Department of Neurology, Akita University Graduate School of Medicine, Akita, Japan.
Department of Molecular and Tumour Pathology, Akita University Graduate School of Medicine, Akita, Japan.
Front Immunol. 2023 Dec 8;14:1188154. doi: 10.3389/fimmu.2023.1188154. eCollection 2023.
Ophelia syndrome is paraneoplastic limbic encephalitis (PLE) with Hodgkin lymphoma. Some Ophelia syndrome patients have been reported as testing positive for anti-metabotropic glutamate receptor 5 (mGluR5) antibodies. However, we experienced a case of anti-mGluR5 antibody-negative Ophelia syndrome. The type of onset, neurological symptoms, and imaging as well as electroencephalographic findings were like previous reports except for a normal cell count in cerebrospinal fluid (CSF). Unfortunately, a lymph node biopsy failed and could not diagnose the patient before death because steroid treatment for limbic encephalitis had shrunk lymph nodes. We believe it is essential to accumulate cases of this syndrome and clarify the association between PLE and Hodgkin lymphoma so chemotherapy can be initiated even if malignant lymphoma cannot be pathologically proven or when antibodies cannot be measured or are negative.
奥菲利娅综合征是副肿瘤性边缘叶脑炎(PLE)伴霍奇金淋巴瘤。已有报道称部分奥菲利娅综合征患者抗代谢型谷氨酸受体 5(mGluR5)抗体检测呈阳性。然而,我们遇到了一例抗 mGluR5 抗体阴性的奥菲利娅综合征。该病例的发病类型、神经症状、影像学和脑电图检查均与既往报道相似,除了脑脊液(CSF)细胞计数正常。不幸的是,由于类固醇治疗边缘性脑炎使淋巴结缩小,淋巴结活检未能进行,在患者死亡前也无法确诊。我们认为,有必要积累此类综合征的病例,并阐明 PLE 与霍奇金淋巴瘤之间的关系,以便即使无法通过病理证实恶性淋巴瘤,或者无法检测或抗体阴性时,也可以启动化疗。