Letter to the Editor
Pseudotumoral neuro-behçet in a patient treated with anti-tumor necrosis factor alpha
Síndrome neurológico seudotumoral de la enfermedad de Behçet en un paciente tratado con antagonistas del factor de necrosis tumoral
Lina Martínez-Estupiñán
, Francisco Javier López-Longo, Indalecio Monteagudo, Luis Carreño Pérez
Corresponding author
Servicio de Reumatología, Hospital General Universitario Gregorio Marañón, Madrid, Spain
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There is another area in the posterior cavity with signal disturbance showing mass effect affecting right-sided cerebellar hemisphere. These lesions are T2-hyperintense with some internal heterogeneous areas.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">One of the main causes of morbimortality is the central nervous system condition caused by Behçet's disease.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a> It is classified in 2 main categories: nonparenchymal, causing vascular deterioration, and parenchymal, which develops like an aseptic meningoencephalitis.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> In some cases, there might be extensive brain lesions known as pseudotumoral neuro-Behçet's disease.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> In 2012, Noel et al. published a compilation of 23 cases reported in medical literature, and they estimated that this presentation had a 1.8% prevalence among neurological manifestations. The most common histopathological finding was inflammatory perivasculitis. The typical image seen on magnetic resonance imaging (MRI) was a space-occupying lesion with mass effect, T1-hypointense enhanced by contrast, and T2-hyperintense; lesions were mostly located at the capsulolenticular, thalamus, brain stem, basal ganglia and cerebral cortex areas.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> We present the case of a patient who developed a severe, treatment-refractory, pseudotumoral syndrome.</p><p id="par0010" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">Clinical case</span>: 23-year old male diagnosed with Behçet's disease in 2008, initially treated with prednisone 20–30<span class="elsevierStyleHsp" style=""></span>mg/day and colchicine, with partial response. In February 2011, he was given a treatment based on infliximab (IFX); 4 months later the patient had an adverse reaction, and the drug was changed for adalimumab (ADA). In October 2011, the patient started experiencing gait alterations, dizziness and vomiting, and had to be admitted. The MRI showed a pseudotumoral lesion at the right hemicerebellum, and less extensive injuries in the white substance at the right-sided inner capsule anterior arm, and in the area near mesial arm temporal structures (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). In addition to ADA, the patient was administered oral azathioprine (1<span class="elsevierStyleHsp" style=""></span>mg/kg/day) and methylprednisolone (MP) by intravenous pulses (IV) (500<span class="elsevierStyleHsp" style=""></span>mg/day/5 days), with disease remission. A month later the patient was readmitted with fever, behavioural disturbances, hallucinations and short-term memory loss. The cerebrospinal fluid analysis evidenced aseptic meningitis without monoclonal expansion. The patient received new pulses of MP (500<span class="elsevierStyleHsp" style=""></span>mg/day/5 days), showing a good response; he was started on a treatment with tocilizumab (TCZ), and when corticosteroids were orally administered, symptoms reappeared. The brain MRI showed an inflammatory extensive lesion at the limbic system structures, more accentuated on the left side. A new MP cycle (500<span class="elsevierStyleHsp" style=""></span>mg/day/5 day) including IV cyclophosphamide IV (500<span class="elsevierStyleHsp" style=""></span>mg/month) was administered. After a month, the patient developed right-sided hemiparesis and suffered deterioration of his supratentorial and infratentorial lesions, as shown by the MRI. A new MP cycle (1<span class="elsevierStyleHsp" style=""></span>g/day/5 days) including rituximab was administered, and the patient received 5 sessions of plasmapheresis. Due to the negative response to the treatment, a brain biopsy was performed, causing a 5<span class="elsevierStyleHsp" style=""></span>cm<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>3<span class="elsevierStyleHsp" style=""></span>cm intraparenchymal bruise above left-sided basal ganglia. The histological study showed reagent-like changes; there were no signs of encephalitis or atypical lymphoid elements. Two days after completing the brain biopsy, the patient had an epileptic seizure with a prolonged post-critical period, having to be admitted to the intensive care unit. After 5 months of undergoing an immunosuppressant treatment, the patient passed away due to an intraparechymal haemorrhage.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">Discussion</span>: no medical strategy to treat neuro-Behçet's disease has been ratified in controlled clinic trials<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">4</span></a> to date. Most patients with pseudotumoral lesions received a treatment with high doses of steroids along with immunosuppressants, and in 2011, the case of a patient who obtained clinical remission<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">3–5</span></a> after being treated with biological therapies (daclizumab, IFX and TCZ) was reported. The neurological symptoms experienced by our patient had first appeared during the treatment with <span class="elsevierStyleItalic">tumour necrosis factor</span> (TNF) blockers, a drug that has been associated with the development of demyelinating lesions that, typically, improve when the treatment is suspended.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">6</span></a> The brain lesions sustained by our patient were extensive and persisted after suspending the anti-TNF treatment; the corticosteroids and immunosuppressants administered proved to be inefficient. In the series compiling 23 cases, 2 patients passed away (8.7%) and 7 (30.5%) had some sort of sequelae,<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> confirming that pseudotumoral neuro-Behçet is one of the presentations of the Behçet's disease, increasing the risk of suffering severe sequelae and threatening life.</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: Martínez-Estupiñán L, López-Longo FJ, Monteagudo I, Carreño Pérez L. Síndrome neurológico seudotumoral de la enfermedad de Behçet en un paciente tratado con antagonistas del factor de necrosis tumoral. Med Clin (Barc). 2015;144:235–236.</p>" ] ] "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" => 872 "Ancho" => 1400 "Tamanyo" => 161561 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Magnetic resonance axial brain image: there is an area at the supratentorial compartment where signal disturbance pertaining the white substance adjacent to the left-side inferior horn is observed. There is another area in the posterior cavity with signal disturbance showing mass effect affecting right-sided cerebellar hemisphere. These lesions are T2-hyperintense with some internal heterogeneous areas.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:6 [ 0 => array:3 [ "identificador" => "bib0035" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "The long-term mortality and morbidity of Behçet syndrome: a 2-decade outcome survey of 387 patients followed at a dedicated center" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "E. Kural-Seyahi" 1 => "I. Fresko" 2 => "N. Seyahi" 3 => "Y. Ozyazgan" 4 => "C. Mat" 5 => "V. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2023 March | 2 | 0 | 2 |
| 2017 October | 2 | 2 | 4 |
| 2017 September | 6 | 2 | 8 |
| 2017 August | 10 | 2 | 12 |
| 2017 July | 5 | 3 | 8 |
| 2017 June | 5 | 3 | 8 |
| 2017 May | 4 | 5 | 9 |
| 2017 April | 10 | 6 | 16 |
| 2017 March | 12 | 9 | 21 |
