Song Yanna, Liu Shan, Song Lei, Chen Huaqiu, Bai Miaoshui, Yan Jinhua, Luo Tianfei, Liu Kangding, Sun Li, Zhao Yang
Department of Neurology and Neuroscience Center, The First Hospital of Jilin University, Changchun, China.
Front Neurol. 2021 Apr 20;12:565387. doi: 10.3389/fneur.2021.565387. eCollection 2021.
Kikuchi-Fujimoto disease (KFD), also known as histiocytic necrotizing lymphadenitis, is a rare, benign, self-limiting disease characterized by local lymphadenopathy. Central nervous system involvement in KFD is extremely rare and remains a diagnostic challenge. Only 41 cases of aseptic meningitis associated with KFD have been reported worldwide, with just four cases (including our case) of KFD with meningitis as the first symptom. We report a case of KFD accompanied by aseptic meningitis with severely high intracranial pressure (400 mmHO), increased white blood cell count (56 × 10/L), and moderately elevated protein level (0.52 g/L). This case is unique in the delayed appearance of lymphadenopathy. After 1 month of treatment with steroids, fever, headache, and lymphadenopathy gradually disappeared, and the result of cerebrospinal fluid examination gradually became normal. In conclusion, based on our case findings and our literature review on KFD with aseptic meningitis, a diagnosis of KFD should be considered when delayed appearance of lymphadenopathy is observed in patients with aseptic meningitis.
菊池-藤本病（KFD），又称组织细胞性坏死性淋巴结炎，是一种罕见的、良性的、自限性疾病，其特征为局部淋巴结病。菊池-藤本病累及中枢神经系统极为罕见，仍然是一个诊断难题。全球仅报道了41例与菊池-藤本病相关的无菌性脑膜炎病例，仅有4例（包括我们的病例）以脑膜炎为首发症状的菊池-藤本病。我们报告1例伴有无菌性脑膜炎的菊池-藤本病病例，其颅内压严重升高（400 mmHO），白细胞计数增加（56×10/L），蛋白水平中度升高（0.52 g/L）。该病例的独特之处在于淋巴结病出现延迟。经类固醇治疗1个月后，发热、头痛和淋巴结病逐渐消失，脑脊液检查结果逐渐恢复正常。总之，根据我们的病例发现以及对菊池-藤本病合并无菌性脑膜炎的文献回顾，对于出现无菌性脑膜炎且淋巴结病出现延迟的患者，应考虑菊池-藤本病的诊断。
