Anti-alpha-amino-3-hydroxy-5-methyl-4-isoxazolepropionic acid receptor encephalitis developed after ovarian cancer cytoreduction surgery: a case report and literature review
Abstract Background Anti-alpha-amino-3-hydroxy-5-methyl-4-isoxazolepropionic acid receptor (AMPAR) encephalitis, a rare subtype of autoimmune encephalitis (AE), is often found associated with tumors such as thymoma, lung cancer, ovarian tumors, and breast cancer, and the tumors were generally detect...
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| Format: | Article |
| Language: | English |
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BMC
2023-09-01
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| Series: | BMC Women's Health |
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| Online Access: | https://doi.org/10.1186/s12905-023-02636-1 |
| _version_ | 1797556783763423232 |
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| author | Yue Huang Muke Zhou Jing Zhou Bo Wu Xi Yang Wenjiao Min Zhengyu Li |
| author_facet | Yue Huang Muke Zhou Jing Zhou Bo Wu Xi Yang Wenjiao Min Zhengyu Li |
| author_sort | Yue Huang |
| collection | DOAJ |
| description | Abstract Background Anti-alpha-amino-3-hydroxy-5-methyl-4-isoxazolepropionic acid receptor (AMPAR) encephalitis, a rare subtype of autoimmune encephalitis (AE), is often found associated with tumors such as thymoma, lung cancer, ovarian tumors, and breast cancer, and the tumors were generally detected during the screening process after the encephalitis initiated. The tumor is considered a trigger of AE, but the mechanism remains unclear. Case Presentation A 53-year-old woman presented short-term memory loss two days after the primary cytoreduction for high-grade serous ovarian cancer (HGSOC, FIGO stage IC3). Cell-based assay found AMPAR CluA2 IgG positive in both serum (1:3.2) and cerebrospinal fluid (1:32). Moreover, mild AMPAR GluA1 and strong GluA2 expressions were also found positive in the paraffin sections of ovarian tumor tissue, indicating the ovarian cytoreduction surgery might stimulate the release of receptor antigens into the circulation system. The patient’s condition deteriorated within two weeks, developing consciousness and autonomic dysfunction, leading to ICU admission. With oral steroids, intravenous immunoglobulin, plasmapheresis, and rituximab treatment, the patient’s consciousness markedly improved after three months. Conclusion We presented the first case of anti-AMPAR encephalitis developed right after the primary cytoreduction of a patient with HGSOC and retrieved paraneoplastic anti-AMPAR encephalitis cases (n = 66). Gynecologists should pay attention to patients who develop cognitive dysfunction or psychiatric symptoms shortly after the ovarian tumor resection and always include AE in the differentiation diagnosis. |
| first_indexed | 2024-03-10T17:06:52Z |
| format | Article |
| id | doaj.art-6570b6bd563f4d1388d0bb226a7e9f7e |
| institution | Directory Open Access Journal |
| issn | 1472-6874 |
| language | English |
| last_indexed | 2024-03-10T17:06:52Z |
| publishDate | 2023-09-01 |
| publisher | BMC |
| record_format | Article |
| series | BMC Women's Health |
| spelling | doaj.art-6570b6bd563f4d1388d0bb226a7e9f7e2023-11-20T10:46:21ZengBMCBMC Women's Health1472-68742023-09-012311810.1186/s12905-023-02636-1Anti-alpha-amino-3-hydroxy-5-methyl-4-isoxazolepropionic acid receptor encephalitis developed after ovarian cancer cytoreduction surgery: a case report and literature reviewYue Huang0Muke Zhou1Jing Zhou2Bo Wu3Xi Yang4Wenjiao Min5Zhengyu Li6Department of Gynecology and Obstetrics, West China Second University Hospital, Sichuan UniversityDepartment of Neurology, West China Hospital, Sichuan UniversityDepartment of Gynecology and Obstetrics, Chengdu Fifth People’s HospitalDepartment of Neurology, West China Hospital, Sichuan UniversityDepartment of Anesthesiology, West China Hospital, Sichuan universityDepartment of Psychosomatic Medicine, Sichuan Academy of Medical Science & Sichuan Provincial People’s HospitalDepartment of Gynecology and Obstetrics, West China Second University Hospital, Sichuan UniversityAbstract Background Anti-alpha-amino-3-hydroxy-5-methyl-4-isoxazolepropionic acid receptor (AMPAR) encephalitis, a rare subtype of autoimmune encephalitis (AE), is often found associated with tumors such as thymoma, lung cancer, ovarian tumors, and breast cancer, and the tumors were generally detected during the screening process after the encephalitis initiated. The tumor is considered a trigger of AE, but the mechanism remains unclear. Case Presentation A 53-year-old woman presented short-term memory loss two days after the primary cytoreduction for high-grade serous ovarian cancer (HGSOC, FIGO stage IC3). Cell-based assay found AMPAR CluA2 IgG positive in both serum (1:3.2) and cerebrospinal fluid (1:32). Moreover, mild AMPAR GluA1 and strong GluA2 expressions were also found positive in the paraffin sections of ovarian tumor tissue, indicating the ovarian cytoreduction surgery might stimulate the release of receptor antigens into the circulation system. The patient’s condition deteriorated within two weeks, developing consciousness and autonomic dysfunction, leading to ICU admission. With oral steroids, intravenous immunoglobulin, plasmapheresis, and rituximab treatment, the patient’s consciousness markedly improved after three months. Conclusion We presented the first case of anti-AMPAR encephalitis developed right after the primary cytoreduction of a patient with HGSOC and retrieved paraneoplastic anti-AMPAR encephalitis cases (n = 66). Gynecologists should pay attention to patients who develop cognitive dysfunction or psychiatric symptoms shortly after the ovarian tumor resection and always include AE in the differentiation diagnosis.https://doi.org/10.1186/s12905-023-02636-1Anti-AMPAR encephalitisParaneoplastic syndromeOvarian cancerCytoreduction surgeryCase report |
| spellingShingle | Yue Huang Muke Zhou Jing Zhou Bo Wu Xi Yang Wenjiao Min Zhengyu Li Anti-alpha-amino-3-hydroxy-5-methyl-4-isoxazolepropionic acid receptor encephalitis developed after ovarian cancer cytoreduction surgery: a case report and literature review BMC Women's Health Anti-AMPAR encephalitis Paraneoplastic syndrome Ovarian cancer Cytoreduction surgery Case report |
| title | Anti-alpha-amino-3-hydroxy-5-methyl-4-isoxazolepropionic acid receptor encephalitis developed after ovarian cancer cytoreduction surgery: a case report and literature review |
| title_full | Anti-alpha-amino-3-hydroxy-5-methyl-4-isoxazolepropionic acid receptor encephalitis developed after ovarian cancer cytoreduction surgery: a case report and literature review |
| title_fullStr | Anti-alpha-amino-3-hydroxy-5-methyl-4-isoxazolepropionic acid receptor encephalitis developed after ovarian cancer cytoreduction surgery: a case report and literature review |
| title_full_unstemmed | Anti-alpha-amino-3-hydroxy-5-methyl-4-isoxazolepropionic acid receptor encephalitis developed after ovarian cancer cytoreduction surgery: a case report and literature review |
| title_short | Anti-alpha-amino-3-hydroxy-5-methyl-4-isoxazolepropionic acid receptor encephalitis developed after ovarian cancer cytoreduction surgery: a case report and literature review |
| title_sort | anti alpha amino 3 hydroxy 5 methyl 4 isoxazolepropionic acid receptor encephalitis developed after ovarian cancer cytoreduction surgery a case report and literature review |
| topic | Anti-AMPAR encephalitis Paraneoplastic syndrome Ovarian cancer Cytoreduction surgery Case report |
| url | https://doi.org/10.1186/s12905-023-02636-1 |
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