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"apellidos" => "de Vicente" "email" => array:1 [ 0 => "jvicente@uniovi.es" ] "referencia" => array:4 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">d</span>" "identificador" => "aff0020" ] 2 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">e</span>" "identificador" => "aff0025" ] 3 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] ] "afiliaciones" => array:6 [ 0 => array:3 [ "entidad" => "Department of Oral and Maxillofacial Surgery, Hospital Universitario Central de Asturias, Oviedo, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Department of Microbiology, Hospital Universitario Central de Asturias, Oviedo, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Department Head Department of Oral and Maxillofacial Surgery, Hospital Universitario Central de Asturias, Oviedo, Spain" "etiqueta" => "c" "identificador" => "aff0015" ] 3 => array:3 [ "entidad" => "School of Medicine and Health Sciences, University of Oviedo, Asturias, Spain" "etiqueta" => "d" "identificador" => "aff0020" ] 4 => array:3 [ "entidad" => "Instituto Universitario de Oncología del Principado de Asturias (IUOPA), Spain" "etiqueta" => "e" "identificador" => "aff0025" ] 5 => array:3 [ "entidad" => "Instituto de Investigación Sanitaria del Principado de Asturias (ISPA), Oviedo, Spain" "etiqueta" => "f" "identificador" => "aff0030" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Tularemia: diagnóstico de un caso oculoglandular inesperado en un área no endémica mediante PCR universal" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 805 "Ancho" => 805 "Tamanyo" => 84127 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Cervical mass observed in physical examination one month after conjunctivitis.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Tularemia is a zoonotic disease caused by <span class="elsevierStyleItalic">Francisella tularensis</span>, a Gram-negative facultative intracellular coccobacillus<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> with four recognized subspecies<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a>: <span class="elsevierStyleItalic">tularensis</span> (type A), <span class="elsevierStyleItalic">holarctica</span> (type B), <span class="elsevierStyleItalic">mediasiatica</span> and <span class="elsevierStyleItalic">novicida</span>. Type A is found in North America, while type B is located, but not exclusively, in the northern hemisphere.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> In Spain, it was an uncommon disease until 1997, when the first tularemia outbreak occurred in Castilla y León.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a> Until now, all cases reported in Spain were caused by <span class="elsevierStyleItalic">F. tularensis</span> subsp. <span class="elsevierStyleItalic">holarctica.</span> Clinical manifestations of tularemia fall into two main forms: ulceroglandular (>90% of cases in Europe)<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> and typhoidal. However, there are three more clinical forms: oculoglandular, oropharyngeal/gastrointestinal and pneumonic.</p><p id="par0010" class="elsevierStylePara elsevierViewall">We have previously published the first case of ulceroglandular tularemia in a non-endemic area (Asturias, Spain).<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> Here, we present the first reported case of oculoglandular tularemia occurred in the same region which worried us.</p><p id="par0015" class="elsevierStylePara elsevierViewall">An 88-year-old male presented to the emergency department of our hospital in April 2017 for diagnosis and management of pain in his right eye and the presence of conjunctival discharge. He did not have other symptoms, history of trauma, drug intake, or any recently local or systemic infection. His laboratory workup only showed a high value of C-reactive protein (5.5<span class="elsevierStyleHsp" style=""></span>mg/dL) and his medical and surgical histories were noncontributory. A diagnostic of viral conjunctivitis was done and he was treated with lubricant and anti-inflammatory drops.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Two months later, he was brought to the Department of Oral and Maxillofacial Surgery with a painful cervical mass (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>), weight loss and discomfort. He did not declared a recent travel, contact to ill people or animals although he lives in a rural area. Clinical examination revealed the presence of a right neck mass measuring 30<span class="elsevierStyleHsp" style=""></span>mm<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>20<span class="elsevierStyleHsp" style=""></span>mm, with gummy consistency, painful on palpation and without other appreciable alterations.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">A CT scan was performed and three neck lymph nodes together with an intraparotid lymph node were seen as pathological, showing necrotic areas.</p><p id="par0030" class="elsevierStylePara elsevierViewall">A universal PCR was performed in fine-needle aspiration biopsy (FNAB), based on amplification of the gene coding for 16S rRNA and subsequent sequencing (Bigdye® Terminator, Thermo Fisher Scientific). This evidenced the presence of <span class="elsevierStyleItalic">F. tularensis</span> subsp. <span class="elsevierStyleItalic">holarctica</span>. A second sample of FNAB was sent 10 days later resulting also positive.</p><p id="par0035" class="elsevierStylePara elsevierViewall">In addition, serum samples were sent to the Spanish National Center for Microbiology (Madrid, Spain) in order to study the presence of antibodies against <span class="elsevierStyleItalic">F. tularensis</span> by microagglutination resulting positive with a titer of 1/1024.</p><p id="par0040" class="elsevierStylePara elsevierViewall">Ziehl-Neelsen stain, culture and PCR for mycobacteria were negative. Lymphadenopathy-causing viruses (CMV, EB, HHV-6, HHV-7, HHV-8, Adenovirus, Picornavirus, Enterovirus, Mumps, LCMV) and <span class="elsevierStyleItalic">Toxoplasma gondii</span> were undetectable by PCR. Serological tests (ELISA) against <span class="elsevierStyleItalic">Coxiella burnetii</span> (IgG), <span class="elsevierStyleItalic">Rickettsia conorii</span> (IgG) and <span class="elsevierStyleItalic">Borrelia burgdorferi</span> (IgG/IgM) were negative.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Once the diagnosis was confirmed, the patient was treated with a 14-day course of intravenous streptomycin at a dose of 10<span class="elsevierStyleHsp" style=""></span>mg/kg/12<span class="elsevierStyleHsp" style=""></span>h, with favorable evolution. No surgical excision of the neck mass was needed.</p><p id="par0050" class="elsevierStylePara elsevierViewall">Oculoglandular form of tularemia is very infrequent in our environment. In Spain some studies show an incidence of this form around 4%<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a> but this microorganism should be considered in a patient with Parinaud's syndrome even in non-endemic areas.</p><p id="par0055" class="elsevierStylePara elsevierViewall">The gold standard for the diagnosis of tularemia is the isolation of the causative agent by culture, however, this is difficult (it requires a medium with cysteine) and hazardous for the laboratory staff (Biosafety Level 2 precautions). Therefore diagnosis is based mainly on serology and results became positive between 10 and 14 days after onset of the disease.<a class="elsevierStyleCrossRefs" href="#bib0080"><span class="elsevierStyleSup">6,7</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">Genome amplification by polymerase chain reaction (PCR) is more sensitive than culture and provides rapid, sensitive and specific diagnosis of tularemia.<a class="elsevierStyleCrossRefs" href="#bib0090"><span class="elsevierStyleSup">8–10</span></a> There are specific targets of <span class="elsevierStyleItalic">F. tularensis</span> genes (e.g. <span class="elsevierStyleItalic">fopA</span>, <span class="elsevierStyleItalic">tul4</span>, <span class="elsevierStyleItalic">ISFtu2</span> or RD1 protein-encoding gen) but when there is no suspicion of a specific etiological agent, it is useful to perform a universal PCR. In this case, if we had not performed the 16S rRNA PCR, the patient wouldn’t have been correctly diagnosed.</p><p id="par0065" class="elsevierStylePara elsevierViewall">The present case shows the importance of molecular techniques that amplify panbacterial genes, especially useful for diagnosis of rare infections with great difficulty of isolation of the etiological agent like <span class="elsevierStyleItalic">Bartonella henselae</span> (also causing Parinaud's syndrome), <span class="elsevierStyleItalic">Tropheryma whipplei</span>, <span class="elsevierStyleItalic">Borrelia</span> spp. or <span class="elsevierStyleItalic">Ehrlichia</span> spp. Also in those cases without bacterial growth due to antibiotic treatment.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Financial disclosure and conflict of interests</span><p id="par0070" class="elsevierStylePara elsevierViewall">The authors declare that they have not received funding to carry out this study and have no conflict of interests.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:2 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Financial disclosure and conflict of interests" ] 1 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "multimedia" => array:1 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 805 "Ancho" => 805 "Tamanyo" => 84127 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Cervical mass observed in physical examination one month after conjunctivitis.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0055" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Tularemia" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "J. 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Year/Month | Html | Total | |
---|---|---|---|
2024 October | 16 | 10 | 26 |
2024 September | 16 | 12 | 28 |
2024 August | 11 | 7 | 18 |
2024 July | 13 | 8 | 21 |
2024 June | 16 | 6 | 22 |
2024 May | 18 | 4 | 22 |
2024 April | 27 | 15 | 42 |
2024 March | 45 | 28 | 73 |
2024 February | 41 | 10 | 51 |
2024 January | 44 | 10 | 54 |
2023 December | 55 | 19 | 74 |
2023 November | 40 | 33 | 73 |
2023 October | 88 | 33 | 121 |
2023 September | 40 | 22 | 62 |
2023 August | 29 | 6 | 35 |
2023 July | 67 | 113 | 180 |
2023 June | 69 | 33 | 102 |
2023 May | 55 | 12 | 67 |
2023 April | 41 | 1 | 42 |
2023 March | 30 | 8 | 38 |
2023 February | 44 | 5 | 49 |
2023 January | 34 | 7 | 41 |
2022 December | 33 | 14 | 47 |
2022 November | 49 | 22 | 71 |
2022 October | 31 | 26 | 57 |
2022 September | 41 | 16 | 57 |
2022 August | 40 | 19 | 59 |
2022 July | 30 | 16 | 46 |
2022 June | 45 | 18 | 63 |
2022 May | 36 | 15 | 51 |
2022 April | 34 | 45 | 79 |
2022 March | 36 | 23 | 59 |
2022 February | 41 | 11 | 52 |
2022 January | 40 | 12 | 52 |
2021 December | 25 | 23 | 48 |
2021 November | 46 | 20 | 66 |
2021 October | 33 | 10 | 43 |
2021 September | 29 | 13 | 42 |
2021 August | 28 | 10 | 38 |
2021 July | 30 | 6 | 36 |
2021 June | 21 | 9 | 30 |
2021 May | 40 | 11 | 51 |
2021 April | 72 | 10 | 82 |
2021 March | 34 | 16 | 50 |
2021 February | 34 | 8 | 42 |
2021 January | 13 | 10 | 23 |
2020 December | 18 | 7 | 25 |
2020 November | 20 | 14 | 34 |
2020 October | 14 | 8 | 22 |
2020 September | 28 | 10 | 38 |
2020 August | 19 | 11 | 30 |
2020 July | 29 | 16 | 45 |
2020 June | 20 | 7 | 27 |
2020 May | 13 | 9 | 22 |
2020 March | 0 | 3 | 3 |
2020 February | 0 | 5 | 5 |
2020 January | 6 | 5 | 11 |
2019 December | 15 | 32 | 47 |
2019 November | 105 | 44 | 149 |