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[伴有明显小脑受累的军团病:病例报告及文献综述]

[Legionnaires' disease with pronounced cerebellar involvement: case report and literature review].

作者信息

Lu M, Shen N, Zhu H, Yao W Z

机构信息

Department of Respiratory and Critical Care Medicine, Peking University Third Hospital, Beijing 100191, China.

出版信息

Zhonghua Jie He He Hu Xi Za Zhi. 2020 Feb 12;43(2):126-131. doi: 10.3760/cma.j.issn.1001-0939.2020.02.010.

Abstract

To summarize the clinical course, neuroimaging and cerebrospinal fluid (CSF) analyses of cerebellar dysfunction in Legionnaires' disease. A case of Legionnaires' disease with pronounced cerebellar involvement was reported. The related literatures published up to February 2019 were reviewed with "Legionella, legionellosis, legionnaires' disease, cerebellum, cerebellar" as the keywords in CNKI, Wanfang and PubMed databases. A 69-year-old man complained of anorexia and diarrhea for several days. He was subsequently admitted to the hospital after he had fever, ataxia, dysarthria and involuntary tremor. Chest CT revealed right lower lobe pneumonia. Routine urinalysis showed hematuria and proteinuria. Serum alanine transaminase was 52 U/L, creatinine 137 μmol/L, sodium 128 mmol/L, and creatine kinase 6 893 U/L. Cranial CT was normal. Analysis of CSF showed mildly elevated total protein. Legionella colonies isolated from bronchoalveolar lavage fluid was positive by PCR. After initial treatment with moxifloxacin and azithromycin for 7 days, the fever and neurological symptoms persisted. Corticosteroid therapy was administered for 3 days, the fever resolved, whereas the neurological symptoms improved gradually and slowly by 4 weeks of antibiotic therapy. Finally, successive serological test confirmed Legionella pneumophila serogroups 6 and 7 by indirect immunofluorescence. Twenty-one literatures with 23 cases were reviewed, and plus our case, there were a total of 24 cases for analysis. There were 16 males and 8 females, aged from 22 to 71 years. Ataxia and dysarthria were the cerebellar symptoms most frequently reported, occurring in 22 and 18 cases, respectively. All patients had various central and peripheral neuropathies during their illness. Neuroimaging and analysis of CSF was reported in 21 cases. There were no abnormalities in 18 cases of cranial imaging, 1 case with slight hydrocephalus on cranial CT, and 3 cases with hyperintensity in the splenium of corpus callosum on cranial MRI. Eighteen cases of CSF analyses were normal, whereas 1 case with elevated lymphocytes and 3 cases with elevated proteins. Nine cases were eventually identified as Legionella pneumophila serotype 1 by urinary antigen detection, 1 case as Legionella pneumophila serogroups 6 and 7, while the remaining 14 were unknown serotype. Long-term neurologic follow-up showed that 3 cases recovered completely in the first week, while 19 cases improved slowly in the following 3 weeks, and 13 cases had persistent deficits of gait or speech after 3 months. Legionellosis with cerebellar insufficiency is rare. It may be misdiagnosed in the onset of illness. After treatment, there is a trend of slow recovery and neurological symptoms may persist in long-term follow-up.

摘要

总结军团病小脑功能障碍的临床病程、神经影像学及脑脊液(CSF)分析。报告1例有明显小脑受累的军团病病例。以“嗜肺军团菌、军团菌病、军团病、小脑、小脑的”为关键词,检索中国知网、万方数据库及PubMed数据库中截至2019年2月发表的相关文献。一名69岁男性,主诉数日来食欲不振和腹泻。随后,在出现发热、共济失调、构音障碍和不自主震颤后入院。胸部CT显示右下叶肺炎。尿常规显示血尿和蛋白尿。血清丙氨酸转氨酶为52 U/L,肌酐137 μmol/L,钠128 mmol/L,肌酸激酶6893 U/L。头颅CT正常。脑脊液分析显示总蛋白轻度升高。从支气管肺泡灌洗液中分离出的军团菌菌落经聚合酶链反应检测呈阳性。最初用莫西沙星和阿奇霉素治疗7天,发热和神经症状持续存在。给予皮质类固醇治疗3天,发热消退,而神经症状在抗生素治疗4周后逐渐缓慢改善。最后,连续血清学检测通过间接免疫荧光法确认嗜肺军团菌血清群6和7。回顾了21篇文献共23例病例,加上我们的病例,共有24例病例进行分析。其中男性16例,女性8例,年龄22至71岁。共济失调和构音障碍是最常报告的小脑症状,分别发生在22例和18例中。所有患者在患病期间均有各种中枢和周围神经病变。21例报告了神经影像学和脑脊液分析情况。头颅影像学检查18例无异常,头颅CT 1例有轻度脑积水,头颅MRI 3例胼胝体压部有高信号。脑脊液分析18例正常,1例淋巴细胞升高,3例蛋白升高。9例最终通过尿抗原检测确定为嗜肺军团菌血清型1,1例为嗜肺军团菌血清群6和7,其余14例血清型不明。长期神经随访显示,3例在第一周完全恢复,19例在接下来的3周缓慢改善,13例在3个月后仍有步态或言语持续缺陷。军团菌病合并小脑功能不全罕见。发病时可能被误诊。治疗后有恢复缓慢的趋势,神经症状在长期随访中可能持续存在。

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