Lemierre syndrome complicated by bronchopleural fistula.

We present a 19-year-old woman, a case of Lemierre syndrome, who presented with fever, sore throat, and left shoulder pain. Imaging revealed a thrombus in the right internal jugular vein, multiple nodular shadows below both pleura with some cavitations, right lung necrotizing pneumonia, pyothorax, abscess in the infraspinatus muscle, and multiloculated fluid collections in the left hip joint. After inserting a chest tube and administering urokinase for the pyothorax, a bronchopleural fistula was suspected. The fistula was identified based on clinical symptoms and computed tomography scan findings. If a bronchopleural fistula is present, thoracic lavage should not be performed as it may cause complications such as contralateral pneumonia due to reflux.

Elderly onset atypical Lemierre's syndrome concurrent with a rheumatoid vasculitis sacral ulcer infection: a case report.

BACKGROUND: Typical Lemierre's syndrome is usually secondary to an oropharyngeal infection. Recently, several cases following a primary infection site other than the oropharynx have been reported as atypical Lemierre's syndrome; although, these primary lesions are limited to the head and neck. This is the first case potentially sequential to infectious foci outside the head and neck. CASE PRESENTATION: We describe an atypical Lemierre's syndrome in a 72-year-old woman with rheumatoid arthritis, which occurred during the treatment of Streptococcus anginosus bacteremia acquired from a sacral ulcer infection related to rheumatoid vasculitis. At first, the symptoms resolved after the initial administration of vancomycin for the bacteremia caused by methicillin-resistant Staphylococcus aureus and Streptococcus anginosus that entered via a sacral ulcer. On the 8th day, the patient developed a fever of 40 °C and unexpectedly required 10 L of oxygen due to rapid deterioration of oxygenation temporarily. Immediately contrast-enhanced computed tomography was performed to investigate systemic thrombosis including pulmonary embolism. Afterward, the newly formed thrombi at the right external jugular vein, bilateral internal jugular veins, and the right small saphenous vein were detected, and apixaban was started. On the 9th day, the patient again had an intermittent fever of 39.7 °C, and continuous Streptococcus anginosus bacteremia was revealed; subsequently, clindamycin was administered. On the 10th day, she developed a left hemothorax; consequently, apixaban was discontinued, and a thoracic drain was inserted. She repeatedly had an intermittent fever of 40.3 °C, and contrast-enhanced computed tomography detected an abscess formation at the left parotid gland, pterygoid muscle group, and masseter muscle. After Lemierre's syndrome was diagnosed in combination with the abovementioned jugular vein thrombus, clindamycin was replaced with meropenem, and vancomycin was increased. Swelling of the lower part of the left ear became prominent with delay and peaked at approximately the 16th day. The subsequent treatment course was favorable, and she was discharged on the 41st day. CONCLUSION: Clinicians should consider Lemierre's syndrome as the differential diagnosis of internal jugular vein thrombosis occurring during sepsis, even though an antibiotic is administered or a primary infection site is anything besides the oropharynx.

Delayed diagnosis of Lemierre's syndrome in a patient with severe coronavirus disease 2019: importance of comprehensive oral and neck examination - a case report.

BACKGROUND: Given the widespread prevalence of the coronavirus disease 2019 (COVID-19), oral and neck examinations tend to be avoided in patients with suspected or confirmed COVID-19. This might delay the diagnosis of conditions such as Lemierre's syndrome, which involves symptoms resembling COVID-19-related throat manifestations. CASE PRESENTATION: A 24-year-old man without any underlying conditions was diagnosed with COVID-19 7 days before presentation. He was admitted to another hospital 1 day before presentation with severe COVID-19 and suspected bacterial pneumonia; accordingly, he was started on treatment with remdesivir and meropenem. Owing to bacteremic complications, the patient was transferred to our hospital for intensive care. On the sixth day, the patient experienced hemoptysis; further, a computed tomography (CT) scan revealed new pulmonary artery pseudoaneurysms. Successful embolization was performed to achieve hemostasis. In blood cultures conducted at the previous hospital, Fusobacterium nucleatum was isolated, suggesting a cervical origin of the infection. A neck CT scan confirmed a peritonsillar abscess and left internal jugular vein thrombus; accordingly, he was diagnosed with Lemierre's syndrome. The treatment was switched to ampicillin/sulbactam, based on the drug susceptibility results. After 6 weeks of treatment, the patient completely recovered without complications. CONCLUSION: This case highlights the significance of thorough oral and neck examinations in patients with suspected or diagnosed COVID-19 for the detection of throat and neck symptoms caused by other conditions.

Common laboratory tests and their correlation with the clinical presentation and prognosis of Lemierre syndrome.

INTRODUCTION: Lemierre syndrome is a thromboembolic complication following an acute bacterial infection of the head/neck area, often due to anaerobes. Data on the prognostic role of laboratory parameters is lacking. METHODS: We analyzed individual-patient level data from a multinational cohort of patients with Lemierre-syndrome. Patients had an infection in the head/neck area, and contiguous vein thrombosis or septic embolism, irrespective of the causal pathogen. We studied the patterns of white blood cell count, platelet count, and C-reactive protein concentration investigating their association with baseline characteristics and in-hospital clinical outcomes (septic embolism, major bleeding, all-cause death). RESULTS: A total of 447 (63%) patients had complete data for analysis. White blood cells were elevated across all subgroups (median 17 × 103/µL; Q1-Q3:12-21). Median platelet count was 61 × 103/µL (Q1-Q3:30-108) with decreasing levels with increasing age. Males, patients with renal failure or cardiopulmonary impairment, and those with typical Lemierre syndrome (tonsillitis, septic thromboembolism, positivity for Fusobacterium spp.) had the lowest platelet count. Median C-reactive protein was 122 (Q1-Q3:27-248) mg/L with higher values in patients who also had more severe thrombocytopenia. The overall risk of complications was similar across subgroups of patients stratified according to white blood cell and C-reactive protein levels. Patients in the lowest third of platelet count (<42 × 103/µL) had the highest rate of complications (26%), as opposed to those in the highest third (11%), notably septic embolic events. CONCLUSIONS: Common laboratory tests correlate with the clinical presentation of Lemierre syndrome. However, extreme values did not appear to be prognostically relevant for in-hospital complications and potentially able to improve clinical management.

High risk and low prevalence diseases: Lemierre's syndrome.

INTRODUCTION: Lemierre's syndrome is a serious condition that carries with it a high rate of morbidity and even mortality. OBJECTIVE: This review highlights the pearls and pitfalls of Lemierre's syndrome, including presentation, diagnosis, and management in the emergency department (ED) based on current evidence. DISCUSSION: Lemierre's syndrome is a condition marked by septic thrombophlebitis of the internal jugular vein (IJV), with the classic triad of pharyngotonsillitis, IJV thrombosis, and septic emboli resulting in metastatic abscess. It typically begins as pharyngitis, often caused by Fusobacterium necrophorum. Patients most commonly present with fever, recently diagnosed pharyngitis, and neck pain or swelling. Septic emboli may affect multiple organ systems, most commonly the pulmonary system. The disease should be considered in patients with prolonged symptoms of pharyngitis, pharyngitis that improves but then worsens, critically ill patients with pharyngitis, patients with pharyngitis and infection at a secondary site, and neck signs/symptoms. Diagnosis includes throat and blood cultures, as well as imaging to include computed tomography of the neck and chest with intravenous contrast. Additional imaging of other areas should be performed as clinically indicated. Initial management includes hemodynamic stabilization with intravenous fluids and vasopressors as needed, as well as broad-spectrum antibiotics. Anticoagulation for the primary thrombus and possible septic emboli is controversial and should be considered in a multidisciplinary approach with admission. CONCLUSIONS: An understanding of Lemierre's syndrome can assist emergency clinicians in diagnosing and managing this potentially deadly disease.

Short blood culture time-to-positivity in Fusobacterium necrophorum bacteremia is associated with Lemierre's syndrome.

The usefulness of blood culture time-to-positivity (TTP) in the diagnosis of endovascular infections has previously been shown. Here, we investigated TTP in 41 patients with F. necrophorum bacteraemia and found an association between short TTP and Lemierre's syndrome (p = 0.026).

Patients with Lemierre syndrome have a high risk of new thromboembolic complications, clinical sequelae and death: an analysis of 712 cases.

BACKGROUND: Lemierre syndrome is characterized by head/neck vein thrombosis and septic embolism usually complicating an acute oropharyngeal bacterial infection in adolescents and young adults. We described the course of Lemierre syndrome in the contemporary era. METHODS: In our individual-level analysis of 712 patients (2000-2017), we included cases described as Lemierre syndrome if these criteria were met: (i) primary site of bacterial infection in the head/neck; (ii) objectively confirmed local thrombotic complications or septic embolism. The study outcomes were new or recurrent venous thromboembolism or peripheral septic lesions, major bleeding, all-cause death and clinical sequelae. RESULTS: The median age was 21 (Q1-Q3: 17-33) years, and 295 (41%) were female. At diagnosis, acute thrombosis of head/neck veins was detected in 597 (84%) patients, septic embolism in 582 (82%) and both in 468 (80%). After diagnosis and during in-hospital follow-up, new venous thromboembolism occurred in 34 (5.2%, 95% CI 3.8-7.2%) patients, new peripheral septic lesions became evident in 76 (11.7%; 9.4-14.3%). The rate of either was lower in patients who received anticoagulation (OR: 0.59; 0.36-0.94), higher in those with initial intracranial involvement (OR: 2.35; 1.45-3.80). Major bleeding occurred in 19 patients (2.9%; 1.9-4.5%), and 26 died (4.0%; 2.7-5.8%). Clinical sequelae were reported in 65 (10.4%, 8.2-13.0%) individuals, often consisting of cranial nerve palsy (n = 24) and orthopaedic limitations (n = 19). CONCLUSIONS: Patients with Lemierre syndrome were characterized by a substantial risk of new thromboembolic complications and death. This risk was higher in the presence of initial intracranial involvement. One-tenth of survivors suffered major clinical sequelae.

Anticoagulation Strategies in the Management of Lemierre Syndrome: A Systematic Review of the Literature.

OBJECTIVE: To determine the optimal anticoagulation strategy in patients diagnosed with Lemierre Syndrome (LS). DATA SOURCES: A systematic review in accordance with PRISMA guidelines was conducted using PubMed, MEDLINE, Scopus, ProQuest, and CINAHL from January to April 2020. Search terms included "Lemierre Syndrome" AND "anticoagulation" NOT "prophylaxis" OR "atrial fibrillation," in addition to a list of parenteral and oral anticoagulants. Adult patients who developed a clot and required systemic anticoagulation as a result of LS were included in this review. STUDY SELECTION AND DATA EXTRACTION: A total of 4180 records were initially identified, though following the removal of duplicates and nonrelevant entries, 216 full-text articles were reviewed for inclusion; 13 articles were ultimately included. DATA SYNTHESIS: The majority (11/14) of patients developed thromboses of the internal jugular veins, which corresponds to the pathophysiology of LS. Anticoagulation strategies were varied in the included literature, though 12/14 patients initially received a parenteral product. Two patients received a direct-acting oral anticoagulant (DOAC) following either intravenous heparin or subcutaneous enoxaparin and had outcomes similar to patients transitioned to warfarin. RELEVANCE TO PATIENT CARE AND CLINICAL PRACTICE: Anticoagulation in LS is a clinical controversy because the thromboembolic events have rarely led to significant complications; thrombi typically resolve independently, and concerns for bleeding risks are well founded; however, this review indicates both the efficacy and safety of anticoagulation. CONCLUSIONS: Anticoagulation is both efficacious and safe in LS, including treatment using a DOAC. Although further studies are needed, clinicians should consider a duration of anticoagulation of 6 to 12 weeks.

Successful perioperative management of a case of infective endocarditis secondary to a Lemierre's syndrome variant with severe neurological manifestations.

Lemierre's syndrome (LS) is characterized by septic thrombophlebitis of the internal jugular vein with septicemia and metastatic infection following an oropharyngeal infection. LS is rare but can cause infective endocarditis (IE), complicating IE management. We report a case of IE secondary to thrombophlebitis in the left vertebral vein following pharyngitis (LS variant) with distinctively severe manifestations, including metastatic infection and severe neurological impairment with multiple cerebral infarctions. A pedunculated abscess was noted on the left ventricular free wall. Despite the patient's highly impaired consciousness level (i.e., comatose state), we performed early surgery to remove the abscess after excluding LS-related brain complications. Preoperative antibiotics included clindamycin to cover LS-related anaerobic bacteria, and thrombophlebitis required postoperative anticoagulation. By managing LS as well as IE, the infection was controlled, and the neurological status normalized. This report provides insights into the perioperative management of IE secondary to LS.

Lemierre Syndrome: Unusual Presentation With Superior Ophthalmic Vein Thrombosis.

ABSTRACT: Lemierre syndrome (LS) is a rare, but serious condition most commonly associated with oropharyngeal bacterial infection. Infection results in thrombophlebitis of the internal jugular vein and its tributaries. It usually affects previously healthy young adults. The authors report an unusual case of an elderly patient with penicillin allergy who developed Lemierre syndrome from an odontogenic infection (Streptococcus intermedius) resistant to clindamycin. She developed thrombosis of the internal jugular vein and superior ophthalmic vein resulting in visual changes. Treatment required incision and drainage, intravenous antibiotics and anticoagulation. Antibiotics were tailored to culture and sensitivities after testing response to a beta-lactam. With antibiotic resistance increasing, investigation of penicillin allergies is warranted for severe head and neck infections.

A rare, but life-threatening sore throat: A case of Lemierre's syndrome.

We present the case of a previously healthy, immunocompetent male with Lemierre's Syndrome. He presented with headache, sore throat and pyrexia. Initial blood tests revealed raised inflammatory markers and electrolyte abnormalities. Blood cultured Fusobacterium necrophorum. He developed respiratory distress and imaging confirmed lung abscess and empyema due to septic emboli. He required surgical drainage and a prolonged course of antibiotics. This case highlights the rare, but life-threatening condition of Lemierre's Syndrome. We discuss the importance of prompt recognition and early antibiotic therapy.

[Cervicofacial cellulitis complicated by internal jugular vein thrombosis]. / Cellulite cervico-faciale compliquée d'une thrombose veineuse jugulaire interne.

INTRODUCTION: Cervicofacial cellulitis (CFC) is one of the most common dermatological infectious emergencies, and related morbidity and mortality are non-negligible. PATIENTS AND METHODS: We describe the case of a 31-year-old male with left parotitis complicated by CFC with worsening over the previous week despite treatment with clindamycin and non-steroidal anti-inflammatory drugs. A cervicofacial computed tomography (CT) scan showed left internal jugular vein thrombosis (IJVT). Chest imaging showed no pleuropulmonary lesion, and bacteriological samples were negative. The patient received broad-spectrum antibiotic therapy and anticoagulants for 6 weeks. The outcome was quickly favourable. DISCUSSION: It is essential to perform a cervicofacial contrast-enhanced CT scan for any CFC to map the affected areas, detect the primary infection and screen for loco-regional complications such as IVJT. The occurrence of IJVT in an infectious setting should prompt screening for septic emboli, especially pulmonary emboli, as well as performance of a chest CT scan. The presence of septic emboli associated with a recent ENT infection and of IJVT or Fusobacteriumnecrophorum in blood cultures are indicative of Lemierre's syndrome. CONCLUSION: IJVT is an uncommon complication of CFC that occurs either alone or as part of Lemierre's syndrome.

Mastoiditis with concomitant Lemierre's syndrome.

Otalgia with mastoiditis is an infrequently encountered complication of acute otitis media (Pfaff and Moore, 2018). Even more rare is the development of infected jugular venous thrombosis, Lemierre's disease. We present a case of a six year-old girl with otalgia for over two months who presented to our Emergency Department (ED) with clinical mastoiditis, confirmed on CT scan, as well as an incidental diagnosis of complete thrombosis of the internal jugular (IJ) vein, Lemierre's syndrome. The true prevalence of Lemierre's from mastoiditis is difficult to discern. This clinical case highlights the importance of the consideration of these pathologies by the emergency physician.

Purpura fulminans with Lemierre's syndrome caused by Gemella bergeri and Eikenella corrodens: a case report.

BACKGROUND: Gemella bergeri is one of the nine species of the genus Gemella and is relatively difficult to identify. We herein describe the first case of septic shock due to a Gemella bergeri coinfection with Eikenella corrodens. CASE PRESENTATION: A 44-year-old Asian man with a medical history of IgG4-related ophthalmic disease who was prescribed corticosteroids (prednisolone) presented to our hospital with dyspnea. On arrival, he was in shock, and a purpuric eruption was noted on both legs. Contrast enhanced computed tomography showed fluid retention at the right maxillary sinus, left lung ground glass opacity, and bilateral lung irregular opacities without cavitation. Owing to suspected septic shock, fluid resuscitation and a high dose of vasopressors were started. In addition, meropenem, clindamycin, and vancomycin were administered. Repeat computed tomography confirmed left internal jugular and vertebral vein thrombosis. Following this, the patient was diagnosed with Lemierre's syndrome. Furthermore, he went into shock again on day 6 of hospitalization. Additional soft tissue infections were suspected; therefore, bilateral below the knee amputations were performed for source control. Cultures of the exudates from skin lesions and histopathological samples did not identify any pathogens, and histopathological findings showed arterial thrombosis; therefore it was concluded that the second time shock was associated with purpura fulminans. Following this, his general status improved. He was transferred to another hospital for rehabilitation. The blood culture isolates were identified as Gemella bergeri and Eikenella corrodens. Gemella bergeri was identified by matrix-assisted laser desorption ionization-time of flight mass spectrometry and confirmed by 16S rRNA gene sequencing later. The primary focus of the infection was thought to be in the right maxillary sinus, because the resolution of the fluid retention was confirmed by repeat computed tomography. CONCLUSIONS: Gemella bergeri can be the causative pathogen of septic shock. If this pathogen cannot be identified manually or through commercial phenotypic methods, 16S rRNA gene sequencing should be considered.

[Dysphagia and tonsillitis in a 25-year-old male patient : The nearly forgotten severe complication of a common ailment]. / Dysphagie und Angina tonsillaris bei einem 25-jährigen Patienten : Die fast vergessene schwere Komplikation einer häufigen Krankheit.

We report the case of a patient with a severe dysphagia accompanying progressive tonsillitis. The clinical examination supported the possibility of a severe septic soft tissue infection. The blood cultures revealed a largely anaerobic sepsis with Fusobacterium necrophorum. This unusual pathogen is the most common cause of Lemierre's syndrome. A duplex sonogram and magnetic resonance imaging (MRI) of the neck region and vessels suggested a thrombophlebitis of the left internal jugular vein with partial occlusion, so that Lemierre's syndrome could be diagnosed. The patient was treated with appropriate antibiotics according to the resistogram and also with rivaroxaban.

Lemierre's Syndrome as a Trigger for Secondary Hemophagocytic Lymphohistiocytosis.

Secondary hemophagocytic lymphohistiocytosis (sHLH) is a rare hyperinflammatory disorder caused by an aberrant immune response to a number of infectious or inflammatory conditions. Successful treatment of this potentially fatal condition requires early recognition and prompt therapy directed at the underlying trigger. In this report, we describe the clinical presentation, diagnostic findings, management, and outcome of a child with Lemierre's syndrome-associated sHLH. This is the first reported association of these 2 rare conditions and expands the number of known triggers for sHLH.

Mycotic Pseudoaneurysm of Internal Carotid Artery Secondary to Lemierre's Syndrome, How to Do It.

We report the case of a patient with internal carotid artery (ICA) mycotic pseudoaneurysm secondary to Lemierre's syndrome, urgently treated. A 75-year-old man presented to E.R. with a left swelling lesion of the neck and complaining left visions lost since that morning, fever, hypotension, and dyspnea. Since 15 days before developing symptoms, he had sore throat and odynophagia treated with a broad coverage of antibiotic therapy for presumed streptococcal pharyngitis. Preoperative computed tomography angiography images revealed a circular lesion, involving the common carotid artery, carotid bulb, and the proximal part of the internal and external carotid arteries. A pseudoaneurysm of the ICA was detected, and the jugular vein was compressed. A Cormier carotid vein graft bypass was performed. Lemierre's syndrome is a rare syndrome, but it is rarer the carotid artery pseudoaneurysm secondary to Lemierre's syndrome. Surgical treatment is safe and durable in patients with severe infection involving the neck.