Scientific Letter
Meningoencephalitis as a presentation form of Behçet
Meningoencefalitis de tronco como presentación de Behçet
a Servicio de Pediatría, Hospital Sant Joan de Déu, Universitat de Barcelona, Esplugues de Llobregat, Barcelona, Spain
b Servicio de Radiología, Hospital Sant Joan de Déu, Universitat de Barcelona, Esplugues de Llobregat, Barcelona, Spain
c Unidad de Reumatología Pediátrica, Servicio de Pediatría, Hospital Sant Joan de Déu, Universitat de Barcelona, Esplugues de Llobregat, Barcelona, Spain
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Rodà, A. Martínez-Monseny, M. Rebollo, E. Iglesias" "autores" => array:4 [ 0 => array:4 [ "nombre" => "D." "apellidos" => "Rodà" "email" => array:1 [ 0 => "droda@hsjdbcn.org" ] "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" => "A." "apellidos" => "Martínez-Monseny" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "M." "apellidos" => "Rebollo" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "E." "apellidos" => "Iglesias" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] ] "afiliaciones" => array:3 [ 0 => array:3 [ "entidad" => "Servicio de Pediatría, Hospital Sant Joan de Déu, Universitat de Barcelona, Esplugues de Llobregat, Barcelona, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Radiología, Hospital Sant Joan de Déu, Universitat de Barcelona, Esplugues de Llobregat, Barcelona, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Unidad de Reumatología Pediátrica, Servicio de Pediatría, Hospital Sant Joan de Déu, Universitat de Barcelona, Esplugues de Llobregat, Barcelona, Spain" "etiqueta" => "c" "identificador" => "aff0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Meningoencefalitis de tronco como presentación de Behçet" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 651 "Ancho" => 1800 "Tamanyo" => 138494 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Axial T1 SPGR: increased enhancement of both cerebellar peduncles. (B) Axial T2 FSE: hyperintense T2 signal in the right cerebral peduncle, with extension of the signal abnormality towards the periaqueductal grey region. (C) Axial T2 FSE: resolution of abnormalities after treatment.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Behçet's disease (BD) is a disease of unknown aetiology that is rare in the paediatric population. Its typical features are recurrent oral and genital ulcers associated with other systemic ocular, cutaneous, neurologic, vascular and articular manifestations. In exceptional cases, the onset occurs with central nervous system manifestations. We describe the case of a female patient aged 10 years with brainstem meningoencephalitis as the initial presentation of BD that responded well to immunosuppressive therapy.</p><p id="par0010" class="elsevierStylePara elsevierViewall">The patient, born to consanguineous Maghrebi parents, sought care for fever of four days’ duration, oral ulcers, headache and sleepiness. The parents reported that in the past 18 months she had experienced recurrent, but not periodical, four-day episodes of fever (maximum temperature, 39.5<span class="elsevierStyleHsp" style=""></span>°C) and oral ulcers that had not been investigated. The physical examination revealed fever, three ulcers in the oral mucosa, lethargy, nuchal rigidity and upbeat nystagmus with no other apparent neurological abnormalities. The following diagnostic tests were performed: complete blood count and blood chemistry panel (normal [<span class="elsevierStyleItalic">N</span>]); erythrocyte sedimentation rate (18<span class="elsevierStyleHsp" style=""></span>mm/h; <span class="elsevierStyleItalic">N</span><span class="elsevierStyleHsp" style=""></span><<span class="elsevierStyleHsp" style=""></span>20); C-reactive protein (89<span class="elsevierStyleHsp" style=""></span>mg/L; <span class="elsevierStyleItalic">N</span><span class="elsevierStyleHsp" style=""></span><<span class="elsevierStyleHsp" style=""></span>15), eye fundus examination (<span class="elsevierStyleItalic">N</span>), lumbar puncture (predominantly mononuclear pleocytosis with 950 white blood cells/mL; <span class="elsevierStyleItalic">N</span><span class="elsevierStyleHsp" style=""></span><<span class="elsevierStyleHsp" style=""></span>10), cerebrospinal fluid protein (mildly elevated at 68<span class="elsevierStyleHsp" style=""></span>mg/mL; <span class="elsevierStyleItalic">N</span>, 15–40) and glucose (<span class="elsevierStyleItalic">N</span>).</p><p id="par0015" class="elsevierStylePara elsevierViewall">Meningoencephalitis was suspected and empirical treatment with intravenous acyclovir initiated while awaiting the results of PCR tests for herpes simplex virus 1 and 2, which turned out negative. The patient was tested for infectious agents (Mantoux test, antibody testing for HIV, <span class="elsevierStyleItalic">Borrelia</span>, <span class="elsevierStyleItalic">Listeria</span> and syphilis) and cancer (chest radiograph, blood differential test and LDH), and no abnormalities were found. Faecal calprotectin was measured to rule out intestinal inflammatory disease, and was found to be normal.</p><p id="par0020" class="elsevierStylePara elsevierViewall">During hospitalisation, she experienced palsy of the VI cranial nerves with ongoing fluctuations in the level of consciousness, leading to performance of a head CT scan with contrast that showed no abnormalities. Forty-eight hours later, the patient underwent a cranial MRI scan that showed a hyperintense T2 signal at the level of the brainstem, centred at the midbrain and extending towards the cerebral peduncles and the pons, with leptomeningeal enhancement and an uptaking punctiform focus at the level of the right cerebellar peduncle (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A and B). Given the presence of a brain lesion probably caused by inflammation, the patient was screened for autoimmune diseases, and the antinuclear antibodies, complement and antineutrophil cytoplasmic antibodies tests were all negative. The patient underwent genetic testing for hereditary autoinflammatory diseases (familial Mediterranean fever, periodic fever syndrome due to mevalonate kinase deficiency, TRAPS, BLAU syndrome/early-onset sarcoidosis) that found no mutation associated with her disease. The test for HLA-B51 was negative.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">Behçet's disease was suspected based on the clinical presentation and imaging findings, leading to initiation of empirical immunosuppressive therapy with corticosteroids (first with intravenous methylprednisolone bolus injection at 30<span class="elsevierStyleHsp" style=""></span>mg/kg/day for 3 days, and then by the oral route at 2<span class="elsevierStyleHsp" style=""></span>mg/kg/day), colchicine (0.5<span class="elsevierStyleHsp" style=""></span>mg every 12<span class="elsevierStyleHsp" style=""></span>h by the oral route) and azathioprine (1.5<span class="elsevierStyleHsp" style=""></span>mg/kg/day by the oral route).</p><p id="par0030" class="elsevierStylePara elsevierViewall">The patient responded well to the treatment, with a gradual improvement of symptoms that allowed the progressive tapering and eventual discontinuation of corticosteroids. The followup MRI at six months after initiation of treatment did not show any pathological changes (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>C). At present, the patient continues treatment with azathioprine and colchicine, with good results.</p><p id="par0035" class="elsevierStylePara elsevierViewall">Behçet's disease is diagnosed based on clinical manifestations, and there are no validated classification criteria for the paediatric population. Koné-Paut et al. created an international registry of paediatric patients with BD with the purpose of defining the characteristics of paediatric patients with the disease and subsequently developing classification criteria adapted to this population.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">1</span></a> In 26% of cases of BD, the onset occurs before age 16 years.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">2</span></a> Neurological involvement has been described in 5% to 30% of BD cases in children, most frequently with central nervous system involvement in the form of thrombosis.<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">3–5</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Parenchymal involvement is typically found at the level of the brainstem, appearing as a single inflammatory oedematous lesion that is hyperintense on T2 and iso- to hypointense on T1, associated with meningitis.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">6</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">To date, no randomised clinical trials have been conducted for the treatment of BD.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">7</span></a> The goals of treatment are symptom control and preventing damage to the affected organ, usually by administration of corticosteroids or immunosuppressants such as azathioprine, ciclosporin or anti-TNF alpha.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">7</span></a> Advances in the knowledge of the pathophysiology of BD will help define a more specific therapeutic approach.</p><p id="par0050" class="elsevierStylePara elsevierViewall">Neurological involvement is an exceptional initial presentation in childhood BD. The characteristic neuroimaging findings and the accompanying characteristic symptoms guide the clinical diagnosis.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2015-06-15" "fechaAceptado" => "2015-09-14" "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Rodà D, Martínez-Monseny A, Rebollo M, Iglesias E. Meningoencefalitis de tronco como presentación de Behçet. An Pediatr (Barc). 2016;84:117–118.</p>" ] ] "multimedia" => array:1 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 651 "Ancho" => 1800 "Tamanyo" => 138494 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Axial T1 SPGR: increased enhancement of both cerebellar peduncles. (B) Axial T2 FSE: hyperintense T2 signal in the right cerebral peduncle, with extension of the signal abnormality towards the periaqueductal grey region. (C) Axial T2 FSE: resolution of abnormalities after treatment.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:7 [ 0 => array:3 [ "identificador" => "bib0040" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Registries in rheumatological and musculoskeletal conditions. Paediatric Behçet's disease: an international cohort study of 110 patients. One-year follow-up data" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "I. Koné-Paut" 1 => "M. Darce-Bello" 2 => "F. Shahram" 3 => "M. Gattorno" 4 => "R. Cimaz" 5 => "S. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 7 | 12 | 19 |
| 2024 October | 31 | 37 | 68 |
| 2024 September | 46 | 41 | 87 |
| 2024 August | 63 | 50 | 113 |
| 2024 July | 42 | 28 | 70 |
| 2024 June | 46 | 40 | 86 |
| 2024 May | 43 | 41 | 84 |
| 2024 April | 44 | 31 | 75 |
| 2024 March | 43 | 26 | 69 |
| 2024 February | 30 | 30 | 60 |
| 2024 January | 27 | 21 | 48 |
| 2023 December | 28 | 22 | 50 |
| 2023 November | 46 | 35 | 81 |
| 2023 October | 27 | 28 | 55 |
| 2023 September | 24 | 17 | 41 |
| 2023 August | 28 | 18 | 46 |
| 2023 July | 31 | 28 | 59 |
| 2023 June | 65 | 25 | 90 |
| 2023 May | 57 | 28 | 85 |
| 2023 April | 33 | 18 | 51 |
| 2023 March | 37 | 21 | 58 |
| 2023 February | 32 | 15 | 47 |
| 2023 January | 22 | 25 | 47 |
| 2022 December | 50 | 26 | 76 |
| 2022 November | 58 | 32 | 90 |
| 2022 October | 57 | 40 | 97 |
| 2022 September | 52 | 34 | 86 |
| 2022 August | 46 | 41 | 87 |
| 2022 July | 55 | 45 | 100 |
| 2022 June | 41 | 35 | 76 |
| 2022 May | 32 | 32 | 64 |
| 2022 April | 44 | 47 | 91 |
| 2022 March | 54 | 41 | 95 |
| 2022 February | 39 | 38 | 77 |
| 2022 January | 41 | 37 | 78 |
| 2021 December | 39 | 33 | 72 |
| 2021 November | 53 | 58 | 111 |
| 2021 October | 50 | 50 | 100 |
| 2021 September | 43 | 40 | 83 |
| 2021 August | 29 | 46 | 75 |
| 2021 July | 36 | 31 | 67 |
| 2021 June | 41 | 36 | 77 |
| 2021 May | 40 | 23 | 63 |
| 2021 April | 85 | 50 | 135 |
| 2021 March | 71 | 26 | 97 |
| 2021 February | 44 | 21 | 65 |
| 2021 January | 32 | 24 | 56 |
| 2020 December | 27 | 33 | 60 |
| 2020 November | 26 | 19 | 45 |
| 2020 October | 21 | 18 | 39 |
| 2020 September | 28 | 25 | 53 |
| 2020 August | 34 | 16 | 50 |
| 2020 July | 42 | 16 | 58 |
| 2020 June | 40 | 17 | 57 |
| 2020 May | 37 | 33 | 70 |
| 2020 April | 25 | 16 | 41 |
| 2020 March | 22 | 21 | 43 |
| 2020 February | 37 | 20 | 57 |
| 2020 January | 19 | 12 | 31 |
| 2019 December | 36 | 21 | 57 |
| 2019 November | 23 | 12 | 35 |
| 2019 October | 19 | 19 | 38 |
| 2019 September | 15 | 12 | 27 |
| 2019 August | 44 | 22 | 66 |
| 2019 July | 38 | 22 | 60 |
| 2019 June | 28 | 25 | 53 |
| 2019 May | 62 | 25 | 87 |
| 2019 April | 70 | 27 | 97 |
| 2019 March | 47 | 16 | 63 |
| 2019 February | 50 | 18 | 68 |
| 2019 January | 50 | 16 | 66 |
| 2018 December | 49 | 31 | 80 |
| 2018 November | 77 | 46 | 123 |
| 2018 October | 92 | 32 | 124 |
| 2018 September | 42 | 21 | 63 |
| 2018 August | 2 | 0 | 2 |
| 2018 July | 1 | 0 | 1 |
| 2018 June | 4 | 0 | 4 |
| 2018 May | 4 | 0 | 4 |
| 2018 April | 57 | 0 | 57 |
| 2018 March | 14 | 0 | 14 |
| 2018 February | 13 | 0 | 13 |
| 2018 January | 18 | 0 | 18 |
| 2017 December | 18 | 0 | 18 |
| 2017 November | 11 | 0 | 11 |
| 2017 October | 12 | 0 | 12 |
| 2017 September | 13 | 0 | 13 |
| 2017 August | 11 | 0 | 11 |
| 2017 July | 11 | 1 | 12 |
| 2017 June | 24 | 4 | 28 |
| 2017 May | 13 | 5 | 18 |
| 2017 April | 8 | 3 | 11 |
| 2017 March | 7 | 17 | 24 |
| 2017 February | 6 | 4 | 10 |
| 2017 January | 9 | 0 | 9 |
| 2016 December | 21 | 6 | 27 |
| 2016 November | 33 | 6 | 39 |
| 2016 October | 35 | 7 | 42 |
| 2016 September | 36 | 1 | 37 |
| 2016 August | 36 | 5 | 41 |
| 2016 July | 22 | 6 | 28 |
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