Association of pathogenic determinants of Fusobacterium necrophorum with bacteremia, and Lemierre's syndrome.

Fusobacterium necrophorum is a Gram-negative anaerobic bacterium responsible for localized infections of the oropharynx that can evolve into bacteremia and/or septic thrombophlebitis of the jugular vein or peritonsillar vein, called Lemierre's syndrome. To identify microbial genetic determinants associated with the severity of this life-threatening disease, 70 F. necrophorum strains were collected and grouped into two categories according to the clinical presentation: (i) localized infection, (ii) bacteremia with/without Lemierre's syndrome. Comparative genomic analyses revealed two clades with distinct genetic content, one clade being significantly enriched with isolates from subjects with bacteremia. To identify genetic determinants contributing to F. necrophorum pathogenicity, genomic islands and virulence factor orthogroups (OVFs) were predicted. The presence/absence profiles of OVFs did not group isolates according to their clinical category, but rather according to their phylogeny. However, a variant of lktA, a key virulence factor, with a frameshift deletion that results in two open reading frames, was associated with bacteremia. Moreover, a genome-wide association study identified three orthogroups associated with bacteremic strains: (i) cas8a1, (ii) a sodium/solute symporter, and (iii) a POP1 domain-containing protein. Further studies must be performed to assess the functional impact of lktA mutation and of these orthogroups on the physiopathological mechanisms of F. necrophorum infections.

Lemierre Syndrome: Clinical Challenges And The Anticoagulation Dilemma In Adolescent Care.

Lemierre syndrome is a rare, life-threatening condition characterized by an acute otorhinolaryngologic infection with septic thrombophlebitis of the internal jugular vein and septic embolism, particularly to the lungs. We describe a case of a previously healthy 15-year-old female patient who initially presents fever and odynophagia but quickly develops neck and pleuritic chest pain. Computed tomography was performed and the radiological findings confirmed the diagnosis of a Lemierre syndrome. She was managed with antibiotics, anticoagulant for three days and symptomatic treatment, with a gradually improving condition. After 17 days of hospitalisation, due to reappearance of pleuritic pain, a new imaging assessment was performed and showed additional septic emboli in the lungs, which prompted the reintroduction of anticoagulant therapy. Awareness of the existence of this syndrome is essential to ensure a radiological evaluation with computed tomography and thus timely diagnosis and treatment.

Epstein-Barr virus as promoter of Lemierre syndrome: systematic literature review.

PURPOSE: To investigate a possible link between acute Epstein-Barr virus infection and Lemierre syndrome, a rare yet life-threatening infection. METHODS: A systematic review was conducted adhering to the 2020 Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines. Diagnosis criteria for Lemierre syndrome were established, and data extraction encompassed demographic data, clinical, and laboratory information. RESULTS: Out of 985 initially identified papers, 132 articles were selected for the final analysis. They reported on 151 cases of Lemierre syndrome (76 female and 75 male patients with a median of 18 years) alongside interpretable results for Epstein-Barr virus serology. Among these, 38 cases (25%) tested positive for acute Epstein-Barr virus serology. There were no differences in terms of age, sex, or Fusobacterium presence between the serologically positive and negative groups. Conversely, instances of cervical thrombophlebitis and pulmonary complications were significantly higher (P = 0.0001) among those testing negative. The disease course was lethal in one case for each of the two groups. CONCLUSIONS: This analysis provides evidence of an association between acute Epstein-Barr virus infection and Lemierre syndrome. Raising awareness of this link within the medical community is desirable.

Surgical intervention of Lemierre's syndrome: a case report and review of the literature.

BACKGROUND: Lemierre's syndrome is a fatal and rare disease that is typically characterized by oropharyngeal infection and internal jugular vein thrombosis. Timely institution of appropriate antibiotics is the standard treatment. CASE PRESENTATION: The authors report a case of Lemierre's syndrome. A 67-year-old male patient of Han ethnicity in China suffered from a large inflammatory neck mass involving left internal jugular vein thrombosis diagnosed as Lemierre's syndrome and finally cured by surgical treatment. In addition, a literature review was carried out through PubMed using the terms "Lemierre's syndrome/disease and review, meta-analysis or retrospective study" and "Lemierre's syndrome/disease and internal jugular vein". This search yielded six articles that recorded surgical methods such as drainage, craniotomy, tooth extraction, and ligation of the occluded vein to give clinicians more ideas about the treatment of the Lemierre's syndrome. CONCLUSION: This is the first review to summarize the conditions under which surgical treatment are conducted. Additionally, this is the first report of such a large inflammatory neck mass that was completely cured by surgical resection and internal jugular vein ligation. The authors also offer several conclusions regarding surgical intervention in Lemierre's syndrome for the first time.

Lemierre's syndrome complicating multiple organ failure caused by Fusobacterium necrophorum subsp. funduliforme F1260: Case report and review.

We described a case of a 24-year-old man with multiple organ failure caused by Fusobacterium necrophorum subsp. funduliforme F1260. This is the first described case of Lemierre's syndrome with multiple organ failure due to F. necrophorum subsp. funduliforme F1260 in an adult in China. Our study highlights that there may be a risk of misdiagnosis based solely on typical manifestations of internal jugular vein thrombophlebitis, metastatic lesions, and F. necrophorum isolated from blood cultures or normally sterile sites. Clinicians should be cognizant of the potential utility of metagenomic next-generation sequencing in facilitating early pathogen detection in severe infections, thus enabling timely and appropriate administration of antibiotics to reduce mortality rates and improve prognosis.

Atypical presentation of Lemierre's syndrome caused by penicillin-susceptible Staphylococcus aureus in a patient with chronic stomatitis and COVID-19.

A healthy man in his late 20s was admitted to the emergency department due to a flare-up in his severe chronic stomatitis, along with flu-like symptoms. CXR showed multiple bilateral consolidations and subsequent CT revealed thrombosis of the left facial and internal jugular vein, together with septic embolism in both lungs. Blood cultures showed penicillin-susceptible Staphylococcus aureus The patient was diagnosed with Lemierre's syndrome, despite atypical bacteria and clinical presentation. During hospitalisation, he developed pulmonary empyema as a complication and was admitted for 4 weeks. During hospitalisation and after discharge, the patient was examined for multiple rheumatic, immunological and dermatological diseases, but no underlying cause for Lemierre's syndrome has been found. We present this case due to the rarity of its nature, with atypical clinical presentation and pathogen for Lemierre's syndrome, but with classic radiological findings.

Lemierre syndrome with pulmonary empyema caused by Prevotella intermedia.

Lemierre syndrome is a rare disease that is most often caused by Fusobacterium necrophorum We present a case caused by Prevotella intermedia in a young, healthy man, complicated by multiple cavitary lung lesions, loculated pleural effusions requiring chest tube placement and trapezius abscess. Our case highlights (a) P. intermedia as a rare cause of Lemierre syndrome and (b) clinical response to appropriate antimicrobial therapy may be protracted.

Case report: Metagenomics next-generation sequencing in the diagnosis of septic shock due to Fusobacterium necrophorum in a 6-year-old child.

Fusobacterium necrophorum (F. necrophorum) infection is rare in pediatrics. In addition, the detection time of F. necrophorum by blood culture is long, and the positive rate is low. Infection with F. necrophorum bacilli usually follows rapid disease progression, resulting in high mortality. In previous reports of F. necrophorum-related cases, the most dangerous moment of the disease occurred after the appearance of Lemierre's syndrome. We report an atypical case of a 6-year-old female patient who developed septic shock within 24 h of admission due to F. necrophorum infection in the absence of Lemierre's syndrome. F. necrophorum was identified in a blood sample by metagenomics next-generation sequencing (mNGS) but not by standard blood culture. The patient was finally cured and discharged after receiving timely and effective targeted anti-infection treatment. In the present case study, it was observed that the heightened virulence and invasiveness of F. necrophorum contribute significantly to its role as a primary pathogen in pediatric septic shock. This can precipitate hemodynamic instability and multiple organ failure, even in the absence of Lemierre's syndrome. The use of mNGS can deeply and rapidly identify infectious pathogens, guide the use of targeted antibiotics, and greatly improve the survival rate of patients.

Lemierre-like syndrome after soft tissue infection due to methicillin-resistant Staphylococcus aureus: A case report and literature review.

RATIONALE: Lemierre-like syndrome (LLS) is characterized by bacteremia, septic thrombophlebitis of the internal jugular vein, and metastatic abscesses. In contrast to classic Lemierre syndrome, sources of infection are not related to oropharyngeal infections, as are frequent soft tissue infections. In recent years, Staphylococcus aureus has been identified as an emergent pathogen that causes this syndrome. The mortality rate of LLS caused by this pathogen is approximately 16%. Timely diagnosis, antibiotic treatment, and infection control are the cornerstones to treat LLS. Anticoagulant therapy as adjuvant treatment remains controversial. PATIENT CONCERNS: A 31-year-old woman from California, United States (US), was admitted to the emergency room with a history of 2 days of fever and severe throbbing pain in the left cervical region. Thorax and neck CT tomography revealed confluent cavities suggestive of septic embolism in the lungs and a filiform thrombus in the lumen of the left internal jugular vein, with moderate swelling of the soft and muscular tissues. Methicillin-resistant Staphylococcus aureus (MRSA) was isolated from the blood culture. DIAGNOSIS: The thrombus in the internal jugular vein associated with cellulitis in the neck and multiple cavitary lesions in the lungs support the diagnosis of LLS caused by MRSA with septic embolization. INTERVENTIONS: During treatment, the patient received vancomycin IV for 25 days and returned to the US with linezolid orally. In addition, assisted video-thoracoscopy and bilateral mini-thoracotomy with pleural decortication were performed for infectious source control, where 1700cc of purulent pleural fluid was drained. OUTCOMES: The patient was discharged with optimal evolution. LESSONS: LLS should be suspected in patients with skin and soft tissue infections who develop thrombosis or metastatic infections. MRSA infections should be considered in patients from areas where this pathogen is prevalent.

Lemierre syndrome: a diagnostic dilemma of critical care in the post-COVID era.

Lemierre syndrome (LS) is referred to as the 'forgotten Disease' owing to its rarity in the postantibiotic era with an estimated yearly incidence of 1/million population. The classic triad of LS includes internal jugular vein thrombosis, oropharyngeal infection and metastatic septic emboli. We present a case of typical LS with Fusobacterium and Prevotella infection, presenting with peritonsillar abscess and jugular vein thrombosis complicated by sepsis, acute hypoxic respiratory failure due to multiple pulmonary emboli and severe thrombocytopaenia in the absence of disseminated intravascular coagulation.

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.

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.

Septic thrombophlebitis of portal and splenic vein secondary to Fusobacterium nucleatum: A case report of an abdominal variant of lemierre syndrome.

RATIONALE: Septic thrombophlebitis of the internal jugular vein also known as Lemierre syndrome occurs secondary to an oropharyngeal infection often leading to septic embolisms to distant sites. Anaerobic gram-negative bacillus, Fusobacterium nucleatum and Fusobacterium necrophorum are commonly isolated organisms. Fusobacterium species has also been reported to complicate an intra-abdominal infection leading to septic thrombophlebitis of portal vein also known as pylephlebitis or abdominal variant of lemierre syndrome. PATIENT CONCERNS: The patient was a middle-aged female patient with chief complaints of abdominal discomfort, intermittent fever and vomiting for one month. DIAGNOSES: The final diagnosis was septic thrombophlebitis of portal and splenic vein secondary to Fusobacterium nucleatum. INTERVENTIONS: Patient was managed with broad spectrum intravenous antibiotics with coverage against gram-negative bacilli, anaerobes, and aerobic streptococcus species with therapeutic anticoagulation. OUTCOMES: Patient gradually improved and was discharged on oral apixaban. She was instructed to follow up with gastrointestinal specialist upon discharge in anticipation of the need for liver transplant in future. LESSONS: Due to its high mortality and associated long term disease morbidity, clinicians should always strive towards early diagnosis and treatment of the condition with involvement of multidisciplinary teams.

A case of Lemierre's syndrome causing panhypopituitarism, cavernous sinus thrombosis, ischaemic stroke and pulmonary embolism.

Infection is a rare cause of panhypopituitarism and has not been reported in the context of Lemierre's syndrome. We present the case of a previously well 19-year-old man, who presented acutely unwell with meningitis and sepsis. Fusobacterium necrophorum was isolated from peripheral blood cultures and identified on cerebrospinal fluid with 16S rDNA Polymerase Chain Reaction (PCR). Imaging demonstrated internal jugular vein thrombosis with subsequent cavernous venous sinus thrombosis. Pituitary function tests were suggestive of panhypopituitarism. The patient was diagnosed with Lemierre's syndrome complicated by meningitis, cavernous sinus thrombosis, base of skull osteomyelitis, ischaemic stroke and panhypopituitarism. He was treated with 13 weeks of intravenous antibiotics followed by 3 weeks of oral amoxicillin, and anticoagulated with dalteparin then apixaban. His panhypopituitarism was managed with hydrocortisone, levothyroxine and desmopressin.

Lung abscess as a complication of Lemierre Syndrome in adolescents: a single center case reports and review of the literature.

BACKGROUND: Fusobacterium necrophorum is an anaerobic, gram-negative, non-motile, filamentous, non-spore forming bacillus found in the oral cavity, gastrointestinal tract, and female genital tract, responsible of a rare disease named Lemierre Syndrome, characterized by septic thrombophlebitis of the internal jugular vein, which mainly affects previously healthy adolescents and young adults; some risk factors are reported, as smoking or primary viral or bacterial infection leading to the disruption of mucosa. The syndrome originates commonly from an upper respiratory infection such as pharyngotonsillitis, acute otitis media, cervical lymphadenitis, sinusitis, or odontogenic abscess, and may result in multiorgan metastasis, more frequently leading to pulmonary complications, especially lung abscesses. CASE PRESENTATION: We describe two cases of adolescents with atypical Lemierre Syndrome evaluated in a tertiary care center, one with a confirmed infection by Fusobacterium necrophorum and one with a presumptive diagnosis based on clinical features, who developed lung abscesses needing a prolonged antibiotic course and hospitalization. Of interest, both were user of electronic cigarette, configuring a possible new risk factor. The proper diagnosis of Lemierre Syndrome is often difficult to establish, so a high degree of suspicion is needed, especially in the case of lung abscesses in otherwise healthy adolescents. CONCLUSION: The current study will contribute to providing insight into Lemierre Syndrome clinical presentation and management in adolescents, promoting awareness for a rare but potentially fatal disease. Moreover, it suggests a possible relationship between Lemierre syndrome and the use of electronic cigarette, that should be investigated by future studies.

Lemierre syndrome.

Lemierre's syndrome is rare and characterized by an oropharyngeal infection with thrombophlebitis of the internal jugular vein (IJV). Septic microemboli can spread to the lungs or abdomen. This case describes a patient who presented with a sore throat, unilateral swelling on the neck and diffuse abdominal pain. Imaging showed a thrombus in the right IJV. The patient developed septic shock and was treated with antibiotics, anticoagulants, and intensive care support. The most common causative organism is Fusobacterium necrophorum. Early diagnosis and treatment are crucial for reducing mortality.

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.