A case report of neuromyelitis optica spectrum disorder induced by pembrolizumab treatment for lung adenocarcinoma: a clinical and immunohistochemical study
Abstract Background We report a case of neuromyelitis optica spectrum disorders (NMOSD), who developed after the pembrolizumab treatment, an immune checkpoint inhibitor, against lung adenocarcinoma. The present case is discussed with the lung adenocarcinoma specimen which was stained by aquaporin-4...
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| Format: | Article |
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BMC
2022-12-01
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| Series: | BMC Neurology |
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| Online Access: | https://doi.org/10.1186/s12883-022-02987-6 |
| _version_ | 1811198368639090688 |
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| author | Shigeki Hirano Akira Kojima Yoko Nakayama Takahiro Takeda Takashi Kishimoto Toshiyuki Takahashi Satoshi Kuwabara Masahiro Mori |
| author_facet | Shigeki Hirano Akira Kojima Yoko Nakayama Takahiro Takeda Takashi Kishimoto Toshiyuki Takahashi Satoshi Kuwabara Masahiro Mori |
| author_sort | Shigeki Hirano |
| collection | DOAJ |
| description | Abstract Background We report a case of neuromyelitis optica spectrum disorders (NMOSD), who developed after the pembrolizumab treatment, an immune checkpoint inhibitor, against lung adenocarcinoma. The present case is discussed with the lung adenocarcinoma specimen which was stained by aquaporin-4 (AQP4) and with literature review of NMOSD linked to immune checkpoint inhibitors. Case presentation A 62-year-old Japanese man presented with acute diencephalic syndrome, left optic neuritis, and myelitis 5 months after initiation of pembrolizumab treatment for lung adenocarcinoma. He was diagnosed with NMOSD based on serum anti-aquaporin-4 (AQP4) antibody positivity. Immunohistochemistry of lung biopsy samples showed AQP4 expression on CD68+ cells. This is the fifth reported case of AQP4+ NMOSD triggered by an immune checkpoint inhibitor and the first with a brain lesion. Four out of five NMOSD cases, including the present case and one case with lung metastasis, had lung cancer. Conclusions Immune checkpoint inhibitors may trigger AQP4+ NMOSD owing to their molecular similarity to AQP4 expressed in lung and glial tissues. Prompt brain/spinal cord imaging and anti-AQP4 antibody testing may facilitate early diagnosis of immune-mediated adverse event in central nervous system associated with immune checkpoint inhibitors. |
| first_indexed | 2024-04-12T01:29:37Z |
| format | Article |
| id | doaj.art-6d44f9c4c5644eb0af0748cf8989a0df |
| institution | Directory Open Access Journal |
| issn | 1471-2377 |
| language | English |
| last_indexed | 2024-04-12T01:29:37Z |
| publishDate | 2022-12-01 |
| publisher | BMC |
| record_format | Article |
| series | BMC Neurology |
| spelling | doaj.art-6d44f9c4c5644eb0af0748cf8989a0df2022-12-22T03:53:31ZengBMCBMC Neurology1471-23772022-12-012211510.1186/s12883-022-02987-6A case report of neuromyelitis optica spectrum disorder induced by pembrolizumab treatment for lung adenocarcinoma: a clinical and immunohistochemical studyShigeki Hirano0Akira Kojima1Yoko Nakayama2Takahiro Takeda3Takashi Kishimoto4Toshiyuki Takahashi5Satoshi Kuwabara6Masahiro Mori7Department of Neurology, Graduate School of Medicine, Chiba UniversityDepartment of Internal Medicine, Funabashi Central HospitalDepartment of Ophthalmology, Funabashi Central HospitalDepartment of Neurology, National Hospital Organization Chiba-Higashi HospitalDepartment of Molecular Pathology, Graduate School of Medicine, Chiba UniversityDepartment of Neurology, Tohoku University Graduate School of MedicineDepartment of Neurology, Graduate School of Medicine, Chiba UniversityDepartment of Neurology, Graduate School of Medicine, Chiba UniversityAbstract Background We report a case of neuromyelitis optica spectrum disorders (NMOSD), who developed after the pembrolizumab treatment, an immune checkpoint inhibitor, against lung adenocarcinoma. The present case is discussed with the lung adenocarcinoma specimen which was stained by aquaporin-4 (AQP4) and with literature review of NMOSD linked to immune checkpoint inhibitors. Case presentation A 62-year-old Japanese man presented with acute diencephalic syndrome, left optic neuritis, and myelitis 5 months after initiation of pembrolizumab treatment for lung adenocarcinoma. He was diagnosed with NMOSD based on serum anti-aquaporin-4 (AQP4) antibody positivity. Immunohistochemistry of lung biopsy samples showed AQP4 expression on CD68+ cells. This is the fifth reported case of AQP4+ NMOSD triggered by an immune checkpoint inhibitor and the first with a brain lesion. Four out of five NMOSD cases, including the present case and one case with lung metastasis, had lung cancer. Conclusions Immune checkpoint inhibitors may trigger AQP4+ NMOSD owing to their molecular similarity to AQP4 expressed in lung and glial tissues. Prompt brain/spinal cord imaging and anti-AQP4 antibody testing may facilitate early diagnosis of immune-mediated adverse event in central nervous system associated with immune checkpoint inhibitors.https://doi.org/10.1186/s12883-022-02987-6Neuromyelitis optica spectrum disordersImmune checkpoint inhibitorPembrolizumabCase reportAquaporin-4 |
| spellingShingle | Shigeki Hirano Akira Kojima Yoko Nakayama Takahiro Takeda Takashi Kishimoto Toshiyuki Takahashi Satoshi Kuwabara Masahiro Mori A case report of neuromyelitis optica spectrum disorder induced by pembrolizumab treatment for lung adenocarcinoma: a clinical and immunohistochemical study BMC Neurology Neuromyelitis optica spectrum disorders Immune checkpoint inhibitor Pembrolizumab Case report Aquaporin-4 |
| title | A case report of neuromyelitis optica spectrum disorder induced by pembrolizumab treatment for lung adenocarcinoma: a clinical and immunohistochemical study |
| title_full | A case report of neuromyelitis optica spectrum disorder induced by pembrolizumab treatment for lung adenocarcinoma: a clinical and immunohistochemical study |
| title_fullStr | A case report of neuromyelitis optica spectrum disorder induced by pembrolizumab treatment for lung adenocarcinoma: a clinical and immunohistochemical study |
| title_full_unstemmed | A case report of neuromyelitis optica spectrum disorder induced by pembrolizumab treatment for lung adenocarcinoma: a clinical and immunohistochemical study |
| title_short | A case report of neuromyelitis optica spectrum disorder induced by pembrolizumab treatment for lung adenocarcinoma: a clinical and immunohistochemical study |
| title_sort | case report of neuromyelitis optica spectrum disorder induced by pembrolizumab treatment for lung adenocarcinoma a clinical and immunohistochemical study |
| topic | Neuromyelitis optica spectrum disorders Immune checkpoint inhibitor Pembrolizumab Case report Aquaporin-4 |
| url | https://doi.org/10.1186/s12883-022-02987-6 |
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