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Carmena de la Asunción, I. Jiménez Pérez de Heredia, A. Belda Mira" "autores" => array:3 [ 0 => array:4 [ "nombre" => "A." "apellidos" => "Carmena de la Asunción" "email" => array:1 [ 0 => "carmena_amp@gva.es" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "I." "apellidos" => "Jiménez Pérez de Heredia" ] 2 => array:2 [ "nombre" => "A." "apellidos" => "Belda Mira" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Servicio de Medicina Interna del Hospital de Sagunto, Valencia, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Infección por virus del Nilo occidental: primer caso autóctono en la Comunidad Valenciana" ] ] "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" => 1478 "Ancho" => 907 "Tamanyo" => 181036 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Maculopapular skin rash on the lower limbs due to West Nile virus.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">West Nile virus (WNV) is a flavivirus transmitted by mosquitoes of the <span class="elsevierStyleItalic">Culex</span> genus that is endemic to tropical and subtropical areas and whose primary host is birds. Mammals, mainly human beings and horses, are accidental hosts that do not transmit the disease. In Europe, it is considered an endemic epidemic zoonosis with 632 human cases reported in 2023.</p><p id="par0010" class="elsevierStylePara elsevierViewall">We present the first human case of West Nile virus infection in the Community of Valencia involving a 72-year-old female with meningoencephalitis, fever, and skin rash (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). The patient presented at the end of July 2023 with symptoms including a headache of six days duration, neck pain, 38.5 ºC fever, maculopapular skin rash on the trunk and lower limbs, and gait instability with non-spinning dizziness and tremor of the upper limbs. The patient lived in Puzol (Valencia) and had not travelled recently. The examination revealed resting tremor of the upper limbs, neck stiffness, dysmetria of the four limbs, positive Romberg test with falling backwards, and unstable gait with no increase in area. CSF showed 231 nucleated cells/μL with 59% mononuclear cells, 10 red blood cells/μL, glucose 51 mg/dl and proteins 1.43 g/L. Gram stain, culture, and Filmarray PCR for meningitis and encephalitis of the CSF were negative and magnetic resonance imaging revealed no significant findings. A PCR of WNV in CSF was requested, which came back negative yet serology (MAC ELISA) for WNV was positive for IgM and IgG; the IgG titre at admission was 18.87 and 105.74 at 8 days, thus confirming seroconversion. The IgM was positive in both tests.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">The neuroinvasive form of WNV infection only occurs in <1% of cases, as the majority of infections in human beings are asymptomatic or present mild symptoms including fever, headache, muscle aches, and eye pain that tend to last 3–10 days.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a> Morbilliform or maculopapular skin rash appears in 25% of patients and characteristically appears on the trunk and upper limbs. It is often associated with a lower risk of meningoencephalitis and death.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> In our patient, rash was mostly present on the trunk and lower limbs and there was indeed neurological involvement. Neuroinvasive disease encompasses three syndromes: meningitis (35–40%), encephalitis (55–60%) that varies in severity from mild confusion to coma and death, and acute flaccid paralysis (5–10%).<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4,5</span></a> Extrapyramidal symptoms are common as are tremor and myoclonus, particularly affecting the upper limbs. In our case, she presented with lymphocytic meningitis and rhombencephalitis with cerebellar syndrome (ataxia, tremor, dysmetria, Romberg+).</p><p id="par0020" class="elsevierStylePara elsevierViewall">In terms of microbiological diagnosis, identification of IgM antibodies in serum or CSF is the cornerstone of diagnosis in most cases (58% sensitivity).<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> PCR of CSF has a 45% sensitivity,<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> which is why the negative result in our case is not considered strange. If a cross-reaction with adenovirus and other flaviviruses (e.g. Dengue, Zika, etc.) is suspected, it is advisable to confirm diagnosis with a plaque reduction neutralization test. On the other hand, if the initial MAC-ELISA test is negative and elevated suspicion remains, it is advisable to repeat the MAC-ELISA serum test after 10 days and the combination of PCR and MAC-ELISA, which increases the combined sensitivity to 94%.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Globally, between 1999 and 2021, WNV infection has caused over 55,000 human cases, 27,000 with neuroinvasive disease, and 2,600 deaths. According to data from the Centers for Disease Control and Prevention (CDC), in 2023 there were 1419 human cases and 923 deaths.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> In Europe, between the start of 2023 and 11 October 2023, 623 human cases and 50 deaths were reported. In 128 regions from EU/EEA countries, the reported cases were locally acquired. The year with the most outbreaks in European Union countries was 2018, with 2,083 human cases reported. The largest most recent outbreak was in 2022 in Camargue in France with 1339 patients and 104 deaths.<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">9,10</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">In Spain, West Nile virus has been known to be in circulation since the 90s via retrospective studies. Animal surveillance (birds, horses, mosquitoes) has been taking place since 2001 and human monitoring since 2007.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">11</span></a> The first autochthonous Spanish human case was published in 2007 in an individual who was in Badajoz in 2004. Over the next two decades, only 6 more sporadic cases have been detected. In 2020, incidence increased in Spain in an unprecedented manner with an outbreak in Andalusia: 77 human cases, of which 97% presented with meningoencephalitis symptoms and 8 of which died.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">11,12</span></a> In the following years the incidence has continued to rise, resulting in a moderate risk of transmission in settings where the virus has been detected in animals and/or where human cases have occurred in the current or previous seasons.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">11</span></a> Regarding the Community of Valencia, the first outbreak in a horse-farming operation was recorded in 2020, but no autochthonous human cases had been recorded to date.</p><p id="par0035" class="elsevierStylePara elsevierViewall">In this case, an epidemiological alert was received regarding two confirmed cases of West Nile virus infection in two horses in the same Health region the month prior to the patient’s consultation. Therefore, we must highlight the importance of epidemiological surveillance and vector control measures given that, despite WNV infection being uncommon in our community, it can result in severe neuroinvasive disease symptoms and should be considered in the differential diagnosis of meningitis and encephalitis, particularly in summer and autumn months.</p></span>" "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" => 1478 "Ancho" => 907 "Tamanyo" => 181036 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Maculopapular skin rash on the lower limbs due to West Nile virus.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:12 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:1 [ "referenciaCompleta" => "Clinical manifestations and diagnosis of West Nile virus infection. 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West Nile virus infection: first autochthonous case in the Community of Valencia
Infección por virus del Nilo occidental: primer caso autóctono en la Comunidad Valenciana
A. Carmena de la Asunción
, I. Jiménez Pérez de Heredia, A. Belda Mira
Corresponding author
Servicio de Medicina Interna del Hospital de Sagunto, Valencia, Spain