Lemierre's Syndrome: Atypical Presentation of the Internal Carotid Artery and Jugular Vein Fistula—CaseReport and Review

Lemierre's Syndrome: Atypical Presentation of the Internal Carotid Artery and Jugular Vein Fistula—CaseReport and Review | Research Square window.SnipcartSettings = { analytics: { enabled: false } }; (function() { var accessVector = localStorage.getItem('access_vector') || ''; window.dataLayer = window.dataLayer || []; if (accessVector) { window.dataLayer.push({ user: { profile: { profileInfo: { snid: accessVector } } } }); } })(); (function(w,d,s,l,i){w[l]=w[l]||[];w[l].push({'gtm.start':new Date().getTime(),event:'gtm.js'});var f=d.getElementsByTagName(s)[0],j=d.createElement(s),dl=l!='dataLayer'?'&l='+l:'';j.async=true;j.src='https://www.googletagmanager.com/gtm.js?id='+i+dl;f.parentNode.insertBefore(j,f);})(window,document,'script','dataLayer','GTM-K279D39R'); Browse Preprints In Review Journals COVID-19 Preprints AJE Video Bytes Research Tools Research Promotion AJE Professional Editing AJE Rubriq About Preprint Platform In Review Editorial Policies Our Team Advisory Board Help Center Sign In Submit a Preprint Cite Share Download PDF Case Report Lemierre's Syndrome: Atypical Presentation of the Internal Carotid Artery and Jugular Vein Fistula—CaseReport and Review Dr. Liza Wanjagi Kariuki, Dr. Jean Aime Musabyemungu, Dr. Jemima kebedi, and 8 more This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-4283446/v1 This work is licensed under a CC BY 4.0 License Status: Posted Version 1 posted You are reading this latest preprint version Abstract Background: Lemierre's syndrome (LS) is a rare yet potentially life-threatening complication of oropharyngeal infections that is typically characterized by septic thrombophlebitis of the internal jugular vein and is commonly associated with Fusobacterium necrophorum . Despite its recognition since 1936, LS can present atypically, as seen in our case where a patient exhibited a rare complication of an internal carotid artery and jugular vein fistula. This case highlights the diagnostic and therapeutic challenges that can arise and underscores the importance of both a broader awareness of LS and the necessity for a multidisciplinary approach. Case Presentation: A 42-year-old male who was initially diagnosed with a periodontal abscess and tonsillitis later developed internal jugular vein thrombophlebitis. This progressed to an internal carotid to internal jugular vein fistula, leading to septic thrombosis of the left internal carotid and jugular vein and eventually culminating in a carotid bulb aneurysm and subsequent cerebral infarct. The patient also experienced septic emboli, resulting in lung and mediastinal abscesses. Treatment involved intravenous antibiotics, anticoagulant therapy, and vascular repair, yielding a favorable outcome. Conclusions: This case underscores the intricate nature of LS, showing its potential to progress to severe septic complications, including rare vascular issues and cerebral infarction. The diagnostic complexities and therapeutic interventions emphasize the critical importance of early identification, prompt diagnosis, and multidisciplinary management of LS to prevent life-threatening outcomes. Lemierre's syndrome Septic thrombophlebitis Pharyngeal space. Figures Figure 1 Figure 2 Figure 3 Introduction Lemierre's syndrome (LS) is an infrequent but potentially deadly complication of oropharyngeal infections and is characterized by septic thrombophlebitis of the internal jugular vein (IJV). This condition was first reported in 1936 by a French physician, Andre Lemierre, who identified 20 cases of anaerobic septicaemia, resulting in 18 fatalities ( 1 ). LS involves an initial infection in the oropharyngeal region and later thrombophlebitis in the internal jugular vein. Bacteria can then spread through the bloodstream or in the lymphatic system to other areas, causing septic metastases, primarily in the brain and lungs but also in the kidneys or joints. The majority of cases involve young and healthy individuals ( 2 , 3 ). Despite the recognized patterns of LS, there have been reports of variants displaying unusual features, including the age of onset and the location of infection or septic metastases. Fusobacterium necrophorum , frequently isolated from both blood and pus cultures, is predominantly acknowledged as the main causative agent of the syndrome, although other bacterial pathogens have also been identified ( 3 – 5 ). We present a patient with LS with an initial typical presentation that eventually progressed to significant septic sequelae, including lung abscess, multiple mediastinal abscesses associated with fistula, aneurysm and thrombosis of the left neck main vessels, resulting in a left middle cerebral artery territory infarct. Case Presentation A 42-year-old male who presented to a peripheral hospital for one month experienced a sore throat and pain while swallowing. He was diagnosed with dental abscess and tonsillitis and treated with IV ceftriaxone and metronidazole for two weeks. The patient reported slight improvement and was discharged from the hospital. One month later, he presented to the same facility with respiratory distress and septic shock. At that time, he was diagnosed with right empyema thoracis and cervical and mediastinal abscesses. A right chest tube was inserted, and the patient was transferred to the intensive care unit (ICU). A presumptive diagnosis of tuberculosis (TB) was made due to the high TB incidence in the region, leading to the initiation of anti-tuberculous medications (RHZE), in addition to other medications he was receiving in the ICU. No central line or other invasive monitoring devices were used. Once the patient improved, he was transferred to the ward for continued treatment. While receiving medication in the ward, he progressively developed painful left neck swelling accompanied by hoarseness of the voice and intermittent right arm weakness. Swallowing remained unaffected. He had no history of trauma but was an active smoker and alcohol user. TB, HIV and hepatitis were negative. He was then referred to Kenyatta National Hospital (KNH) for further treatment. Initial physical examination at KNH revealed the following vital signs : BP, 130/92 mmHg; PR, 92 bpm; T, 36.6; SpO2, 96% on room air; RR, 15 bpm; and GCS, 15/15 . Neck Exam: A Level II and III left neck mass measuring 8 x 10 cm was palpable anterior to the sternocleidomastoid muscle. It exhibited a firm consistency, was tender, was pulsatile, and was accompanied by bruits on auscultation, with no apparent skin changes. No palpable lymph nodes were detected. Oral Exam: The uvula and tongue deviated to the right, with the medial lobe bulk on the left. Tonsillar hypertrophy was noted, with Grade III on the left and Grade II right tonsillar crypts, along with a pharyngeal cobblestone. Otoscopy revealed normal findings. Cranial Nerve Assessment : Left Hypoglossal (CNXII), glossopharyngeal (CNIX) and recurrent laryngeal nerve parts of the Vagus nerve (X) palsy were observed. Motor and sensory exam : The right arm had reduced power, with normal tone, bulk, reflexes and sensory exam. Imaging studies : The imaging studies revealed a complex array of vascular and neurological abnormalities. Initial findings, identified through an IV contrast CT scan, included septic left internal jugular vein thrombophlebitis, an aneurysm of the left carotid bulb/internal carotid artery, and an arteriovenous fistula between the left carotid bulb and internal jugular vein. The situation was further complicated by large neck abscesses extending into the mediastinum, right pulmonary septic emboli, and left vocal cord paralysis. Subsequent diagnostic modalities, including repeat chest and brain CT scans, MRI of the brain with magnetic resonance angiography (MRA) and magnetic resonance venography (MRV), as well as neck vessel Doppler ultrasound, revealed additional critical findings. These included the resolution of the abscess, thrombosis in both the left internal jugular vein (IJV) and the internal carotid artery (ICA), spontaneous closure of the AV fistula, chronic infarcts in the left frontal lobe and the territory of the left middle cerebral artery (MCA), a normal status of the circle of Willis, and a reduction in the caliber of various arteries and veins. There was persistence of the thrombotic aneurysm and left vocal cord paralysis. Figures 1-3. Coronal MIP, 3D reconstruction, and sagittal MIP images of the neck showing a bilobed aneurysm of the left carotid bulb extending to the internal carotid artery, with a surrounding hematoma. 3D reconstruction also revealed left sigmoid sinus thrombosis. The ECA was normal. Multidisciplinary management, involving specialists from various medical fields to ensure comprehensive care and optimal treatment outcomes, was undertaken to address the complex nature of the patient's condition. Medical Management: Upon suspicion of LS, broad-spectrum intravenous antibiotics, including ceftriaxone and metronidazole, and anticoagulation therapy were initiated under close monitoring. Surgical management of the aneurysm revealed several critical findings. The aneurysm sac extended from the bifurcation of the left common carotid artery, affecting both the external and internal carotid arteries. Notably, the IJV was embedded within the wall of the sac. There was extensive thrombosis throughout the entire length of the left IJV and in the left ICA. Additionally, cranial nerves (CNs) IX, X, and XII could not be clearly delineated from the wall of the aneurysm sac or the surrounding inflammatory tissue, complicating the surgical approach. Furthermore, there was no backflow from the left internal carotid artery, indicating significant vascular impairment. During the operation, the surgical team evacuated a massive thrombus and ligated the left common carotid artery at its bifurcation. Postoperatively, the patient quickly regained consciousness, showing no new neurological deficits. Remarkably, just five days after the operation, the patient was evaluated and considered ready for discharge, after which he subsequently returned home. A follow-up examination two weeks later demonstrated a complete recovery of function in the CNIX, CNX and CNXII groups as his voice returned to normal. Nevertheless, the patient continued to experience persistent left hemiparesis, which was attributed to an infarct in the MCA. Discussion Our case report describes a unique manifestation of LS, characterized by the rare complication of an internal carotid artery and internal jugular vein fistula, followed by the development of septic thrombosis leading to aneurysm formation and cerebral infarction. This anomaly was observed following a periodontal abscess and tonsillitis, evolving into deep neck space, mediastinal, and chest abscesses, along with left IJV thrombophlebitis. This sequence of events underscores the critical nature of LS and its potential to progress to life-threatening complications, despite the initial administration of broad-spectrum antibiotics. The rarity of this presentation within the context of LS contributes significantly to the medical literature, highlighting the need for vigilance in cases of persistent oropharyngeal infection symptoms, especially when accompanied by vascular complications. Once labeled the 'forgotten disease' in the 1980s due to effective antibiotic use, LS, a deadly disease in the preantibiotic era, has experienced a documented increase in incidence mainly attributed to the increase in antibiotic resistance (6). Despite its rarity, the syndrome presents diagnostic challenges, necessitating a comprehensive, multidisciplinary approach for effective management (5,7,8). Our case underscores the crucial importance of early diagnosis and prompt initiation of targeted therapy to address potentially life-threatening complications (6). Demographically, LS primarily affects young individuals, typically aged 15 to 24 years, with a peak incidence in the second and third decades of life and a slightly greater male incidence (male-to-female ratio of 2:1). However, it has been documented in individuals across a wide age range, from as young as 2 months to as old as 78 years (3,9). Fusobacterium necrophorum , identified as the causative agent in 10% of peritonsillar abscesses and 81% of LS cases, is an anaerobic bacterium prevalent in the oropharynx, genitourinary system, and gastrointestinal tract (5,8,10). Heightened suspicion should arise when oropharyngeal pharyngitis or periodontal abscess persists beyond 3 to 5 days and is accompanied by systemic or respiratory symptoms, lateral cervical pain, dysphagia, progression to sepsis or multiple pulmonary abscesses (5,8,9,11) The common presentation of LS is unilateral neck discomfort and inflammation at the anterior border of the sternocleidomastoid muscle after a recent throat infection (3,12). The detection of this syndrome may be easily overlooked or delayed since patients typically exhibit a palpable, tender cord (or 'cord sign') of the internal jugular vein in only 26% to 45% of cases (1,3,8,13). Furthermore, microbiology laboratories often encounter challenges in cultivating Fusobacterium necrophorum , as observed in our case. The conclusive identification of pathognomonic anaerobes through blood cultures in the laboratory may require several days for growth on specialized media that are not routinely utilized in most laboratory settings (9,10) . Moreover, the identification process may be impeded by the widespread use of antibiotics for outpatient treatment of pharyngitis (1,5,14). The peritonsillar abscesses spread via hematogenesis or through the lymphatic system. Venous drainage of the palatine tonsils occurs through the peritonsillar plexus of veins, which drains into the pharyngeal and lingual veins and ultimately into the internal jugular vein. The palatine tonsils drain to the jugulodigastric node, a node of the deep cervical lymph nodes, where the posterior belly of the digastric muscle crosses the internal jugular vein (15). The most common metastatic abscess sites are the lung (empyema or cavitary nodules), mediastinum, brain, and IJV (2,16). Other organs that may be affected include the bone/joint (septic arthritis), the soft tissue and muscle, intraabdominal abscesses in the liver or spleen, and the central nervous system (in the form of chronic infarcts or abscesses and/or meningitis) (4,10,17,18). In our patient, the infection spread in the cervical, thoracic, or mediastinal spaces up to the dura sinus, and left IJV thrombosis and then left ICA thrombosis resulted in chronic infarction of the left MCA territory (19). The aneurysm at the left common carotid artery CCA-carotid bulb resulted from thrombophlebitis and mechanical pressure from the fistula, leading to damage to the left CCA wall and subsequent aneurysm formation, as typically described in the literature on aneurysm pathophysiology (20–23). However, to the best of our knowledge, this is the first case of LS involving a carotid-jugular vein fistula, a left carotid bulb CCA and left ECA aneurysm, and extensive vascular thrombosis leading to cerebral infarction. Historically, LS diagnosis has relied on criteria such as primary head or neck infection, thrombosis of head/neck veins, and isolation of Fusobacterium necrophorum from blood cultures (2,5). However, evolving diagnostic criteria currently acknowledge challenges in isolating Fusobacterium necrophorum, making it nonmandatory, as a result of potential negative cultures resulting from empirical antibiotic therapy (10,17). Radiological investigations, particularly duplex ultrasonography, CT scans, and magnetic resonance imaging, play a crucial role in diagnosis. Contrast-enhanced CT is considered optimal because it offers accessibility, cost-effectiveness, and visualization of adjacent structures (6,24). The radiological findings reported in other studies align with our case, where similar modalities were employed and yielded comparable results, confirming the diagnosis of LS. Our patient received ceftriaxone and metronidazole in combination with anti-TB therapy following the standard treatment protocol because of the high TB prevalence in this region. This combination was effective in treating extensive abscesses. Although neither Ceftriaxone nor anti-TB drugs are known for treating LS, evidence indicates that similar cephalosporins have been effective against LS due to their action on anaerobic bacteria (10)(25). Given the occurrence of beta-lactamase-producing Fusobacterium. For necrophorum strains resistant to macrolides and penicillin (6,26), the literature recommends the use of a beta-lactamase-resistant beta-lactam antibiotic in conjunction with metronidazole, which is initially advised for two weeks, transitioning to metronidazole alone for six weeks (13,14). This approach ensures deep penetration into fibrin clots and addresses potential resistance issues. The use of antituberculosis therapy underscores the diagnostic challenges of LS, a condition easily mistaken for others due to its severity and rarity, as well as the complexities involved in identifying the responsible bacteria. The use of prophylactic anticoagulant agents for managing LS remains a subject of considerable debate within the medical community. This controversy stems from the disparate findings reported in the literature, compounded by the disease's rarity, which inherently limits the feasibility of conducting statistically robust trials to assess the effectiveness of anticoagulation therapy comprehensively (27). Despite these challenges, a consensus among several authors advocates for the utilization of anticoagulation therapy in LS patients as a strategy to mitigate neurological complications. This underscores the importance of a tailored approach that weighs the estimated risk of venous thromboembolism against potential contraindications (5,6,13,14). Specifically, the deployment of anticoagulant therapy is recommended for patients identified as high risk, emphasizing a judicious evaluation of benefits and risks (28,29). In the case presented, the incorporation of an anticoagulant regimen is strongly corroborated by evidence from the literature, which posits that anticoagulation, particularly in scenarios of carotid thrombosis, is associated with a diminished recurrence of ischemic events (30,31). In the context of LS, surgical procedures have predominantly focused on the drainage of pus, as indicated by the literature (10). To our knowledge, the present case is unprecedented in necessitating vascular surgery as a part of LS management. Surgery was required to mitigate the effect of the mass on cranial nerves IX and XII. The standard of care for treating infected carotid artery pseudoaneurysms predominantly involves traditional open repair. This approach includes aneurysmectomy, debridement of surrounding infected tissues, and carotid artery ligation (32–35). Revascularization of the ICA is generally considered only in cases where it is deemed essential, such as in patients with bilateral ICA disease (36,37). This cautious approach is due to the potential risk of embolic events in the brain (38,39). In line with recommendations from several studies, our patient underwent ligation of both the left ICA and the ECA without attempting revascularization of the left IJV and ICA, owing to thrombosis. During the repair process, we observed evidence of spontaneous closure of the internal jugular vein-carotid artery (IJV-ICA) fistula, which was secondary to thrombosis in both the IJV and ICA. These observations are consistent with findings reported in the literature, specifically the spontaneous resolution of direct carotid-cavernous fistulas documented in 2019 (40,41) Conclusion Our case of Lemierre's syndrome, featuring an unusual presentation of an internal carotid artery and internal jugular vein fistula, emphasizes the critical importance of prompt recognition, a multidisciplinary treatment approach, and aggressive management to prevent life-threatening complications. This case highlights the atypical manifestations of Lemierre’s syndrome and stresses the need to consider it in patients with recent oropharyngeal infections who develop complex vascular symptoms. This study offers valuable insights into therapeutic strategies for managing complex cases of Lemierre’s syndrome, emphasizing the significance of personalized care, including targeted antibiotic therapy, judicious use of anticoagulation agents, and surgical intervention. Recommendations Prompt Antibiotic Therapy: Early initiation of broad-spectrum antibiotics tailored based on microbiological culture findings. Selective anticoagulation: Case-by-case assessment for anticoagulation, especially in patients with significant vascular thrombosis. Integrated Care Approach: Collaborative effort among specialists, including those involved in infectious diseases, radiology, vascular surgery, otorhinolaryngology, and intensive care. Abbreviations LS - Lemierre's Syndrome IJV - Internal jugular vein CT - Computed Tomography MRI - magnetic resonance imaging MCA - middle cerebral artery ICU - Intensive Care Unit TB - Tuberculosis CN - Cranial Nerve RHZE - Rifampicin, Isoniazid, Pyrazinamide, Ethambutol CCA - common carotid artery ICA - Internal carotid artery ECA - External carotid artery MRA - magnetic resonance angiography MRV - Magnetic Resonance Venography Declarations Author Contribution L.W.K., J.A.M., and J.K. prepared and wrote the main manuscript text. P.K. prepared figures 1-3. All authors, including M.S., F.H., M.A., G.O., C.I., and S.N.,T.O. alongside those mentioned above, reviewed the manuscript. Acknowledgement The authors sincerely thank Kathryn Parker for her valuable contribution to reviewing the manuscript, which significantly improved its quality. They appreciate her time and expertise in maintaining the integrity and accuracy of the research. Ethics Statement and Informed Consent: Ethical approval was waived, and the patient provided written permission to publish this case report and associated images. References Valerio L, Corsi G, Sebastian T, Barco S. Lemierre syndrome: Current evidence and rationale of the Bacteria-Associated Thrombosis, Thrombophlebitis and LEmierre syndrome (BATTLE) registry. Thrombosis Research. Volume 196. Elsevier Ltd; 2020. pp. 494–9. Lucian, Giubelan. 1,2,† Livia Dragonu,1,2,† Vlad Pădureanu,3,* Alexandru Neacșu,2,* Mirela Mănescu,2 Andreea Cristina Stoian,1 and Florentina Dumitrescu1,2. 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Interventional Neuroradiology [Internet]. 2019 Feb 1 [cited 2024 Feb 11];25(1):71. Available from:/pmc/articles/PMC6378520/ . Spontaneous resolution of direct carotid-. cavernous fistulas: case series and literature review - PMC [Internet]. [cited 2024 Feb 11]. https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6378520/ . Additional Declarations No competing interests reported. Cite Share Download PDF Status: Posted Version 1 posted You are reading this latest preprint version Research Square lets you share your work early, gain feedback from the community, and start making changes to your manuscript prior to peer review in a journal. As a division of Research Square Company, we’re committed to making research communication faster, fairer, and more useful. We do this by developing innovative software and high quality services for the global research community. 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Also discoverable on Platform About Our Team In Review Editorial Policies Advisory Board Help Center Resources Author Services Accessibility API Access RSS feed Manage Cookie Preferences © Research Square 2026 | ISSN 2693-5015 (online) Privacy Policy Terms of Service Do Not Sell My Personal Information {"props":{"pageProps":{"initialData":{"identity":"rs-4283446","acceptedTermsAndConditions":true,"allowDirectSubmit":true,"archivedVersions":[],"articleType":"Case Report","associatedPublications":[],"authors":[{"id":293069490,"identity":"08b6b066-958a-4b85-821a-659b90523bd9","order_by":0,"name":"Dr. Liza Wanjagi Kariuki","email":"","orcid":"","institution":"University of Nairobi","correspondingAuthor":false,"prefix":"Dr.","firstName":"Liza","middleName":"Wanjagi","lastName":"Kariuki","suffix":""},{"id":293069491,"identity":"a2249d98-e5ea-448c-9ad5-071a55a03fb8","order_by":1,"name":"Dr. Jean Aime Musabyemungu","email":"data:image/png;base64,iVBORw0KGgoAAAANSUhEUgAAAZAAAAAyAQMAAABI0h/eAAAABlBMVEX///8AAABVwtN+AAAACXBIWXMAAA7EAAAOxAGVKw4bAAAAy0lEQVRIiWNgGAWjYJACAyBk4AexEgpI0SLZANJiQJJVByC6CQPdGekPCn4UbJMzPr868cMDAwZ5frED+LWY3cgxMOwxuG1sduPtZgmgwwxnzk4gqIXBgMfgduK2G2c3gLQkGNwmqCX9geEfg9v1m2ec3fyDSC0JBsZAWxIM+Hu3EWnLmTcGxjIGtw1n3ODdZpFgIEGEX46nPzN88+e2PH//2c03f1TYyPNLE9ACBGyQyJAAq5QgqBwEmB+AKf4DRKkeBaNgFIyCEQgAihZHHMt2gBgAAAAASUVORK5CYII=","orcid":"","institution":"University of Nairobi","correspondingAuthor":true,"prefix":"Dr.","firstName":"Jean","middleName":"Aime","lastName":"Musabyemungu","suffix":""},{"id":293069492,"identity":"4ad2d82a-48ab-4efa-8e13-e5cf114115ec","order_by":2,"name":"Dr. Jemima kebedi","email":"","orcid":"","institution":"University of Nairobi","correspondingAuthor":false,"prefix":"Dr.","firstName":"Jemima","middleName":"","lastName":"kebedi","suffix":""},{"id":293069493,"identity":"4a79115e-b226-4437-ab4b-1c887f2cd401","order_by":3,"name":"Dr. Mitali Shah","email":"","orcid":"","institution":"University of Nairobi","correspondingAuthor":false,"prefix":"Dr.","firstName":"Mitali","middleName":"","lastName":"Shah","suffix":""},{"id":293069494,"identity":"9b9db3e2-42a2-4f42-ab11-9055183f3640","order_by":4,"name":"Fadhil Hussein","email":"","orcid":"","institution":"University of Nairobi","correspondingAuthor":false,"prefix":"","firstName":"Fadhil","middleName":"","lastName":"Hussein","suffix":""},{"id":293069495,"identity":"4c8a5f5d-df1e-4ac9-b375-26fe403e35f9","order_by":5,"name":"Dr. Mohamednoor Ahmed","email":"","orcid":"","institution":"University of Nairobi","correspondingAuthor":false,"prefix":"Dr.","firstName":"Mohamednoor","middleName":"","lastName":"Ahmed","suffix":""},{"id":293069496,"identity":"b13f9b5a-e035-4396-8ff7-7be51d694c8d","order_by":6,"name":"Dr. Gilbert Oburu","email":"","orcid":"","institution":"Kenyatta National Hospital","correspondingAuthor":false,"prefix":"Dr.","firstName":"Gilbert","middleName":"","lastName":"Oburu","suffix":""},{"id":293069499,"identity":"aa0e7560-f695-4ea3-8438-4d5cfa9ad873","order_by":7,"name":"Dr. Catherine Irungu","email":"","orcid":"","institution":"University of Nairobi","correspondingAuthor":false,"prefix":"Dr.","firstName":"Catherine","middleName":"","lastName":"Irungu","suffix":""},{"id":293069502,"identity":"81093c88-903c-4a9e-9f14-9342348ed565","order_by":8,"name":"Dr. Pauline Kibaru","email":"","orcid":"","institution":"Kenyatta National Hospital","correspondingAuthor":false,"prefix":"Dr.","firstName":"Pauline","middleName":"","lastName":"Kibaru","suffix":""},{"id":293069506,"identity":"c20c8fc8-399e-4186-b433-46a671bfd731","order_by":9,"name":"Dr. Simon Ndiritu","email":"","orcid":"","institution":"Kenyatta National Hospital","correspondingAuthor":false,"prefix":"Dr.","firstName":"Simon","middleName":"","lastName":"Ndiritu","suffix":""},{"id":293069513,"identity":"2095d382-9695-4d0c-8c5b-3a0d702be581","order_by":10,"name":"Dr. Tom Omulo","email":"","orcid":"","institution":"University of Nairobi","correspondingAuthor":false,"prefix":"Dr.","firstName":"Tom","middleName":"","lastName":"Omulo","suffix":""}],"badges":[],"createdAt":"2024-04-17 17:44:23","currentVersionCode":1,"declarations":"","doi":"10.21203/rs.3.rs-4283446/v1","doiUrl":"https://doi.org/10.21203/rs.3.rs-4283446/v1","draftVersion":[],"editorialEvents":[],"editorialNote":"","failedWorkflow":false,"files":[{"id":55325536,"identity":"a1f3dbfb-d884-4158-b7b3-af8778367ce8","added_by":"auto","created_at":"2024-04-25 16:58:04","extension":"jpg","order_by":1,"title":"Figure 1","display":"","copyAsset":false,"role":"figure","size":327023,"visible":true,"origin":"","legend":"\u003cp\u003eFigures 1-3. Coronal MIP, 3D reconstruction, and sagittal MIP images of the neckshowing a bilobed aneurysm of the left carotid bulb extending to the internal carotid artery, with a surrounding hematoma. 3D reconstruction also revealed left sigmoid sinus thrombosis. The ECA was normal.\u003c/p\u003e","description":"","filename":"Figure1.jpg","url":"https://assets-eu.researchsquare.com/files/rs-4283446/v1/42cc008f9d8bb5e73638f6cc.jpg"},{"id":55325957,"identity":"1d2fd976-f446-49f0-ab5a-6804f4b3cb05","added_by":"auto","created_at":"2024-04-25 17:06:04","extension":"jpg","order_by":2,"title":"Figure 2","display":"","copyAsset":false,"role":"figure","size":303344,"visible":true,"origin":"","legend":"\u003cp\u003eFigures 1-3. Coronal MIP, 3D reconstruction, and sagittal MIP images of the neckshowing a bilobed aneurysm of the left carotid bulb extending to the internal carotid artery, with a surrounding hematoma. 3D reconstruction also revealed left sigmoid sinus thrombosis. The ECA was normal.\u003c/p\u003e","description":"","filename":"Figure2.jpg","url":"https://assets-eu.researchsquare.com/files/rs-4283446/v1/c32178e5f2284d8421b57ccb.jpg"},{"id":55325958,"identity":"b171d2ee-5cae-43d1-b818-bbab96d0d42b","added_by":"auto","created_at":"2024-04-25 17:06:04","extension":"jpg","order_by":3,"title":"Figure 3","display":"","copyAsset":false,"role":"figure","size":318175,"visible":true,"origin":"","legend":"\u003cp\u003eFigures 1-3. Coronal MIP, 3D reconstruction, and sagittal MIP images of the neckshowing a bilobed aneurysm of the left carotid bulb extending to the internal carotid artery, with a surrounding hematoma. 3D reconstruction also revealed left sigmoid sinus thrombosis. The ECA was normal.\u003c/p\u003e","description":"","filename":"Figure3.jpg","url":"https://assets-eu.researchsquare.com/files/rs-4283446/v1/533d06ff01f5d2b6a4a7623d.jpg"},{"id":67345055,"identity":"09597a5a-4ee9-4542-a1a1-20200937d5dd","added_by":"auto","created_at":"2024-10-24 02:16:51","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":1266609,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-4283446/v1/6aa3f1a9-f99a-4d2a-bd0c-911d82018ae7.pdf"}],"financialInterests":"No competing interests reported.","formattedTitle":"Lemierre's Syndrome: Atypical Presentation of the Internal Carotid Artery and Jugular Vein Fistula—CaseReport and Review","fulltext":[{"header":"Introduction","content":"\u003cp\u003eLemierre's syndrome (LS) is an infrequent but potentially deadly complication of oropharyngeal infections and is characterized by septic thrombophlebitis of the internal jugular vein (IJV). This condition was first reported in 1936 by a French physician, Andre Lemierre, who identified 20 cases of anaerobic septicaemia, resulting in 18 fatalities (\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e).\u003c/p\u003e \u003cp\u003eLS involves an initial infection in the oropharyngeal region and later thrombophlebitis in the internal jugular vein. Bacteria can then spread through the bloodstream or in the lymphatic system to other areas, causing septic metastases, primarily in the brain and lungs but also in the kidneys or joints. The majority of cases involve young and healthy individuals (\u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e2\u003c/span\u003e, \u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e). Despite the recognized patterns of LS, there have been reports of variants displaying unusual features, including the age of onset and the location of infection or septic metastases. \u003cem\u003eFusobacterium necrophorum\u003c/em\u003e, frequently isolated from both blood and pus cultures, is predominantly acknowledged as the main causative agent of the syndrome, although other bacterial pathogens have also been identified (\u003cspan additionalcitationids=\"CR4\" citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR5\" class=\"CitationRef\"\u003e5\u003c/span\u003e).\u003c/p\u003e \u003cp\u003eWe present a patient with LS with an initial typical presentation that eventually progressed to significant septic sequelae, including lung abscess, multiple mediastinal abscesses associated with fistula, aneurysm and thrombosis of the left neck main vessels, resulting in a left middle cerebral artery territory infarct.\u003c/p\u003e"},{"header":"Case Presentation","content":"\u003cp\u003eA 42-year-old male\u0026nbsp;who\u0026nbsp;presented to\u0026nbsp;a\u0026nbsp;peripheral hospital for one month\u0026nbsp;experienced\u0026nbsp;a sore throat and pain while swallowing. He was diagnosed with dental abscess and\u0026nbsp;tonsillitis and\u0026nbsp;treated\u0026nbsp;with\u0026nbsp;IV ceftriaxone and\u0026nbsp;metronidazole\u0026nbsp;for two weeks. The patient\u0026nbsp;reported slight improvement and\u0026nbsp;was\u0026nbsp;discharged\u0026nbsp;from the hospital.\u003c/p\u003e\n\u003cp\u003eOne\u0026nbsp;month later, he presented to the same facility\u0026nbsp;with\u0026nbsp;respiratory distress and septic shock. At\u0026nbsp;that\u0026nbsp;time, he was diagnosed\u0026nbsp;with\u0026nbsp;right empyema thoracis\u0026nbsp;and\u0026nbsp;cervical and mediastinal abscesses. A right chest tube was\u0026nbsp;inserted, and\u0026nbsp;the patient\u0026nbsp;was transferred to the intensive care unit (ICU). A presumptive diagnosis of tuberculosis (TB)\u0026nbsp;was made\u0026nbsp;due to\u0026nbsp;the\u0026nbsp;high TB\u0026nbsp;incidence\u0026nbsp;in the region, leading to the initiation of anti-tuberculous medications (RHZE), in addition to other medications he was receiving in the ICU. No central line or other invasive monitoring devices\u0026nbsp;were\u0026nbsp;used.\u0026nbsp;Once\u0026nbsp;the patient improved,\u0026nbsp;he\u0026nbsp;was transferred to the ward for continued treatment.\u003c/p\u003e\n\u003cp\u003eWhile receiving medication in the ward,\u0026nbsp;he\u0026nbsp;progressively\u0026nbsp;developed painful left neck swelling accompanied by hoarseness of\u0026nbsp;the\u0026nbsp;voice and intermittent right arm weakness. Swallowing remained unaffected. He had no history of trauma but was an active smoker and alcohol user. TB, HIV and hepatitis were negative.\u0026nbsp;He was then referred to Kenyatta National Hospital (KNH) for further treatment.\u003c/p\u003e\n\u003cp\u003e\u003cem\u003eInitial physical\u0026nbsp;\u003c/em\u003e\u003cem\u003eexamination at KNH revealed the following vital\u003c/em\u003e\u003cem\u003esigns\u003c/em\u003e: BP,\u0026nbsp;130/92 mmHg;\u0026nbsp;PR,\u0026nbsp;92 bpm;\u0026nbsp;T,\u0026nbsp;36.6;\u0026nbsp;SpO2,\u0026nbsp;96% on\u0026nbsp;room air;\u0026nbsp;RR,\u0026nbsp;15 bpm;\u0026nbsp;and\u0026nbsp;GCS,\u0026nbsp;15/15\u003cem\u003e. Neck Exam:\u003c/em\u003e A Level II and III left neck mass\u0026nbsp;measuring 8 x 10 cm\u0026nbsp;was palpable\u0026nbsp;anterior to the sternocleidomastoid muscle. It exhibited a firm consistency,\u0026nbsp;was\u0026nbsp;tender,\u0026nbsp;was\u0026nbsp;pulsatile,\u0026nbsp;and was\u0026nbsp;accompanied by bruits on auscultation,\u0026nbsp;with no apparent skin changes. No palpable lymph nodes were detected.\u003cem\u003e\u0026nbsp;Oral Exam:\u003c/em\u003e The uvula and tongue deviated to the right,\u0026nbsp;with\u0026nbsp;the\u0026nbsp;medial lobe bulk on the left. Tonsillar hypertrophy was noted, with Grade III on the left and Grade II right tonsillar crypts, along with\u0026nbsp;a\u0026nbsp;pharyngeal cobblestone. Otoscopy revealed normal findings.\u003cem\u003e\u0026nbsp;Cranial Nerve Assessment\u003c/em\u003e: Left Hypoglossal (CNXII),\u0026nbsp;glossopharyngeal\u0026nbsp;(CNIX) and recurrent laryngeal nerve\u0026nbsp;parts\u0026nbsp;of\u0026nbsp;the Vagus\u0026nbsp;nerve (X) palsy\u0026nbsp;were\u0026nbsp;observed. \u003cem\u003eMotor and sensory exam\u003c/em\u003e: The right arm had reduced power, with normal tone, bulk, reflexes and sensory exam.\u003c/p\u003e\n\u003cp\u003e\u003cem\u003eImaging\u0026nbsp;\u003c/em\u003e\u003cem\u003estudies\u003c/em\u003e\u003cem\u003e:\u0026nbsp;\u003c/em\u003eThe imaging studies revealed a complex array of vascular and neurological abnormalities. Initial findings, identified through an IV contrast CT scan, included septic left internal jugular vein thrombophlebitis, an aneurysm of the left carotid bulb/internal carotid artery, and an arteriovenous fistula between the left carotid bulb and internal jugular vein. The situation was further complicated by large neck abscesses extending into the mediastinum, right pulmonary septic emboli, and left vocal cord paralysis.\u003c/p\u003e\n\u003cp\u003eSubsequent diagnostic modalities, including repeat chest and brain CT scans, MRI of the brain with magnetic resonance angiography (MRA) and magnetic resonance venography (MRV), as well as neck vessel Doppler ultrasound, revealed additional critical findings. These included the resolution of the abscess, thrombosis in both the left internal jugular vein (IJV) and the internal carotid artery (ICA), spontaneous closure of the AV fistula, chronic infarcts in the left frontal lobe and the territory of the left middle cerebral artery (MCA), a normal status of the circle of Willis, and a reduction in the caliber of various arteries and veins. There was persistence of the thrombotic aneurysm and left vocal cord paralysis.\u003c/p\u003e\n\u003cp\u003eFigures 1-3. Coronal MIP, 3D reconstruction, and sagittal MIP images of the neck showing a bilobed aneurysm of the left carotid bulb extending to the internal carotid artery, with a surrounding hematoma. 3D reconstruction also revealed left sigmoid sinus thrombosis. The ECA was normal.\u003c/p\u003e\n\u003cp\u003eMultidisciplinary management, involving specialists from various medical fields to ensure comprehensive care and optimal treatment\u0026nbsp;outcomes, was undertaken to address the complex nature of the patient\u0026apos;s condition.\u003c/p\u003e\n\u003cp\u003e\u003cem\u003eMedical Management:\u003c/em\u003e Upon suspicion of LS, broad-spectrum intravenous antibiotics, including ceftriaxone and metronidazole, and anticoagulation therapy were initiated under close monitoring.\u003c/p\u003e\n\u003cp\u003e\u003cem\u003eSurgical management of the aneurysm\u0026nbsp;\u003c/em\u003erevealed several critical findings. The aneurysm sac extended from the bifurcation of the left common carotid artery, affecting both the external and internal carotid arteries. Notably, the IJV was embedded within the wall of the sac. There was extensive thrombosis throughout the entire length of the left IJV and in the left ICA. Additionally, cranial nerves (CNs) IX, X, and XII could not be clearly delineated from the wall of the aneurysm sac or the surrounding inflammatory tissue, complicating the surgical approach. Furthermore, there was no backflow from the left internal carotid artery, indicating significant vascular impairment. During the operation, the surgical team evacuated a massive thrombus and ligated the left common carotid artery at its bifurcation.\u003c/p\u003e\n\u003cp\u003ePostoperatively, the patient quickly regained consciousness, showing no new neurological deficits. Remarkably, just five days after the operation, the patient was evaluated and considered ready for discharge, after which he subsequently returned home. A follow-up examination two weeks later demonstrated a complete recovery of function in the CNIX, CNX and CNXII groups as his voice returned to normal. Nevertheless, the patient continued to experience persistent left hemiparesis, which was attributed to an infarct in the MCA.\u003c/p\u003e"},{"header":"Discussion","content":"\u003cp\u003eOur case report\u0026nbsp;describes\u0026nbsp;a unique manifestation of LS, characterized by the rare complication of an internal carotid artery and internal jugular vein fistula, followed by the development of septic thrombosis leading to aneurysm formation and cerebral infarction.\u0026nbsp;This anomaly was observed following a periodontal abscess and tonsillitis, evolving into deep neck space, mediastinal, and chest abscesses, along with left IJV thrombophlebitis. This sequence of events underscores the critical nature of LS and its potential to progress to life-threatening complications, despite the initial administration of broad-spectrum antibiotics. The rarity of this presentation within the context of LS contributes significantly to the medical literature, highlighting the need for vigilance in cases of persistent oropharyngeal infection symptoms, especially when accompanied by vascular complications.\u003c/p\u003e\n\u003cp\u003eOnce labeled the 'forgotten disease' in the 1980s due to effective antibiotic use, LS, a deadly disease in the\u0026nbsp;preantibiotic\u0026nbsp;era, has experienced a documented increase in incidence mainly attributed to the\u0026nbsp;increase in\u0026nbsp;antibiotic resistance (6). Despite its rarity, the syndrome presents diagnostic challenges, necessitating a comprehensive, multidisciplinary approach for effective management (5,7,8). Our case underscores the crucial importance of early diagnosis and prompt initiation of targeted therapy to address potentially life-threatening complications (6).\u003c/p\u003e\n\u003cp\u003eDemographically, LS primarily affects young individuals, typically aged 15 to 24 years, with a peak incidence in the second and third decades of life and a slightly greater male incidence (male-to-female ratio of 2:1). However, it has been documented in individuals across a wide age range, from as young as 2 months to as old as 78 years (3,9). \u003cem\u003eFusobacterium necrophorum\u003c/em\u003e, identified as the causative agent in 10% of peritonsillar abscesses and 81% of LS cases, is an anaerobic bacterium prevalent in the oropharynx, genitourinary system, and gastrointestinal tract (5,8,10). Heightened suspicion should arise when oropharyngeal pharyngitis or periodontal abscess persists beyond 3 to 5 days and is accompanied by systemic or respiratory symptoms, lateral cervical pain, dysphagia, progression to sepsis or multiple pulmonary abscesses (5,8,9,11)\u003c/p\u003e\n\u003cp\u003eThe common presentation of LS is unilateral neck discomfort and inflammation at the anterior border of the sternocleidomastoid muscle after a recent throat infection (3,12). The detection of this syndrome may be easily overlooked or delayed since patients typically exhibit a palpable, tender cord (or 'cord sign') of the internal jugular vein in only 26% to 45% of cases (1,3,8,13). Furthermore, microbiology laboratories often encounter challenges in cultivating \u003cem\u003eFusobacterium necrophorum\u003c/em\u003e, as observed in our case. The conclusive identification of pathognomonic anaerobes through blood cultures in the laboratory may require several days for growth on specialized media that are not routinely utilized in most laboratory settings (9,10)\u003cstrong\u003e.\u003c/strong\u003e Moreover, the identification process may be impeded by the widespread use of antibiotics for outpatient treatment of pharyngitis (1,5,14).\u003c/p\u003e\n\u003cp\u003eThe peritonsillar abscesses spread via\u0026nbsp;hematogenesis\u0026nbsp;or through the lymphatic system.\u0026nbsp;Venous\u0026nbsp;drainage of the palatine tonsils occurs\u0026nbsp;through\u0026nbsp;the peritonsillar plexus of veins,\u0026nbsp;which drains into the pharyngeal and lingual veins and ultimately into the internal jugular vein. The\u0026nbsp;palatine tonsils drain to the jugulodigastric node, a node of the deep cervical lymph nodes, where the posterior belly of the digastric muscle crosses the internal jugular vein (15). The most common metastatic abscess sites are the lung (empyema\u0026nbsp;or cavitary nodules), mediastinum, brain, and IJV (2,16). Other organs that may be affected include the bone/joint (septic arthritis), the soft tissue and muscle, intraabdominal abscesses in the liver or spleen, and the central nervous system (in the form of chronic\u0026nbsp;infarcts\u0026nbsp;or abscesses and/or meningitis)\u0026nbsp;(4,10,17,18). In our\u0026nbsp;patient,\u0026nbsp;the infection spread in the cervical, thoracic, or mediastinal spaces up to the dura sinus, and left IJV thrombosis and then left ICA thrombosis\u0026nbsp;resulted\u0026nbsp;in\u0026nbsp;chronic infarction of the\u0026nbsp;left MCA territory (19). The aneurysm at the left common\u0026nbsp;carotid artery\u0026nbsp;CCA-carotid bulb resulted from thrombophlebitis and mechanical pressure from the fistula, leading to damage\u0026nbsp;to\u0026nbsp;the left CCA wall and subsequent aneurysm formation, as typically described in the literature on aneurysm pathophysiology (20–23).\u0026nbsp;However, to the best of our knowledge, this is the first case of LS\u0026nbsp;involving\u0026nbsp;a\u0026nbsp;carotid-jugular vein\u0026nbsp;fistula,\u0026nbsp;a\u0026nbsp;left carotid bulb CCA and left ECA aneurysm, and extensive vascular thrombosis leading to cerebral infarction.\u003c/p\u003e\n\u003cp\u003eHistorically, LS diagnosis\u0026nbsp;has\u0026nbsp;relied on criteria such as primary head or neck infection, thrombosis of head/neck veins, and isolation of \u003cem\u003eFusobacterium necrophorum\u003c/em\u003e from blood cultures (2,5). However, evolving diagnostic criteria\u0026nbsp;currently\u0026nbsp;acknowledge challenges in isolating \u003cem\u003eFusobacterium necrophorum,\u003c/em\u003e making it nonmandatory, as a result of potential negative cultures resulting from empirical antibiotic therapy (10,17). Radiological investigations, particularly duplex ultrasonography, CT\u0026nbsp;scans, and magnetic resonance imaging, play a crucial role in diagnosis. Contrast-enhanced CT is considered optimal\u0026nbsp;because it offers\u0026nbsp;accessibility, cost-effectiveness, and visualization of adjacent structures (6,24). The radiological findings reported in other studies align with our case, where similar modalities were employed and yielded comparable results, confirming the diagnosis of LS.\u003c/p\u003e\n\u003cp\u003eOur patient received\u0026nbsp;ceftriaxone and metronidazole in combination with\u0026nbsp;anti-TB\u0026nbsp;therapy following the standard treatment protocol\u0026nbsp;because of\u0026nbsp;the high TB prevalence in\u0026nbsp;this\u0026nbsp;region. This combination was effective in treating extensive abscesses.\u0026nbsp;Although\u0026nbsp;neither Ceftriaxone nor anti-TB drugs are known for treating LS, evidence indicates that similar cephalosporins have been effective against LS due to their action on anaerobic bacteria (10)(25). Given the occurrence of beta-lactamase-producing \u003cem\u003eFusobacterium.\u0026nbsp;\u003c/em\u003eFor\u0026nbsp;\u003cem\u003enecrophorum\u003c/em\u003e strains resistant to\u0026nbsp;macrolides\u0026nbsp;and penicillin (6,26), the\u0026nbsp;literature recommends\u0026nbsp;the use of a beta-lactamase-resistant beta-lactam antibiotic in conjunction with\u0026nbsp;metronidazole, which is\u0026nbsp;initially\u0026nbsp;advised\u0026nbsp;for two weeks, transitioning to\u0026nbsp;metronidazole\u0026nbsp;alone for six weeks (13,14). This approach ensures deep penetration into fibrin clots and addresses potential resistance issues. The use of\u0026nbsp;antituberculosis\u0026nbsp;therapy underscores the diagnostic challenges of LS, a condition easily mistaken for others due to its severity and rarity, as well as the complexities involved in identifying the responsible bacteria.\u003c/p\u003e\n\u003cp\u003eThe use of prophylactic anticoagulant agents\u0026nbsp;for\u0026nbsp;managing LS remains a subject of considerable debate within the medical community. This controversy stems from the disparate findings reported in the literature, compounded by the disease's rarity, which inherently limits the feasibility of conducting statistically robust trials to assess the effectiveness of anticoagulation therapy comprehensively (27).\u0026nbsp;Despite these challenges, a consensus among several authors advocates for the utilization of anticoagulation\u0026nbsp;therapy in\u0026nbsp;LS patients\u0026nbsp;as a strategy to mitigate neurological complications.\u0026nbsp;This underscores the importance of a tailored approach that weighs the estimated risk of venous thromboembolism against potential contraindications (5,6,13,14). Specifically, the deployment of anticoagulant therapy is recommended for patients identified as high\u0026nbsp;risk, emphasizing a judicious evaluation of benefits and risks (28,29). In the case presented, the incorporation of an anticoagulant regimen is strongly corroborated by evidence from the literature, which posits that anticoagulation, particularly in scenarios of carotid thrombosis, is associated with a diminished recurrence of ischemic events (30,31).\u003c/p\u003e\n\u003cp\u003eIn the context of LS, surgical procedures have predominantly focused on the drainage of pus, as indicated by the literature (10). To our knowledge, the present case is unprecedented in necessitating vascular surgery as a part of LS management. Surgery was required to mitigate the effect of the mass on cranial nerves IX and XII. The standard of care for treating infected carotid artery pseudoaneurysms predominantly involves traditional open repair. This approach includes aneurysmectomy, debridement of surrounding infected tissues, and carotid artery ligation (32–35). Revascularization of the ICA is generally considered only in cases where it is deemed essential, such as in patients with bilateral ICA disease (36,37). This cautious approach is due to the potential risk of embolic events in the brain (38,39). In line with recommendations from several studies, our patient underwent ligation of both the left ICA and the ECA without attempting revascularization of the left IJV and ICA, owing to thrombosis. During the repair process, we observed evidence of spontaneous closure of the internal jugular vein-carotid artery (IJV-ICA) fistula, which was secondary to thrombosis in both the IJV and ICA. These observations are consistent with findings reported in the literature, specifically the spontaneous resolution of direct carotid-cavernous fistulas documented in 2019 (40,41)\u003c/p\u003e"},{"header":"Conclusion","content":"\u003cp\u003eOur case of Lemierre's syndrome, featuring an unusual presentation of an internal carotid artery and internal jugular vein fistula, emphasizes the critical importance of prompt recognition, a multidisciplinary treatment approach, and aggressive management to prevent life-threatening complications. This case highlights the atypical manifestations of Lemierre’s syndrome and stresses the need to consider it in patients with recent oropharyngeal infections who develop complex vascular symptoms.\u0026nbsp;This\u0026nbsp;study offers valuable insights into therapeutic strategies for managing complex cases of Lemierre’s syndrome, emphasizing the significance of personalized care, including targeted antibiotic therapy, judicious use of anticoagulation agents, and surgical intervention.\u003c/p\u003e\n\u003cp\u003e\u003cem\u003eRecommendations\u003c/em\u003e\u003c/p\u003e\n\u003col\u003e\n \u003cli\u003ePrompt Antibiotic Therapy: Early initiation of broad-spectrum antibiotics tailored based on microbiological culture findings.\u003c/li\u003e\n \u003cli\u003eSelective\u0026nbsp;anticoagulation: Case-by-case assessment for anticoagulation, especially in patients with significant vascular thrombosis.\u003c/li\u003e\n \u003cli\u003eIntegrated Care Approach: Collaborative effort among specialists, including those involved in infectious diseases, radiology, vascular surgery, otorhinolaryngology, and intensive care.\u003c/li\u003e\n\u003c/ol\u003e"},{"header":"Abbreviations","content":"\u003cp\u003eLS - Lemierre's Syndrome\u003c/p\u003e\n\u003cp\u003eIJV - Internal jugular vein\u003c/p\u003e\n\u003cp\u003eCT - Computed Tomography\u003c/p\u003e\n\u003cp\u003eMRI - magnetic resonance imaging\u003c/p\u003e\n\u003cp\u003eMCA - middle cerebral artery\u003c/p\u003e\n\u003cp\u003eICU - Intensive Care Unit\u003c/p\u003e\n\u003cp\u003eTB - Tuberculosis\u003c/p\u003e\n\u003cp\u003eCN - Cranial Nerve\u003c/p\u003e\n\u003cp\u003eRHZE - Rifampicin, Isoniazid, Pyrazinamide, Ethambutol\u003c/p\u003e\n\u003cp\u003eCCA - common carotid artery\u003c/p\u003e\n\u003cp\u003eICA - Internal carotid artery\u003c/p\u003e\n\u003cp\u003eECA - External carotid artery\u003c/p\u003e\n\u003cp\u003eMRA - magnetic resonance angiography\u003c/p\u003e\n\u003cp\u003eMRV - Magnetic Resonance Venography\u003c/p\u003e"},{"header":"Declarations","content":"\u003ch2\u003eAuthor Contribution\u003c/h2\u003e\u003cp\u003eL.W.K., J.A.M., and J.K. prepared and wrote the main manuscript text. P.K. prepared figures 1-3. All authors, including M.S., F.H., M.A., G.O., C.I., and S.N.,T.O. alongside those mentioned above, reviewed the manuscript.\u003c/p\u003e\u003ch2\u003eAcknowledgement\u003c/h2\u003e\u003cp\u003eThe authors sincerely thank Kathryn Parker for her valuable contribution to reviewing the manuscript, which significantly improved its quality. They appreciate her time and expertise in maintaining the integrity and accuracy of the research.\u003c/p\u003e\n\u003cp\u003e\u003cem\u003eEthics Statement and\u0026nbsp;\u003c/em\u003e\u003cem\u003eInformed Consent:\u003c/em\u003e Ethical approval was waived, and the patient provided written permission to publish this case report and associated images.\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\u003cli\u003e\u003cspan\u003eValerio L, Corsi G, Sebastian T, Barco S. 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Spontaneous resolution of direct carotid-cavernous fistulas: case series and literature review. Interventional Neuroradiology [Internet]. 2019 Feb 1 [cited 2024 Feb 11];25(1):71. Available \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003efrom:/pmc/articles/PMC6378520/\u003c/span\u003e\u003cspan address=\"http://from:/pmc/articles/PMC6378520/\" targettype=\"URL\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eSpontaneous resolution of direct carotid-. cavernous fistulas: case series and literature review - PMC [Internet]. [cited 2024 Feb 11]. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC6378520/\u003c/span\u003e\u003cspan address=\"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6378520/\" targettype=\"URL\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e.\u003c/span\u003e\u003c/li\u003e\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":true,"hideJournal":true,"highlight":"","institution":"","isAcceptedByJournal":false,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"researchsquare","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":true,"externalIdentity":"","sideBox":"","snPcode":"","submissionUrl":"/submission","title":"Research Square","twitterHandle":"researchsquare","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"","reportingPortfolio":"","inReviewEnabled":false,"inReviewRevisionsEnabled":true},"keywords":"Lemierre's syndrome, Septic thrombophlebitis, Pharyngeal space.","lastPublishedDoi":"10.21203/rs.3.rs-4283446/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-4283446/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003cp\u003e\u003cstrong\u003eBackground: \u003c/strong\u003eLemierre's syndrome (LS) is a rare yet potentially life-threatening complication of oropharyngeal infections that is typically characterized by septic thrombophlebitis of the internal jugular vein and is commonly associated with \u003cem\u003eFusobacterium necrophorum\u003c/em\u003e. Despite its recognition since 1936, LS can present atypically, as seen in our case where a patient exhibited a rare complication of an internal carotid artery and jugular vein fistula. This case highlights the diagnostic and therapeutic challenges that can arise and underscores the importance of both a broader awareness of LS and the necessity for a multidisciplinary approach.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eCase Presentation: \u003c/strong\u003eA 42-year-old male who was initially diagnosed with a periodontal abscess and tonsillitis later developed internal jugular vein thrombophlebitis. This progressed to an internal carotid to internal jugular vein fistula, leading to septic thrombosis of the left internal carotid and jugular vein and eventually culminating in a carotid bulb aneurysm and subsequent cerebral infarct. The patient also experienced septic emboli, resulting in lung and mediastinal abscesses. Treatment involved intravenous antibiotics, anticoagulant therapy, and vascular repair, yielding a favorable outcome.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConclusions:\u003c/strong\u003e This case underscores the intricate nature of LS, showing its potential to progress to severe septic complications, including rare vascular issues and cerebral infarction. The diagnostic complexities and therapeutic interventions emphasize the critical importance of early identification, prompt diagnosis, and multidisciplinary management of LS to prevent life-threatening outcomes.\u003c/p\u003e","manuscriptTitle":"Lemierre's Syndrome: Atypical Presentation of the Internal Carotid Artery and Jugular Vein Fistula—CaseReport and Review","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2024-04-25 16:57:59","doi":"10.21203/rs.3.rs-4283446/v1","editorialEvents":[{"type":"communityComments","content":0}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"researchsquare","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":true,"externalIdentity":"","sideBox":"","snPcode":"","submissionUrl":"/submission","title":"Research Square","twitterHandle":"researchsquare","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"","reportingPortfolio":"","inReviewEnabled":false,"inReviewRevisionsEnabled":true}}],"origin":"","ownerIdentity":"f7cf5de5-df70-4832-b5e0-a898e427b615","owner":[],"postedDate":"April 25th, 2024","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"posted","subjectAreas":[],"tags":[],"updatedAt":"2024-10-24T02:08:38+00:00","versionOfRecord":[],"versionCreatedAt":"2024-04-25 16:57:59","video":"","vorDoi":"","vorDoiUrl":"","workflowStages":[]},"version":"v1","identity":"rs-4283446","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-4283446","identity":"rs-4283446","version":["v1"]},"buildId":"8U1c8b4HqxoKbykW_rLl7","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}