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  • Title: [Legionnaires' disease with pronounced cerebellar involvement: case report and literature review].
    Author: Lu M, Shen N, Zhu H, Yao WZ.
    Journal: Zhonghua Jie He He Hu Xi Za Zhi; 2020 Feb 12; 43(2):126-131. PubMed ID: 32062882.
    Abstract:
    Objective: To summarize the clinical course, neuroimaging and cerebrospinal fluid (CSF) analyses of cerebellar dysfunction in Legionnaires' disease. Methods: 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. Results: 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. Conclusions: 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. 目的： 探讨表现为小脑功能不全的军团菌病的临床、头颅影像学和脑脊液特点。 方法： 报道1例表现为小脑功能不全的军团菌病病例。以"军团菌，小脑"为检索词检索中国知网和万方数据库，检索时限为1978年1月至2019年2月。以"Legionella，legionellosis，legionnaires′ disease，cerebellum，cerebellar"为检索词检索PubMed数据库，检索时限为1967年1月1日至2019年2月28日。 结果： 患者男，69岁。因"纳差2周，腹泻1周，发热、共济失调步态、言语不利、全身不自主震颤3 d"入院，胸部CT显示右下肺实变。尿常规潜血（+++），蛋白（+）。血丙氨酸转氨酶为52 U/L，肌酐为137 μmol/L，血钠为128 mmol/L，肌酸激酶为6 893 U/L。头颅CT未见异常，脑脊液检测蛋白轻度升高。支气管肺泡灌洗液中军团菌核酸检测阳性。给予莫西沙星联合阿奇霉素抗感染1周后症状无改善，加用糖皮质激素3 d体温较前下降，并继续抗感染治疗4周后肺炎和神经系统症状逐渐缓解，2个月后痊愈。血清间接免疫荧光法检测嗜肺军团菌血清型6型和7型抗体阳性，且抗体滴度有4倍以上动态变化，最终确诊为重症军团菌病。共检索到的资料相对完整的21篇文献（中文1篇，英文20篇），对其中的23例患者以及本例共24例进行分析，其中男16例，女8例，年龄22~71岁。军团菌病并合并小脑受累最常见的神经系统症状为共济失调（22例）和构音障碍（18例）。24例均不同程度伴有其他中枢和外周神经系统病变，18例头颅影像学检查未见异常，1例头颅CT显示轻度脑积水，3例头颅MRI显示胼胝体压部高信号。18例脑脊液检测完全正常，1例淋巴细胞轻度增多，3例蛋白轻度升高。9例最终确诊为嗜肺军团菌血清型1型（通过尿抗原检测），1例为嗜肺军团菌血清型6型和7型混合感染，其余14例血清型不详。3例治疗后肺炎和神经系统症状快速好转，19例治疗1周至1个月后缓慢改善，其中13例在3个月后仍有步态和言语异常。 结论： 军团菌病合并小脑功能不全临床少见，发病初期干扰因素多，容易误诊。治疗见效慢，远期可能遗留步态和言语异常。.
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