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Immune Checkpoint Inhibitor-Associated Kelch-Like Protein-11 IgG Brainstem Encephalitis.
Aboseif, Albert; Vorasoot, Nisa; Pinto, Marcus V; Guo, Yong; Hasan, Shemonti; Zekeridou, Anastasia; Chen, John J; Dubey, Divyanshu.
Afiliação
  • Aboseif A; From the Center for Multiple Sclerosis and Autoimmune Neurology (A.A., N.V., Y.G., S.H., A.Z., J.J.C., D.D.), Department of Neurology; Department of Laboratory Medicine and Pathology (N.V., Y.G., A.Z., D.D.); Department of Neurology (N.V.), Mayo Clinic College of Medicine, Rochester, MN; Division of
  • Vorasoot N; From the Center for Multiple Sclerosis and Autoimmune Neurology (A.A., N.V., Y.G., S.H., A.Z., J.J.C., D.D.), Department of Neurology; Department of Laboratory Medicine and Pathology (N.V., Y.G., A.Z., D.D.); Department of Neurology (N.V.), Mayo Clinic College of Medicine, Rochester, MN; Division of
  • Pinto MV; From the Center for Multiple Sclerosis and Autoimmune Neurology (A.A., N.V., Y.G., S.H., A.Z., J.J.C., D.D.), Department of Neurology; Department of Laboratory Medicine and Pathology (N.V., Y.G., A.Z., D.D.); Department of Neurology (N.V.), Mayo Clinic College of Medicine, Rochester, MN; Division of
  • Guo Y; From the Center for Multiple Sclerosis and Autoimmune Neurology (A.A., N.V., Y.G., S.H., A.Z., J.J.C., D.D.), Department of Neurology; Department of Laboratory Medicine and Pathology (N.V., Y.G., A.Z., D.D.); Department of Neurology (N.V.), Mayo Clinic College of Medicine, Rochester, MN; Division of
  • Hasan S; From the Center for Multiple Sclerosis and Autoimmune Neurology (A.A., N.V., Y.G., S.H., A.Z., J.J.C., D.D.), Department of Neurology; Department of Laboratory Medicine and Pathology (N.V., Y.G., A.Z., D.D.); Department of Neurology (N.V.), Mayo Clinic College of Medicine, Rochester, MN; Division of
  • Zekeridou A; From the Center for Multiple Sclerosis and Autoimmune Neurology (A.A., N.V., Y.G., S.H., A.Z., J.J.C., D.D.), Department of Neurology; Department of Laboratory Medicine and Pathology (N.V., Y.G., A.Z., D.D.); Department of Neurology (N.V.), Mayo Clinic College of Medicine, Rochester, MN; Division of
  • Chen JJ; From the Center for Multiple Sclerosis and Autoimmune Neurology (A.A., N.V., Y.G., S.H., A.Z., J.J.C., D.D.), Department of Neurology; Department of Laboratory Medicine and Pathology (N.V., Y.G., A.Z., D.D.); Department of Neurology (N.V.), Mayo Clinic College of Medicine, Rochester, MN; Division of
  • Dubey D; From the Center for Multiple Sclerosis and Autoimmune Neurology (A.A., N.V., Y.G., S.H., A.Z., J.J.C., D.D.), Department of Neurology; Department of Laboratory Medicine and Pathology (N.V., Y.G., A.Z., D.D.); Department of Neurology (N.V.), Mayo Clinic College of Medicine, Rochester, MN; Division of
Neurol Neuroimmunol Neuroinflamm ; 11(3): e200218, 2024 May.
Article em En | MEDLINE | ID: mdl-38484218
ABSTRACT

OBJECTIVES:

Kelch-like protein-11 (KLHL11)-IgG is associated with rhombencephalitis and seminoma. It has not previously been described as a neurologic immune checkpoint inhibitor (ICI)-related adverse event (nirAE) or in association with esophageal adenocarcinoma.

METHODS:

We describe a 61-year-old man with metastatic esophageal adenocarcinoma treated with folinic acid, fluorouracil, oxaliplatin (FOLFOX), and nivolumab, who subsequently developed diplopia, vertigo, and progressive gait ataxia after 8 weeks of treatment.

RESULTS:

Owing to a concern for ICI-associated myasthenia gravis, nivolumab was held and he was treated with prednisone and pyridostigmine. EMG showed no neuromuscular junction dysfunction, and acetylcholine-receptor antibodies were negative. Brain MRI was unrevealing. Murine brain tissue immunofluorescence assay revealed KLHL11-IgG in both serum and CSF, confirmed by cell-based assay. Tumor histopathology demonstrated poorly differentiated, highly proliferative adenocarcinoma with increased mitotic figures and cytoplasmic KLHL11 immunoreactivity. He was initiated on 6 months of cyclophosphamide in addition to FOLFOX for post-ICI-associated KLHL11-IgG rhombencephalitis.

DISCUSSION:

We report KLHL11-IgG rhombencephalitis associated with poorly differentiated esophageal cancer as a novel nirAE. Tumor staining revealed KLHL11 immunoreactivity, supporting a cancer-antigen-driven ICI-associated paraneoplastic syndrome. Recognition of novel nirAEs can expedite treatment and potentially prevent progressive neurologic disability.
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Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Assunto principal: Neoplasias Testiculares / Neoplasias Esofágicas / Adenocarcinoma / Encefalite Limite: Animals / Humans / Male / Middle aged Idioma: En Ano de publicação: 2024 Tipo de documento: Article
Texto completo
Imprimir
XML
PubMed Links
Buscar no Google

Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Assunto principal: Neoplasias Testiculares / Neoplasias Esofágicas / Adenocarcinoma / Encefalite Limite: Animals / Humans / Male / Middle aged Idioma: En Ano de publicação: 2024 Tipo de documento: Article