关键词: Anti-AMPAR encephalitis Case report Cytoreduction surgery Ovarian cancer Paraneoplastic syndrome

Mesh : Female Humans Middle Aged Cytoreduction Surgical Procedures Breast Neoplasms Ovarian Neoplasms / complications surgery Encephalitis

来  源:   DOI:10.1186/s12905-023-02636-1   PDF(Pubmed)

Abstract:
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.
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.
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.
摘要:
背景:抗α-氨基-3-羟基-5-甲基-4-异恶唑丙酸受体(AMPAR)脑炎,一种罕见的自身免疫性脑炎(AE)亚型,经常发现与胸腺瘤等肿瘤有关,肺癌,卵巢肿瘤,和乳腺癌,肿瘤通常是在脑炎开始后的筛查过程中检测到的。肿瘤被认为是AE的触发因素,但机制尚不清楚。
方法:一名53岁女性在高级别浆液性卵巢癌原发性细胞减灭术后两天出现短期记忆丧失(HGSOC,图IC3阶段)。基于细胞的测定发现AMPARCluA2IgG在血清(1:3.2)和脑脊液(1:32)中均为阳性。此外,在卵巢肿瘤组织石蜡切片中也发现轻度AMPARGluA1和强GluA2阳性表达,表明卵巢细胞减灭术可能刺激受体抗原释放到循环系统中。病人的病情在两周内恶化,发展意识和自主神经功能障碍,导致ICU入院。口服类固醇,静脉注射免疫球蛋白,血浆置换,利妥昔单抗治疗,3个月后患者的意识明显改善。
结论:我们介绍了第一例抗AMPAR脑炎发生在原发性细胞减灭术后的HGSOC患者,并检索了副肿瘤抗AMPAR脑炎病例(n=66)。妇科医生应注意卵巢肿瘤切除后不久出现认知功能障碍或精神症状的患者,并始终将AE纳入鉴别诊断。
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