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易栓症倾向患者中由[具体病因未给出]引起的并可能合并立克次体感染的复杂性勒米埃尔综合征

Complicated Lemierre Syndrome Caused by and Possible Rickettsial Co-Infection in a Patient with Thrombophilia Predisposition.

作者信息

Ioannou Antreas, Dimitriou Dimitra, Dimitriou Panagiotis, Katsios Aram, Petrikkos Georgios

机构信息

Internal Medicine Department, Nicosia General Hospital, 2029 Strovolos, Nicosia, Cyprus.

Infectious Diseases Research Laboratory, 4th Dept. of Internal Medicine, National and Kapodistrian University of Athens, Medical School, "Attikon" University Hospital, Athens, Greece.

出版信息

Eur J Case Rep Intern Med. 2017 Mar 17;4(5):000606. doi: 10.12890/2017_000606. eCollection 2017.

DOI:10.12890/2017_000606
PMID:30755946
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC6346786/
Abstract

AIMS

Lemierre syndrome is a life-threating condition characterized by recent oropharyngeal infection, internal jugular vein thrombosis, and anaerobic septicemia. It is usually caused by .

METHODS

A young Romanian male presented with fever and rigors, mild tachypnea, hypoxia, sore throat, decayed teeth, and tenderness of the left carotid triangle. Laboratory examination indicated severe sepsis with disseminated intravascular coagulation, acute renal failure, and acute respiratory distress syndrome while the Doppler ultrasonography of the carotids revealed left internal jugular venous thrombosis. The patient was administered piperacillin/tazobactam and vancomycin intravenously, doxycycline orally, and anti-coagulation by enoxaparin based on the diagnosis of Lemierre syndrome. Meanwhile, he was complicated by bilateral diffuse pulmonary cavities and encapsulated pleural effusions and so transcutaneous drainage was performed. The patient was discharged after a month and continued his treatment with oral phenoxypenicillin and doxycycline until full radiographic improvement. He was switched to oral anti-coagulation by vitamin-K antagonists and was referred to a hematologist, a vascular-surgeon, and a dentist.

RESULTS

was isolated from the patient's blood and pleural fluid cultures and serology for . IgM was positive. Thrombophilia genetic tests revealed three minor mutations for fibrinogen-455, plasminogen activator inhibitor-1, and methylenetetrahydrofolate reductase. According to the literature, is not usually a causative agent and Rickettsial spp. have as yet not been correlated with Lemierre syndrome. The failure of left jugular vein recanalization shows a possible causative role of the underlying thrombophilic predisposition.

DISCUSSION

Because of the syndrome's rarity and the atypical microorganisms isolated in this case, increased awareness is advised for its diagnosis and the underlying mechanisms involved in its genesis. The role of anti-coagulation is debatable.

LEARNING POINTS

Very rare condition. Medical practitioners should be vigilant in its diagnosis since septic jugular vein thrombophlebitis could be misdiagnosed as neck lymphadenitis.Duration of antibiotic therapy and additional anticoagulation treatment are still under discussion.Underlying thrombophilia predisposition should be excluded.

摘要

目的

勒米尔综合征是一种危及生命的疾病,其特征为近期口咽感染、颈内静脉血栓形成和厌氧败血症。通常由……引起。

方法

一名年轻的罗马尼亚男性出现发热、寒战、轻度呼吸急促、缺氧、咽痛、龋齿以及左颈动脉三角区压痛。实验室检查显示严重脓毒症伴弥散性血管内凝血、急性肾衰竭和急性呼吸窘迫综合征,而颈动脉多普勒超声检查显示左颈内静脉血栓形成。根据勒米尔综合征的诊断,患者接受了静脉注射哌拉西林/他唑巴坦和万古霉素、口服多西环素以及依诺肝素抗凝治疗。同时,他并发双侧弥漫性肺空洞和包裹性胸腔积液,因此进行了经皮引流。患者在一个月后出院,继续口服苯氧西林和多西环素治疗,直至影像学完全改善。随后改用维生素K拮抗剂进行口服抗凝治疗,并转诊至血液科医生、血管外科医生和牙医处。

结果

从患者的血液和胸腔积液培养物中分离出……,并进行了……的血清学检查。IgM呈阳性。血栓形成倾向基因检测显示纤维蛋白原-455、纤溶酶原激活物抑制剂-1和亚甲基四氢叶酸还原酶存在三个微小突变。根据文献,……通常不是病原体,立克次体属尚未与勒米尔综合征相关联。左颈静脉再通失败表明潜在的血栓形成倾向可能起致病作用。

讨论

由于该综合征罕见,且本病例中分离出非典型微生物,建议提高对其诊断及发病机制的认识。抗凝的作用存在争议。

学习要点

非常罕见的疾病。医生在诊断时应保持警惕,因为化脓性颈静脉血栓性静脉炎可能被误诊为颈部淋巴结炎。抗生素治疗的持续时间和额外的抗凝治疗仍在讨论中。应排除潜在的血栓形成倾向。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/de4b/6346786/95cee92b7e40/606_Fig3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/de4b/6346786/2fc4beb0c202/606_Fig1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/de4b/6346786/0e31c2e20b7c/606_Fig2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/de4b/6346786/95cee92b7e40/606_Fig3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/de4b/6346786/2fc4beb0c202/606_Fig1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/de4b/6346786/0e31c2e20b7c/606_Fig2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/de4b/6346786/95cee92b7e40/606_Fig3.jpg

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本文引用的文献

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Lemierre syndrome-should we anticoagulate? A case report and review of the literature.勒米尔综合征——我们应该进行抗凝治疗吗?一例病例报告及文献综述。
Int J Angiol. 2013 Jun;22(2):137-42. doi: 10.1055/s-0033-1336828.
2
Human infection with Fusobacterium necrophorum (Necrobacillosis), with a focus on Lemierre's syndrome.人类感染坏死梭杆菌(坏死杆菌病),重点关注勒米尔综合征。
Clin Microbiol Rev. 2007 Oct;20(4):622-59. doi: 10.1128/CMR.00011-07.
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Rickettsiae and rickettsial infections: the current state of knowledge.立克次氏体及立克次氏体感染:当前的知识状况
Clin Infect Dis. 2007 Jul 15;45 Suppl 1:S39-44. doi: 10.1086/518145.
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Lemierre's syndrome and genetic polymorphisms: a case report.勒米尔综合征与基因多态性:一例报告
BMC Infect Dis. 2006 Jul 17;6:115. doi: 10.1186/1471-2334-6-115.
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Incidence and clinical epidemiology of necrobacillosis, including Lemierre's syndrome, in Denmark 1990-1995.1990 - 1995年丹麦坏死杆菌病(包括勒米尔氏综合征)的发病率及临床流行病学情况。
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