Letter to the Editor
Atypical Lemierre’s syndrome due to thrombophlebitis of the anterior jugular vein. A case report
Síndrome de Lemierre atípico por tromboflebitis de la vena yugular anterior. A propósito de un caso
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A case report" ] ] "contieneTextoCompleto" => array:1 [ "es" => true ] "contienePdf" => array:1 [ "es" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figura 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 500 "Ancho" => 1401 "Tamanyo" => 71261 ] ] "descripcion" => array:1 [ "es" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A) Ecografía Doppler de tiroides en la que se aprecia la ocupación y distensión de la vena yugular anterior derecha. 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A case report" "tieneTextoCompleto" => true "saludo" => "Dear Editor," "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "424" "paginaFinal" => "425" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Enrique Rodríguez-Guerrero, Azahara Fernández-Carbonell, María López-Soldado" "autores" => array:3 [ 0 => array:4 [ "nombre" => "Enrique" "apellidos" => "Rodríguez-Guerrero" "email" => array:1 [ 0 => "enriquerg83@gmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "Azahara" "apellidos" => "Fernández-Carbonell" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "María" "apellidos" => "López-Soldado" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] ] "afiliaciones" => array:3 [ 0 => array:3 [ "entidad" => "Servicio de Microbiología, Hospital Universitario Virgen de las Nieves, Granada, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Unidad de Cuidados Intensivos, Hospital Universitario de Jaén, Jaén, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Servicio de Urgencias, Hospital Universitario Virgen de las Nieves, Granada, Spain" "etiqueta" => "c" "identificador" => "aff0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Síndrome de Lemierre atípico por tromboflebitis de la vena yugular anterior. A propósito de un caso" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 500 "Ancho" => 1401 "Tamanyo" => 67960 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Doppler ultrasound of the thyroid showing right anterior jugular vein filling and distension. B) Computed tomography of the neck with intravenous contrast showing total filling of the lumen of the right anterior jugular vein by a hypodense content, compatible with thrombophlebitis.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Lemierre’s syndrome is characterised by the development of internal jugular vein thrombophlebitis and metastatic septic emboli secondary to acute pharyngeal infection.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We report an extremely rare case of a young man with Lemierre’s syndrome due to thrombophlebitis of the right anterior jugular vein without associated oropharyngeal disease.</p><p id="par0015" class="elsevierStylePara elsevierViewall">The patient, a 38-year-old doctor, presented with a 5-day history of progressive fever (from 37.5 °C to 39.5 °C) with odynophagia, fatigue and pain in the right anterior neck region. He went to the emergency department for no improvement despite self-medication with anti-inflammatory drugs, antipyretics and amoxicillin-clavulanic acid (875/125 mg every 8 h) for 3 days. Haemodynamic constants were normal, with slight pallor of the skin and a normal physical examination except for erythema, flushing and pain in the right anterior neck region.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Chest X-ray was normal. Biochemical analysis showed parameters of acute renal failure (creatinine 1.78 mg/dl), proteinuria and microscopic haematuria. Regarding the liver, there was elevation of enzymes GOT (136 U/L), GPT (216 U/L), GGT (210 U/L) and alkaline phosphatase (277 U/L). Acute phase reactants were increased (C-reactive protein 268 mg/L and procalcitonin 4.13 ng/mL). The CBC was normal except for mild leukocytosis with neutrophilia (8,180 neutrophils/μL) and thrombocytopenia (75,000 platelets/μL). Thyroid tests were normal, with positive antithyroglobulin antibodies and positive ANA with nucleolar pattern, without associated systemic disease. Multiple infectious serologies and blood cultures were negative.</p><p id="par0025" class="elsevierStylePara elsevierViewall">After ruling out oropharyngeal disease, the patient was admitted to the Internal Medicine ward for clinical stabilisation and diagnostic study. Empirical treatment was started with 2 g of intravenous amoxicillin-clavulanic acid every 8 h. The abdominal ultrasound was normal and the thyroid Doppler ultrasound showed thrombophlebitis of the right anterior jugular vein (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A), hospitalisation was therefore maintained to continue intravenous antibiotic treatment for 2 weeks, as well as subcutaneous anticoagulation with prophylactic doses of 2,500 IU/24 h bemiparin and 1 g paracetamol intravenously/orally in case of fever. A neck-chest-abdomen-pelvis CT scan with intravenous contrast confirmed thrombophlebitis of the right anterior jugular vein, as well as bilateral pleural effusion and multiple bilateral pulmonary nodules suggestive of septic embolisms (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>B). No abnormalities were observed on head CT or echocardiogram.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">The patient showed improvement in both clinical and laboratory parameters over the next 2 weeks. A contrast-enhanced neck and chest CT scan showed complete resolution of the anterior jugular vein thrombus and reduction of the pulmonary septic embolisms. Home discharge was decided with outpatient treatment of 875/125 mg amoxicillin-clavulanic acid every 8 h for 2 more weeks, as well as antiplatelet therapy with ASA (100 mg/24 h). Patient fully recovered with no significant sequelae after 6 months of regular monitoring. Anti-platelet therapy was withdrawn.</p><p id="par0035" class="elsevierStylePara elsevierViewall">Lemierre’s syndrome is characterised by thrombosis of the internal jugular vein, although atypical variants,<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> such as anterior jugular vein thrombophlebitis, are rare and represent an additional diagnostic difficulty. This syndrome is more common in young males, with an incidence of 3.6 cases per million per year and a mortality of 4.1%.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Risk factors include persistent fever, history of head and neck infections, diabetes, dental caries, among others.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Although the most commonly associated microorganism is <span class="elsevierStyleItalic">Fusobacterium necrophorum</span>, it is not always isolated microbiologically. In our case, the absence of oropharyngeal involvement made it difficult to identify the infectious origin, as blood cultures were negative, which occurs in 7.4% of cases.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">To date, only two other cases of thrombophlebitis of the anterior jugular vein have been reported. Both required surgical intervention, in one case due to the presence of a dental abscess<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> and in the other case due to extensive neck cellulitis secondary to acute bacterial tonsillitis.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">In our case, the action taken in the emergency department made it possible to obtain an early clinical radiological diagnosis: an effective intravenous antibiotic therapy with amoxicillin-clavulanic acid was administered in time. However, there is controversy about the indication for anticoagulation in Lemierre’s syndrome, as it has not been shown to improve vascular recanalization or mortality rate compared to non-anticoagulated patients.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Therefore, in our case, the indication was chosen for prophylactic rather than therapeutic purposes.</p><p id="par0055" class="elsevierStylePara elsevierViewall">In conclusion, due to the severity and high mortality risk of Lemierre’s syndrome, it is very important that this syndrome is considered in the emergency department; not only in cases of high fever with signs of sepsis and oropharyngeal focus, but also in patients with pain in the anterior neck region without oropharyngeal disease.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Ethical considerations</span><p id="par0060" class="elsevierStylePara elsevierViewall">Informed consent was obtained from the patient for publication, as required by current legislation.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Funding</span><p id="par0065" class="elsevierStylePara elsevierViewall">There are no sources of funding.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflict of interest</span><p id="par0070" class="elsevierStylePara elsevierViewall">There are no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Ethical considerations" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Funding" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Conflict of interest" ] 3 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "multimedia" => array:1 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 500 "Ancho" => 1401 "Tamanyo" => 67960 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Doppler ultrasound of the thyroid showing right anterior jugular vein filling and distension. B) Computed tomography of the neck with intravenous contrast showing total filling of the lumen of the right anterior jugular vein by a hypodense content, compatible with thrombophlebitis.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Lemierre’s syndrome: a forgotten and re-emerging infection" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "W.S. Lee" 1 => "S.S. Jean" 2 => "F.L. Chen" 3 => "S.M. Hsieh" 4 => "P.R. 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