.doi.org/10.3389/fneur.2021.669493 Text en Copyright © 2021 Bruno, Palmiero, Ferrero, Franchino, Pellerino, Milanesi, Soffietti and Rudà. https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Neurology
Bruno, Francesco
Palmiero, Rosa Antonietta
Ferrero, Bruno
Franchino, Federica
Pellerino, Alessia
Milanesi, Enrica
Soffietti, Riccardo
Rudà, Roberta
Pembrolizumab-Induced Isolated Cranial Neuropathy: A Rare Case Report and Review of Literature
title Pembrolizumab-Induced Isolated Cranial Neuropathy: A Rare Case Report and Review of Literature
title_full Pembrolizumab-Induced Isolated Cranial Neuropathy: A Rare Case Report and Review of Literature
title_fullStr Pembrolizumab-Induced Isolated Cranial Neuropathy: A Rare Case Report and Review of Literature
title_full_unstemmed Pembrolizumab-Induced Isolated Cranial Neuropathy: A Rare Case Report and Review of Literature
title_short Pembrolizumab-Induced Isolated Cranial Neuropathy: A Rare Case Report and Review of Literature
title_sort pembrolizumab-induced isolated cranial neuropathy: a rare case report and review of literature
topic Neurology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8144636/
https://www.ncbi.nlm.nih.gov/pubmed/34046006
http://dx.doi.org/10.3389/fneur.2021.669493
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