Síndrome de Lemierre / Lemierre's Syndrome

Paciente masculino de 32 años de edad sin comorbilidades, acude a servicio de urgencias con cuadro clínico caracterizado por dolor en región cervical anterior, tos con expectoración de color marrón de mal olor y dolor torácico. Refiere como antecedente, diagnóstico de faringoamigdalitis hace un mes, para el cual recibió tratamiento antibiótico intramuscular que no especifica. Al examen físico presenta masa palpable a nivel de región cervical anterior, dolor a la palpación torácica en séptimo espacio intercostal izquierdo. A la auscultación frote pericárdico, murmullo vesicular bilateral disminuido y frote pleural. Los estudios de laboratorio demostraron leucocitosis (17230 k/ul), neutrofilia (79,25%), anemia leve (Hb 11 g/dl, Hcto 35,5%). La tomografía computarizada contrastada de tórax, demostró presencia de edema y líquido en los tejidos blandos superficiales anteriores infrahioideos del cuello por encima de la horquilla esternal, y, en la fase contrastada trombosis de la vena yugular interna izquierda

Acometimento pulmonar e articular secundário à síndrome de Lemierre: relato de caso e revisão de literatura / Pulmonary and joint involvement secondary to Lemierre syndrome: case report and literature review

A síndrome de Lemierre caracteriza-se por uma rara entidade que gera tromboflebite da veia jugular interna e embolismo séptico em história da infecção recente da orofaringe, além de sinais radiológicos e isolamento de patógenos anaeróbicos, principalmente Fusobacterium necrophorum. Relatamos o caso de uma paciente do sexo feminino, 13 anos de idade, com histórico de carcinoma de nasofaringe associado ao vírus Epstein-Barr (estadiamento T4N2M0), submetida a procedimentos cirúrgicos e quimiorradioterapia. Iniciou com queixa de mialgia intensa, diplopia, lesões infectadas em membros e choque séptico. Por meio de exames de ultrassonografia cervical com Doppler colorido e tomografia computadorizada de pescoço com contraste endovenoso, foram identificados trombos intraluminais na veia jugular interna, além de trombos sépticos pulmonares, por meio da tomografia computadorizada de tórax. Posteriormente, ainda evoluiu com artrite piogênica coxofemoral esquerda. Foi isolada, por hemocultura, a bactéria Klebsiella pneumoniae Carpemenase, e o tratamento se deu pela associação entre vancomicina, amicacina, meropenem, metronidazol e anfotericina B. Conclui-se que, após o diagnóstico de SL e, embora com múltiplas complicações e diagnóstico tardio, a paciente encontra-se bem e assintomática, além do relato comprovar a dificuldade diagnóstica e de seu tratamento

Lemierre's syndrome is a rare condition that leads to thrombophlebitis of the internal jugular vein and septic embolism following recent oropharyngeal infection, being characterized by radiological signs and isolation of anaerobic pathogens, especially Fusobacterium necrophorum. We report the case of a 13-year-old female patient with history of nasopharyngeal carcinoma associated with Epstein-Barr virus (T4N2M0 staging), who underwent surgical procedures and chemoradiotherapy. Her initial complaint was severe myalgia, diplopia, infected limb injuries, and septic shock. Cervical color Doppler ultrasound and computed tomography scan of the neck with intravenous contrast showed intraluminal thrombi in the internal jugular vein, and chest computed tomography showed pulmonary septic thrombi. Subsequently, she progressed with left coxofemoral pyogenic arthritis. The bacterium Klebsiella pneumoniae Carpemenase was isolated in blood culture, and the patient was treated with the association of vancomycin, amikacin, meropenem, metronidazole, and amphotericin B. It is concluded that, despite the multiple complications and late diagnosis, the patient is well and asymptomatic after the diagnosis of Lemierre's syndrome; in addition, the report proves the difficulty of diagnosis and treatme

Síndrome de Lemierre. Caso y revisión de la literatura / Lemierre's syndrome. Clinical case and literature review / Síndrome de Lemierre. Caso clínico e revisão da literatura

Resumen: El síndrome de Lemierre constituye una entidad poco frecuente y potencialmente grave que puede complicar una infección orofaríngea. Incluye una tromboflebitis de la vena yugular interna y embolias sépticas a distancia. Presentamos el caso clínico de un hombre joven que se presentó, luego de una infección respiratoria alta, con una trombosis de vena yugular interna, seno sigmoideo y transverso izquierdos, asociando además tromboembolismo pulmonar séptico y empiema pleural. Mostró buena evolución con antibioticoterapia empírica, drenaje pleural y anticoagulación. Realizamos una revisión de la literatura sobre este tema enfatizando los aspectos clínicos, epidemiológicos y microbiológicos.

Summary: Lemierre's syndrome constitutes a rare and potentially serious entity, that may complicate oropharyngeal infections. This condition includes thrombophlebitis of the internal jugular vein and distant septic embolisms. The study presents the clinical case of a young man who consulted after an upper respiratory infection, with internal jugular vein thrombosis, left sigmoid and transverse seins, also associating septic pulmonary embolism and pleural empyema. The patient had a positive evolution after empirical antibiotic, pleural drainage and anticoagulants. We conducted literature review on this condition, focusing on clinical, epidemiological and microbiological aspects.

Resumo: A síndrome de Lemierre é uma doença rara e potencialmente grave que pode complicar uma infecção orofaríngea. Inclui uma tromboflebite da veia jugular interna e embolias sépticas a distância. Descrevemos o caso clínico de um homem jovem que depois de uma infecção respiratória alta apresentou trombose de veia jugular interna, seio sigmoide e transverso esquerdos, associado com tromboembolismo pulmonar séptico e empiema pleural. Teve boa evolução com antibioticoterapia empírica, drenagem pleural e anticoagulação. Realizamos uma revisão da literatura sobre este tema enfatizando os aspectos clínicos, epidemiológicos e microbiológicos.

Síndrome de Lemierre simulando síndrome paraneoplásico: reporte de un caso y revisión de la literatura / Lemierre syndrome simulating signs and symptoms of paraneoplastic syndrome: case report and literature review

El síndrome de Lemierre es una enfermedad rara, ocasionada por la complicación grave de una infección orofaringea que se manifiesta con una tromboflebitis séptica en la yugular interna y una embolización séptica a distancia. En principio, el síndrome cursa con fiebre, dolor en el área del ángulo de la mandíbula, inflamación de la región periamigdalina y un crecimiento unilateral del cuello luego de una infección en las estructuras de la cabeza y el cuello. En el artículo se presenta el caso de una mujer de 45 años quien consultó por una complicación infecciosa posterior a la realización de una exodoncia. El diagnóstico clínico se realizó con base en los signos y síntomas que indicaban tromboflebitis séptica del seno cavernoso. Se realiza antibióticoterapia con resultados positivos en la paciente y, como resultado, una mejoría total del estado de salud. Se presenta este caso por lo infrecuente de la entidad.

Lemierre syndrome is a disease caused by the serious complication of an oropharyngeal infection that manifests with septic thrombophlebitis in internal jugular and remote septic embolization. It presents with fever, pain in the area of the jaw angle, inflammation of the peritonsillar region and unilateral neck growth after an infection in the head and neck structures. We present the case of a 45-year-old woman who consulted due to an infectious complication after carrying out the extraction. The clinical diagnosis was made based on the signs and symptoms that indicated septic thrombophlebitis of the cavernous sinus. Antibiotic therapy is performed with positive results in the patient and total improvement of the state of health.

Infecciones profundas del cuello / Deep neck infections

Deep neck infections (DNIs) are special entities among infectious diseases for their versatility and potential for severe complications. Complex head and neck anatomy often makes early recognition of DNIs challenging, and a high index of suspicion is necessary to avoid any delay in treatment. The diagnosis is made by clinical history, physical examination findings and imaging studies. The treatment consists in securing the airway, intravenous antibiotics and surgical drainage, when needed. To make decisions the surgeon must understand the anatomy of the region, the etiology of infection, appropriate diagnostic tools, and medical and surgical management. This article provides a review of these pertinent topics. (AU)

A Case of Lemierre's Syndrome, Misdiagnosed as a Simple Deep Neck Infection on Initial Ultrasonography Followed by an Abscess Aspiration Trial / 대한두경부종양학회지

Lemierre's syndrome is rare disease characterized by anaerobic sepsis, internal jugular vein thrombosis, septic emboli that resulted from head and neck infection. Lemierre's syndrome has significant morbidity, so immediate, accurate diagnosis and treatment is needed. It is necessary to perform contrast-enhanced computed tomography (CT) for diagnosis. Systemic antibiotics is recommended, and surgical interventions, anticoagulation may beis considered for treatment. We report misdiagnosed case as a simple deep neck infection on initial ultrasonography with simultaneous abscess aspiration but finally diagnosed and treated internal jugular vein thrombophlebitis (Lemierre's syndrome) on CT scan.We report a case of a 45-year-old patient, who was diagnosed with a simple deep neck infection and treated with simultaneous abscess aspiration, but finally diagnosed and treated internal jugular vein thrombophlebitis (Lemierre's syndrome) on CT scan.

Enfermedad de Lemierre, a propósito de un caso / Lemierre's Disease, Case Report

La enfermedad de Lemierre, descrita por primera vez en el año 1936 por el médico Francés André Lemierre, es una complicación inusual de una infección orofaríngea, que progresa con tromboflebitis séptica secundaria e infecciones embólicas frecuentes. Es producida por microorganismos anaerobios, siendo el Fusobacterium necrophorum el germen aislado con mayor frecuencia. Para su diagnóstico, además del estudio microbiológico, se emplean los estudios por imágenes como la ecografía Doppler y la tomografía computada (TC). La antibióticoterapia precoz y prolongada, a dosis altas, ha mejorado considerablemente el pronóstico; no obstante, en ocasiones, se hace necesario recurrir a la escisión quirúrgica de las venas yugulares. En la actualidad con la terapia antimicrobiana, casos como éste son cada vez más raros, incluso a veces olvidados, pero dada su gravedad deben sospecharse ante cuadros faríngeos de evolución tórpida

Síndrome de Lemierre: relato de caso / Lemierre syndrome: case report

síndrome de Lemierre caracteriza-se pela tromboflebite séptica da veia jugular interna, após uma orofaringite, com embolização séptica para o pulmão ou outros órgãos. Neste relato de caso, apresentamos uma paciente feminina, 37 anos de idade, com história de edema e dor em hemiface direita há três dias, associada a fadiga e dispneia progressiva há um dia. História de extração dentária do elemento 48 há três dias. No exame físico admissional, apresentava-se taquipneica, saturando 60% (em ar ambiente), com edema em ângulo da mandíbula direita, redução difusa do murmúrio vesicular e panturrilhas sem empastamento. Angiotomografia de tórax e exames laboratoriais foram compatíveis com quadro de embolia séptica, e tomografia computadorizada da cervical corroborou o diagnóstico de tromboflebite séptica da veia jugular interna. Foi tratada com antibióticos e sintomáticos. A síndrome de Lemierre afeta mais homens jovens e tem embolização para o pulmão em até 97% dos casos. Extrações dentárias raramente podem ser a etiologia dessa síndrome. A tomografia computadorizada é o método de imagem mais utilizado no diagnóstico, e o tratamento é, essencialmente, com antibióticos; portanto, a abordagem cirúrgica é raramente necessária

Lemierre syndrome is characterized by septic thrombophlebitis of the internal jugular vein, after an oropharyngeal infection, with septic embolization to the lungs or other organs. This case report describes a 37-year-old female patient who presented with edema and pain in the right hemiface with onset 3 days previously and progressive fatigue and dyspnea since the previous day. She had had tooth 48 extracted 3 days previously. Physical examination at admission found tachypnea, with 60% saturation (in room air), edema at the angle of the right mandible, diffuse reduction of vesicular murmur, and calves free from clubbing. Angiotomography of the chest and laboratory tests were compatible with septic emboli, and cervical computed tomography confirmed a diagnosis of septic thrombophlebitis of the internal jugular vein. She was managed with antibiotics and given treatment for her symptoms. Lemierre syndrome most often occurs in young men and there is embolization to the lungs in up to 97% of cases. Rarely, the etiology of this syndrome may be tooth extraction. Computed tomography is the imaging method most often used for diagnosis and treatment is basically antibiotic. Surgery is thus rarely necessary

Tromboembolismo pulmonar séptico secundário à síndrome de Lemierre / Septic pulmonary thromboembolism due to Lemierre syndrome

Descrita pela primeira vez em 1900 por Coumont e Cade, a tromboflebite séptica da veia jugular interna (síndrome de Lemierre) é uma condição rara. Acomete indivíduos jovens e possui elevada morbimortalidade. Relatamos o caso de uma paciente atendida inicialmente como portadora de amigdalite bacteriana e que retornou com piora do quadro, associado à trombose da veia jugular interna, evoluindo, na internação, com embolia séptica pulmonar. Além de relatar o caso, fazemos breve revisão da literatura e chamamos a atenção sobre este importante assunto.(AU)

First described in 1900 by Coumont and Cade, septic thrombophlebitis of the internal jugular vein (Lemierre's syndrome) is relatively rare. It affects young patients and has high morbidity and mortality. We describe the case of a woman first diagnosed with a bacterial tonsillitis, who returned to the hospital with worsening of the condition, associated with internal jugular vein thrombophlebitis, that developed to pulmonary embolism during her hospitalization. We reported the case, and made a brief review of the literature, highlighting the details of this important condition.(AU)

Lemierre Syndrome in Adolescent with Active Ulcerative Colitis / 대한소아소화기영양학회지

Inflammatory bowel disease (IBD) is a well-recognized risk factor for thrombotic events in adults but data on children are scarce. In the great majority of adult patients, thrombotic events are usually deep vein thrombosis and pulmonary embolism. Other sites such as jugular veins are extremely rare. We present a case of Lemierre syndrome in an adolescent girl with active ulcerative colitis and discuss possible risk factors. This is the first reported case of severe Lemierre syndrome with thrombus extension to cranial veins in a patient with ulcerative colitis. Early recognition of Lemierre syndrome in patients who present with rapidly worsening symptoms of neck pain, fever and signs of pharyngitis is imperative because it increases a chance of favorable prognosis. It is important for pediatricians treating IBD patients not to underestimate possible thrombotic events in children with IBD. Recognition of additional risk factors is crucial for prompt diagnosis and adequate treatment.

An atypical case of Lemierre syndrome following oropharyngeal infection / 고신대학교의과대학학술지

Lemierre syndrome is characterized by anaerobic bacterial infection in the head and neck and clinical or radiological evidence of internal jugular vein thrombophlebitis. The most common pathogens are Fusobacterium species, particularly Fusobacterium necrophorum. Septic emboli resulting from infected thrombophlebitis of the internal jugular vein leads to metastatic infections involving lung, liver, kidney, bone and central nervous system. The accurate diagnosis and treatment is important because it may be associated with a high mortality rate if untreated. We present a case of 28-year-old man with an atypical history for the diagnosis of Lemierre syndrome, which showed no definite evidence of internal jugular thrombophlebitis.

El síndrome de Lemierre: una patología resurgente: a propósito de una observación pediátrica / Lemierres syndrome: a resurgent pathology about a pediatric observation

El síndrome de Lemierre (SL) es una emergencia diagnóstica y requiere una terapéutica urgente. Se define como una tromboflebitis séptica de la vena yugular interna (VYI) secundaria a una infección orofaríngea. En la mayoría de los casos el germen implicado es el Fusobacterium necrophorum (FN). Afecta tanto al adulto joven como al adolescente pero muy poco al niño. Esta rara afección debe beneficiarse de un diagnóstico precoz. En el caso contrario, se acompaña con un riesgo elevado de mortalidad. Les exponemos en este trabajo, el caso de una niña de 6 años con un SL tras una otitis media aguda asociada a manifestaciones cutáneas de la septicemia. La paciente mejoró bajo una combinación de antibióticos (8 semanas) y anticoagulantes (3 meses).

Lemierre’s syndrome is a rare, acute and severe entity It is characterized by thrombosis of the internal jugular vein and metastatic infections especially lung localization. The main pathogen is Fusobacterium necrophorum. This pathology concerns rarely the child. Early diagnosis is crucial otherwise the mortality will increase. We report of Lemierre’s syndrome in a girl of 6 years old with favourable outcome. Antibiotherapy targeting anaerobes has been rapidly Introduced and maintained 8 weeks. The anticoagulation has been maintained 3 months.

A Case of Lemierre Syndrome Manifests with Persistent Fever and Neck Stiffness Following Acute Oropharyngeal Infection

Lemierre syndrome is a rare disease involving multiple organs affected by septic emboli following oropharyngeal infection. After the introduction of penicillin in the 1940s, it became a "forgotten" disease. However, due to the development of diagnostic image modalities including neck computed tomography (CT) scan, the number of published reports of Lemierre syndrome and diagnosis has been increasing since the 1990s. In this report, we describe a case of Lemierre syndrome, following oropharyngeal infection in a 16-year-old patient, who manifested with persistent fever and neck stiffness. Neck ultrasonography confirmed thrombus formation in the right internal jugular vein without definite evidence of septic emboli to other organs. After the three-week-long antibiotics therapy was completed, the thrombus in the right internal jugular vein finally disappeared.

Ultrasound-guided superficial cervical plexus block for carotid endarterectomy in a patient with Lemierre syndrome: A case report

A 70-year-old woman with an infectious thrombus in her left internal jugular vein (IJV) underwent carotid endarterectomy for stenosis and a highly movable plaque in her right carotid artery. She had been treated with antibiotics for four weeks before surgery due to Lemierre syndrome, a rare septic thrombophlebitis in the IJV secondary to an oropharyngeal infection. The right IJV was in a two-fold dilated state due to compensation for a thrombotic left IJV. Accordingly, superficial cervical plexus block was performed under ultrasound guidance to ensure safety and accuracy. During surgery, the alertness was maintained and the patient did not complain of pain in the absence of additional analgesics. No block-related complications were encountered. The authors report for the first time their regional anesthetic experiences in a patient with Lemierre syndrome.

Síndrome de Lemierre: relato de caso / Lemierre syndrome: a case report

A tromboflebite supurativa da veia jugular interna ou síndrome de Lemierre foi descrita pela primeira vez em 1900. O evento inicial mais frequente é a infecção de orofaringe associada à trombose da veia jugular interna. Embora uma entidade rara, a síndrome de Lemierre continua a ser uma doença de morbidade e mortalidade consideráveis devido à sua progressão e atrasos de diagnóstico.

Suppurative thrombophlebitis of the internal jugular vein, or Lemierre syndrome, was described for the first time in 1900. The most common initial event is an infection of the oropharynx associated with thrombosis of the internal jugular vein. While it is a rare entity, Lemierre syndrome remains a disease that causes considerable morbidity and mortality, due to its progression and to delays in diagnosis.

Síndrome de Lemierre: una entidad clínica casi en el olvido: reporte de tres casos y revisión de la literatura / Lemierre´s syndrome: an almost forgotten clinical entity: three case reports and literature review

Lemierre’s syndrome is a rare disease that affects young adults and is mainly caused by Fusobacterium necrophorum and occasionally by other anaerobic bacteria of the species. This syndrome is characterized by a throat infection complicated with septic thrombophlebitis of the internal jugular vein and septic emboli mainly to the lungs. In the pre-antibiotic era its evolution was often fatal. Since the 1960’s this syndrome has rarely been reported given the extensive use of penicillin en pharyngeal infections. Currently the incidence of Lemierre’s syndrome is about one in a million. Currently, since imaging has a key role in the early diagnosis of this syndrome, the radiologist should be aware of and recognize its manifestations. Three cases of Lemierre’s syndrome and a literature review are presented.

El síndrome de Lemierre es una enfermedad rara que afecta a adultos jóvenes y es causada principalmente por Fusobacterium necrophorum y ocasionalmente por otras bacterias anaerobias de la especie. Este síndrome se caracteriza por una infección faríngea complicada con tromboflebitis séptica de la vena yugular interna y embolias sépticas principalmente a los pulmones. En la era pre-antibióticos su evolución era frecuentemente de curso fatal. Desde los años 60 este síndrome ha sido rara vez reportado dado el extensivo uso de penicilina en infecciones faríngeas. Actualmente la incidencia del síndrome de Lemierre es de aproximadamente uno en un millón. Dado que hoy en día las imágenes tienen un rol fundamental en el diagnóstico temprano de este síndrome el radiólogo debe estar al tanto y reconocer sus manifestaciones. Se presentan tres casos de Síndrome de Lemierre y una revisión de la literaratura.