Letter to the Editor
Acute Meningitis in Behcet's Disease
Meningitis aguda en la enfermedad de Behçet
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"Andrade-Rodado" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 1 => array:4 [ "nombre" => "Robert" "apellidos" => "Hurtado-García" "email" => array:2 [ 0 => "robelx2@hotmail.com" 1 => "robelx2@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" ] ] ] 2 => array:3 [ "nombre" => "Antonio" "apellidos" => "Álvarez Cienfuegos" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "Santiago" "apellidos" => "Mola Caballero" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] 4 => array:3 [ "nombre" => "Juan Carlos" "apellidos" => "Barreras Mateos" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">d</span>" "identificador" => "aff0020" ] ] ] 5 => array:3 [ "nombre" => "Juan" "apellidos" => "Custardoy Olavarrieta" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:4 [ 0 => array:3 [ "entidad" => "Servicio de Medicina Interna, Hospital Vega Baja, Orihuela, Alicante, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Reumatología, Hospital Vega Baja, Orihuela, Alicante, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Servicio de Neurología, Hospital Vega Baja, Orihuela, Alicante, Spain" "etiqueta" => "c" "identificador" => "aff0015" ] 3 => array:3 [ "entidad" => "Servicio de Radiología, Hospital Vega Baja, Orihuela, Alicante, Spain" "etiqueta" => "d" "identificador" => "aff0020" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Meningitis aguda en la enfermedad de Behçet" ] ] "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" => 903 "Ancho" => 900 "Tamanyo" => 83819 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Brain MRI.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Behçet's disease (BD) is a chronic relapsing occlusive vasculitis of unknown etiology, characterized by the presence of oral and genital ulcers and intraocular inflammation, with less common cutaneous, joint, vascular, digestive and neurological involvement.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">The presence of neurological symptoms in the context of BD constitutes what is called neuroBehçet's, characterized in most cases by the presence of aseptic meningitis, cerebellar signs, intracranial hypertension, and pyramidal alterations.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> We report the case of a 34 year old male with a history of BD, who presented an altered consciousness and meningeal signs, diagnosed with neuroBehçet and showing a favorable clinical response to treatment with triple immunomodulatory therapy.</p><p id="par0015" class="elsevierStylePara elsevierViewall">The patient is a Spanish man of 34 years with BD, in whom the diagnosis was established by the presence of oral and genital ulcers and repeated episodes of anterior uveitis three years prior, treated with prednisone at a dose of 5<span class="elsevierStyleHsp" style=""></span>mg/day and cyclosporin A. He came to the emergency department 24<span class="elsevierStyleHsp" style=""></span>h before due to a self-limited episode of loss of consciousness and subsequent headache and vomiting. The general physical examination and vital signs were normal except for the presence of nuchal rigidity upon neurological examination. There were no signs of disease activity at the time of the initial evaluation.</p><p id="par0020" class="elsevierStylePara elsevierViewall">He was put in reverse isolation for suspected intracranial meningeal syndrome secondary to infection due to immunosuppression. During observation in the emergency department a blood count was performed which showed 4130 WBC E9/l and no left shift, hemoglobin 11.5<span class="elsevierStyleHsp" style=""></span>mg/dl, hematocrit 36%, ESR 12<span class="elsevierStyleHsp" style=""></span>mm/h, lactate dehydrogenase 97<span class="elsevierStyleHsp" style=""></span>U/L, total protein 6.2<span class="elsevierStyleHsp" style=""></span>g/l albumin 3<span class="elsevierStyleHsp" style=""></span>g/l, and a computed tomography that revealed no significant structural alterations.</p><p id="par0025" class="elsevierStylePara elsevierViewall">In accordance with the headache and meningeal signs a lumbar puncture was performed, resulting inconclusive due to a traumatic technique, and no second sample was obtained. However, the patient was admitted to the neurology department with empirical intravenous antibiotic therapy based on ceftriaxone and vancomycin as well as acyclovir for antiherpetic coverage.</p><p id="par0030" class="elsevierStylePara elsevierViewall">During admission, he underwent a second lumbar puncture with glucose 56<span class="elsevierStyleHsp" style=""></span>mg/dl, protein 68<span class="elsevierStyleHsp" style=""></span>mg/dl and WBC 600/μl (90% polymorphonuclear cells, 10% lymphocytes). Due to the polymorphonuclear pleiocitosis we performed a magnetic resonance imaging (MRI) scan (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>), which showed an extrusion affection with extension to the left cerebral peduncle which appeared isointense on T1, hyperintense on T2 (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>), and flair, and hypointense on T1 IR.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0035" class="elsevierStylePara elsevierViewall">Microbiological studies with Gram and Ziehl–Neelsen stains of the cerebrospinal fluid cultures (including Lowenstein), polymerase chain reaction for herpes virus, Brucella and Borrelia serologies were negative. We also excluded other possible differential diagnoses considering the history of immunosuppression: varicella zoster virus, herpes virus, pox, cytomegalovirus, Candida and meningeal lymphomatosis.</p><p id="par0040" class="elsevierStylePara elsevierViewall">He was assessed by neurology who considered neuroBehçet given the persistence of symptoms despite antibiotic treatment and the medical history of the patient and who initiated infliximab therapy associated with tuberculosis prophylaxis with isoniazid, prior suspension of antibiotic treatment and cyclosporin A, the latter due to the relationship described in the literature with worsening neurologic manifestations.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,4</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Regarding the imaging differential diagnosis it should be noted that, because of its topography and signal intensity, similar images can be seen in early forms of multiple sclerosis and postinfectious encephalopathy. Viral rhomboencephalitis would be a differential diagnosis also, but it is a rare condition in which the injury is unique, extensive and of pontine dominance. Given the history, the clinical and laboratory tests, we oriented the diagnosis toward neuroBehçet.</p><p id="par0050" class="elsevierStylePara elsevierViewall">The patient showed significant clinical improvement and resolution of the headache and nuchal rigidity. A third lumbar puncture was performed 72<span class="elsevierStyleHsp" style=""></span>h after treatment initiation, with glucose 52<span class="elsevierStyleHsp" style=""></span>mg/dl, protein 48<span class="elsevierStyleHsp" style=""></span>mg/dl and 100/μl with predominantly mononuclear leukocytes.</p><p id="par0055" class="elsevierStylePara elsevierViewall">Considering the rapid radiographic and clinical improvement after receiving anti-TNF therapy,<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> we conclude that the patient had BD related aseptic meningitis; the patient was discharged with prednisone 1<span class="elsevierStyleHsp" style=""></span>mg/kg/day, isoniazide-pyridoxine, infliximab and azathioprine. Control brain MRI showed no lesions described in the previous study and presently the patient had no recurrence of neurological affectation.</p><p id="par0060" class="elsevierStylePara elsevierViewall">The central and/or peripheral nervous system affection occurs in about 5%–25% of patients with BD. It is more common in males and usually presents between 3 and 6 years following the onset of illness. Presentation as the first manifestation of the disease is rare.<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6,7</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">Involvement of the central nervous system (CNS) is the most prevalent and there are two clinical forms: parenchymal and non-parenchymal affection.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> This clinical, pathological, radiological differentiation is important and influences prognosis since parenchymal injury is more prevalent (80% of cases) and has a worse prognosis.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> The clinical presentation is nonspecific and varied, including loss of consciousness, seizures, confusion, lethargy, psychiatric disorders, personality changes and dementia.<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">10,11</span></a> Unlike immunosuppressed patients without BD, meningitis can be present as oligosymptomatic, subacute, or associated to cranial nerve disorders.</p><p id="par0070" class="elsevierStylePara elsevierViewall">In the context of an BD our patient presented neurological manifestations and we established the diagnosis of neuroBehçet supported in patient study and prior exclusion of other causes. The presence of signs of meningeal inflammation is common in the CNS parenchymal form; however, the presentation as isolated meningitis is quite rare. This case highlights, therefore, the importance of ruling out an infectious etiology, particularly in patients undergoing immunomodulator treatment.</p><p id="par0075" class="elsevierStylePara elsevierViewall">MRI findings in relation to the progression of the disease and the initial phase T2 hyperintensity areas in the brainstem, basal ganglia and brain hemispheres, followed by an intermediate stage of edema with a mass effect and microhemorrhages and a third stage with brainstem atrophy have been described.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">12,13</span></a> In our case, there was no clinical but rather imaging evidence of thromboencephalic involvement, with findings corresponding in our patient with his initial changes.</p><p id="par0080" class="elsevierStylePara elsevierViewall">In accordance with the above, it is advisable to perform MRI controls for tracking injuries once the treatment is started although these lesions can take months or years to disappear<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">14</span></a>; in our case, we verified resolution after 5 weeks of triple therapy with immunomodulatory therapy.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please, cite this article as: Andrade-Rodado FM, et al. Meningitis aguda en la enfermedad de Behçet. Reumatol Clin. 2014;10:59–61.</p>" ] ] "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 903 "Ancho" => 900 "Tamanyo" => 83819 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Brain MRI.</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Fig. 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1114 "Ancho" => 900 "Tamanyo" => 77085 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Involvement of the pons with extension into the left cerebral peduncle.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:14 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Clinicopathologic features and outcomes of neuro Behçet disease in Spain: a study of 20 patients" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "A. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 17 | 21 | 38 |
| 2024 October | 125 | 67 | 192 |
| 2024 September | 170 | 39 | 209 |
| 2024 August | 149 | 52 | 201 |
| 2024 July | 121 | 66 | 187 |
| 2024 June | 133 | 61 | 194 |
| 2024 May | 126 | 51 | 177 |
| 2024 April | 95 | 42 | 137 |
| 2024 March | 121 | 43 | 164 |
| 2024 February | 105 | 33 | 138 |
| 2024 January | 123 | 29 | 152 |
| 2023 December | 98 | 43 | 141 |
| 2023 November | 102 | 29 | 131 |
| 2023 October | 134 | 50 | 184 |
| 2023 September | 141 | 47 | 188 |
| 2023 August | 113 | 36 | 149 |
| 2023 July | 93 | 43 | 136 |
| 2023 June | 100 | 32 | 132 |
| 2023 May | 146 | 39 | 185 |
| 2023 April | 79 | 20 | 99 |
| 2023 March | 91 | 43 | 134 |
| 2023 February | 97 | 43 | 140 |
| 2023 January | 92 | 29 | 121 |
| 2022 December | 108 | 29 | 137 |
| 2022 November | 101 | 37 | 138 |
| 2022 October | 83 | 38 | 121 |
| 2022 September | 130 | 38 | 168 |
| 2022 August | 89 | 59 | 148 |
| 2022 July | 74 | 58 | 132 |
| 2022 June | 70 | 59 | 129 |
| 2022 May | 74 | 58 | 132 |
| 2022 April | 108 | 60 | 168 |
| 2022 March | 120 | 67 | 187 |
| 2022 February | 154 | 48 | 202 |
| 2022 January | 102 | 53 | 155 |
| 2021 December | 70 | 54 | 124 |
| 2021 November | 84 | 51 | 135 |
| 2021 October | 177 | 66 | 243 |
| 2021 September | 117 | 62 | 179 |
| 2021 August | 119 | 50 | 169 |
| 2021 July | 78 | 47 | 125 |
| 2021 June | 89 | 53 | 142 |
| 2021 May | 141 | 46 | 187 |
| 2021 April | 221 | 98 | 319 |
| 2021 March | 229 | 64 | 293 |
| 2021 February | 150 | 26 | 176 |
| 2021 January | 146 | 41 | 187 |
| 2020 December | 121 | 31 | 152 |
| 2020 November | 142 | 21 | 163 |
| 2020 October | 140 | 22 | 162 |
| 2020 September | 115 | 28 | 143 |
| 2020 August | 94 | 23 | 117 |
| 2020 July | 52 | 21 | 73 |
| 2020 June | 38 | 32 | 70 |
| 2020 May | 35 | 17 | 52 |
| 2020 April | 30 | 20 | 50 |
| 2020 March | 16 | 11 | 27 |
| 2020 February | 1 | 0 | 1 |
| 2018 May | 7 | 0 | 7 |
| 2018 April | 89 | 8 | 97 |
| 2018 March | 117 | 13 | 130 |
| 2018 February | 72 | 3 | 75 |
| 2018 January | 118 | 7 | 125 |
| 2017 December | 59 | 8 | 67 |
| 2017 November | 67 | 7 | 74 |
| 2017 October | 63 | 4 | 67 |
| 2017 September | 61 | 5 | 66 |
| 2017 August | 77 | 5 | 82 |
| 2017 July | 45 | 11 | 56 |
| 2017 June | 93 | 9 | 102 |
| 2017 May | 87 | 6 | 93 |
| 2017 April | 89 | 9 | 98 |
| 2017 March | 79 | 5 | 84 |
| 2017 February | 192 | 8 | 200 |
| 2017 January | 90 | 3 | 93 |
| 2016 December | 113 | 16 | 129 |
| 2016 November | 93 | 0 | 93 |
| 2016 October | 142 | 6 | 148 |
| 2016 September | 177 | 4 | 181 |
| 2016 August | 115 | 7 | 122 |
| 2016 July | 57 | 5 | 62 |
| 2016 June | 0 | 3 | 3 |
| 2016 April | 1 | 0 | 1 |
| 2016 March | 1 | 0 | 1 |
| 2016 February | 1 | 0 | 1 |
| 2015 December | 2 | 0 | 2 |
| 2015 October | 1 | 0 | 1 |
| 2015 August | 2 | 0 | 2 |
| 2015 July | 39 | 5 | 44 |
| 2015 June | 63 | 9 | 72 |
| 2015 May | 83 | 17 | 100 |
| 2015 April | 50 | 12 | 62 |
| 2015 March | 63 | 7 | 70 |
| 2015 February | 70 | 5 | 75 |
| 2015 January | 80 | 13 | 93 |
| 2014 December | 62 | 9 | 71 |
| 2014 November | 45 | 8 | 53 |
| 2014 October | 57 | 18 | 75 |
| 2014 September | 50 | 11 | 61 |
| 2014 August | 38 | 14 | 52 |
| 2014 July | 56 | 19 | 75 |
| 2014 June | 75 | 15 | 90 |
| 2014 May | 67 | 23 | 90 |
| 2014 April | 67 | 19 | 86 |
| 2014 March | 86 | 31 | 117 |
| 2014 February | 61 | 22 | 83 |
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