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: 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.

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.

[Anticoagulation in a patient with Lemierre syndrome and pulmonary septic embolisms]. / Anticoagulación en paciente con síndrome de Lemierre y embolias sépticas pulmonares.

Lemierre's syndrome, also called septic thrombophlebitis of the internal jugular vein, necrobacillosis or postanginal sepsis, is an infection that begins in the oropharyngeal space, is complicated by septic thrombophlebitis of the internal jugular vein and infectious metastases. The rapid progression to serious clinical conditions that compromise the patient's life and its low frequency justify the disclosure of clinical cases. We present the case of a 27-year-old woman who developed facial edema and trismus 48 hours after completing treatment with phenoxymethylpenicillin for an odontogenic infection. An angio-CT of the craniofacial massif revealed an extensive thrombus in the internal jugular vein and a computed tomography of the chest showed septic pulmonary emboli. Treatment consisted of broad-spectrum intravenous antibiotics and early anticoagulation.

El síndrome de Lemierre, también denominado tromboflebitis séptica de la vena yugular interna, necrobacilosis o sepsis postanginal es una infección que inicia en el espacio orofaríngeo, se complica con tromboflebitis séptica de la vena yugular interna y metástasis infecciosas. La rápida progresión a cuadros clínicos graves que comprometen la vida del paciente y su baja frecuencia justifican la divulgación de casos clínicos. Se presenta el caso de una mujer de 27 años de edad, que a las 48 horas de completar el tratamiento con fenoximetilpenicilina por una infección odontógena evolucionó con edema facial y trismus. En la angio-TC de macizo craneofacial se evidenció extenso trombo en la vena yugular interna y en la tomografía computarizada de tórax, embolias sépticas pulmonares. El tratamiento consistió en antibióticos endovenosos de amplio espectro y anticoagulación de manera precoz.

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.

Severe atypical Lemierre syndrome caused by methicillin-sensitive Staphylococcus aureus: Two pediatric case reports.

BACKGROUND: Lemierre syndrome is typically associated with ear, nose, and throat (ENT) infections caused by Fusobacterium necrophorum. Since 2002, cases of atypical Lemierre-like syndrome secondary to Staphylococcus aureus have been reported. CASES: We report two pediatric cases of atypical Lemierre syndrome with a similar presentation: exophthalmia, absence of pharyngitis, metastatic lung infection, and intracranial venous sinus thrombosis. Both patients had a favorable outcome following treatment with antibiotics, anticoagulation, and corticosteroids. CONCLUSION: Regular therapeutic monitoring of antibiotic levels helped to optimize antimicrobial treatment in both cases.

Atypical thrombophlebitis patterns in head and neck infections.

Thrombophlebitis is a known complication of head and neck infection, which can result in propagation of infected thrombi and associated complications. While antegrade/downstream propagation of thrombus is well described-the Lemierre syndrome-thrombophlebitis can spread in atypical patterns which has specific diagnostic and treatment considerations. In this series, we highlight 6 cases of atypical thrombophlebitis patterns complicating head and neck infection, 5 of which extend intracranially. Through these pathways, head and neck infections such as oral cavity, oropharynx, or skin can be the culprit of remote, including intracranial, complications, which may be overlooked. Furthermore, understanding pathways of venous drainage and communication can help accurately diagnose thrombophlebitis patterns and associated complications.

Forget-me-not: Lemierre's syndrome, a case report.

Background: Familiarity breeds complacency. One case of sore throat can start to sound like another. However, even common viral and bacterial infections can lead to serious sequelae. Clinicians cannot make a diagnosis if they do not consider it in their differential.Case: The presentation - and subsequent hospitalization - of a septic 19 year-old male college student is described. Despite brief interval improvement, he became hypoxic one week after developing a sore throat and was ultimately diagnosed with Lemierre's syndrome (septic thrombophlebitis of the internal jugular vein) via blood cultures and advanced imaging.Conclusions: Though rare, Lemierre's syndrome carries a high mortality rate. It should be considered an emergent complication of head and neck infections, particularly in young adults.

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.

Atypical presentation of Lemierre syndrome in a young healthy man with acute jaundice.

This report presents a case of Lemierre syndrome caused by Fusobacterium necrophorum in a healthy young adult who presented atypically with shortness of breath and jaundice but no clinical or diagnostic evidence of thrombophlebitis. Due to this unusual presentation with jaundice, diagnosis was challenging and delayed. However, the patient was successfully initiated on a prolonged course of intravenous antibiotics; he required a period in the intensive care unit and was discharged without significant complications. This report aims to raise awareness of the diagnosis and treatment of this rare condition and to highlight both common and unusual presentations of the syndrome.

Paralysis: A Rare Presentation of Lemierre's Syndrome: A Case Report.

CASE: A patient presented with acute paralysis in the setting of undiagnosed Lemierre's syndrome. Streptococcus pyogenes was the causative organism leading to internal jugular vein thrombosis, sternocleidomastoid abscess, vertebral osteomyelitis, and epidural phlegmon. The patient was treated both surgically, with abscess evacuation, hemicorpectomy, and fusion, and medically, with antibiotics and anticoagulation. Postoperatively, the patient regained upper extremity strength and lower extremity sensation, but the paraplegia remained. CONCLUSION: Lemierre's syndrome is a rare complication of oropharyngeal infection with a constellation of findings including jugular thrombophlebitis, metastatic abscesses, and frequent involvement of anaerobic pathogens that may present acutely with paralysis.

[Lemierre's syndrome: a rare disease, but not forgotten]. / Het syndroom van Lemierre.

BACKGROUND: Lemierre's syndrome is a rare disease with different clinical and microbiological criteria. Without adequate treatment mortality is high. It often concerns healthy adolescents who present with common and usually harmless complaints such as fever and sore throat. CASE DESCRIPTION: We describe a 19-year-old male who was admitted to our hospital with a septic shock after a few days of fever and a sore throat. He was admitted to our intensive care unit. Further examination showed a jugular vein thrombosis and blood cultures showed Fusobacterium necrophorum; a classic presentation of Lemierre's syndrome. However, a jugular vein thrombosis is not a requirement for the diagnosis. CONCLUSION: We recommend considering Lemierre's syndrome when a young adult presents with fever and a sore throat. Early recognition of Lemierre's syndrome is very important to decrease complications and mortality.

Septic Thrombophlebitis of the Internal Jugular Vein in an Immunocompromised Patient with Lemierre Syndrome: A Case Report.

Lemierre syndrome is a rare complication of oropharyngeal infection, especially acute pharyngotonsillitis, associated with septicemia and thrombophlebitis of the internal jugular vein (IJV). We present the case of a 52-year-old patient who underwent liver transplantation and returned with symptoms of pain, redness and left cervical bulging 1 month after surgery. After investigation, the diagnosis of septic thrombophlebitis of the IJV was made. The patient responded well to treatment with antibiotic therapy and full anticoagulation. To the best of our knowledge, the present report is the first report of Lemierre syndrome in a post-liver transplant patient.

Cardiac and Cerebral Arterial Complications of Lemierre Syndrome: Results from a Systematic Review and Individual Patient Data Meta-analysis.

BACKGROUND: Lemierre syndrome is a potentially life-threatening disease, which affects otherwise healthy young adults and adolescents. It is characterized by acute neck vein thrombosis and septic embolism, usually complicating a bacterial infection. Data on the syndrome are sparse, particularly concerning arterial complications. METHODS: We evaluated the frequency and patterns of cerebral arterial and cardiac involvement ("arterial complications") in an individual patient level cohort of 712 patients, representing all cases described over the past 20 years in the medical literature who fulfilled the criteria: (1) bacterial infection in the neck/head site and (2) objectively confirmed thrombotic complication or septic embolism. The study outcomes were defined as all-cause in-hospital deaths and the occurrence of clinical sequelae at discharge or in the postdischarge period. RESULTS: A total of 55 (7.7%) patients had an arterial complication. The most frequent arterial complications were carotid involvement (52.7%), stroke (38.2%), and pericardial complications (20%). Patients with an arterial involvement were more likely to be treated with a greater number of antibiotics (23 vs. 10%) and to receive anticoagulation. In addition, patients with arterial complications had a greater risk of all-cause death (n = 20/600, 3.3% vs. n = 6/52, 12%; odds ratio [OR]: 3.8; 95% confidence interval [CI]: 1.5-9.9) and late clinical sequelae (n = 49/580, 9.0% vs. n = 15/46, 35%; OR: 5.2; 95% CI: 2.65-10.37). CONCLUSIONS: While Lemierre syndrome is known to be primarily characterized by venous thromboembolic events, our results suggest that local or distant arterial complications may occur in approximately one-tenth of patients and may be associated with a greater risk of long-term sequelae and death.

Ophthalmic complications of Lemierre syndrome.

PURPOSE: Lemierre syndrome is a life-threatening condition characterized by head/neck bacterial infection, local suppurative thrombophlebitis and septic embolic complications in a range of sites of distant organs. No prior study focused on the course and characteristics of ophthalmic complications of Lemierre syndrome. METHODS: We analysed data of 27 patients with ophthalmic complications from a large cohort of 712 cases with Lemierre syndrome reported globally between 2000 and 2017. We focused on initial manifestations, early (in-hospital) course and long-term ophthalmic deficits at the time of hospital discharge or during postdischarge follow-up. The study protocol was registered in the International Prospective Register of Systematic Reviews PROSPERO (CRD42016052572). RESULTS: Nine (33%) patients were women; the median age was 20 (Q1-Q3: 15-33) years. Fusobacterium spp. was involved in 56% of cases. The most prevalent initial manifestations were decreased vision (35%) and periocular oedema (38%), followed by impaired eye movements/nerve palsy (28%) and proptosis (28%). Venous involvement, notably cerebral vein thrombosis (70%) and ophthalmic vein thrombosis (55%), explained the symptomatology in most cases. Septic embolism (7%), orbital abscesses (2%) and carotid stenosis (14%) were also present. Ophthalmic sequelae were reported in 9 (33%) patients, often consisting of blindness or reduced visual acuity, and nerve paralysis/paresis. CONCLUSION: Ophthalmic complications represent a severe manifestation of Lemierre syndrome, often reflecting an underlying cerebral vein thrombosis. Visual acuity loss and long-term severe complications are frequent. We call for an interdisciplinary approach to the management of patients with Lemierre syndrome and the routine involvement of ophthalmologists.