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A: secuencia FLAIR axial; B: secuencia sagital T2; C: secuencia axial T2. TC craneal posoperatoria sin contraste. Muestra la posición normal del catéter ventricular en el tercer ventrículo. Se observó reducción del tamaño del sistema ventricular y mejoría del edema transependimario. D: secuencia FLAIR axial. E: secuencia sagital T2. F: secuencia axial T2. 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A: axial FLAIR sequence; B: sagittal T2 sequence; C: axial T2 sequence. Postoperative cranial CT scan without contrast. Shows the normal position of the ventricular catheter in the third ventricle. Reduction of the size of the ventricular system and improvement of the transependymal oedema were observed. D: axial FLAIR sequence. E: sagittal T2 sequence. F: axial T2 sequence. G: 3D reconstruction of a cranial CT scan showing normal positioning of the Medtronic fixed pressure VPS valve reservoir.</p> <p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">CT: computed tomography; FLAIR: fluid attenuated inversion recovery; VPS: ventriculoperitoneal shunt.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">POEMS syndrome is a rare paraneoplastic syndrome due to an underlying plasma cell disorder. It includes the presence of polyneuropathy, organomegaly, endocrinopathy, monoclonal (M) protein production secondary to monoclonal plasma cell proliferation, skin changes, thrombocytosis, papilledema and elevated vascular endothelial growth factor (VEGF) levels.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> The approximate prevalence rate is 0.3 per 100,000 population.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Early diagnosis is essential to limit morbidity and mortality and is based on the criteria established by Dispenzieri.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> There are two main criteria that must be met (typically demyelinating polyneuropathy and monoclonal plasma cell proliferative disorder); one major criterion (Castleman's disease, sclerotic bone lesions or VEGF elevation); and one minor criterion (organomegaly, extravascular volume overload, endocrinopathy, skin changes, papilledema and thrombocytosis). Median survival is around 14 months, probably due to the high rate of complications.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2–4</span></a> Several studies report increased intracranial pressure (ICP) and papilledema, requiring treatment with acetazolamide and repeated lumbar punctures, although none describe the presence of hydrocephalus.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,4</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">A 51-year-old woman previously diagnosed with monoclonal gammopathy of uncertain significance, immunoglobulin G (IgG) Kappa reported distal dysesthesias in both hands and progressive weakness of all four limbs in the last five years. Examination revealed moderate dysarthria, with muscle balance of 3/5 predominantly proximal and areflexia in all four limbs, as well as hypoaesthesia and hypopallesthesia in the lower limbs, walking with support. The neurophysiological study showed a very severe generalised sensory-motor polyneuropathy in all four limbs (absence of sensory and motor potentials in all nerves studied), with presence of signs of reinnervation. Blood tests showed platelets 540,000/μL (150–370), gamma fraction 4.21 g/dL (0.8–1.35), serum IgG of 4227 mg/dL (725−1900), and VEGF > 1000. Lumbar puncture showed a significantly elevated spinal fluid protein count with protein 503.8 mg/dL (15–45), lactate 4.5 and nucleated cells 75/mm3. Initial cerebrospinal fluid (CSF) opening pressure was 17 cm H2O. Bilateral papilledema was observed in the fundus of the eye. With these clinical findings a diagnosis of IgG Kappa POEMS syndrome (polyneuropathy, monoclonal plasma cell proliferation, elevated VEGF, papilledema, thrombocytosis) was made and treatment with lenalidomide 25 mg daily and dexamethasone 40 mg daily for 21 days was started. Three months after diagnosis, she presented with general motor impairment (muscle balance 1/5) and worsening dysarthria, requiring assistance with basic activities of daily living. A cranial MRI scan was then performed which showed tetraventricular hydrocephalus with transependymal oedema (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A–C). An external ventricular drain was implanted for seven days with clear improvement in strength and dysarthria, so a Medtronic (Medtronic Sofamor Danek, Memphis, TN, USA) fixed pressure ventriculoperitoneal shunt (VPS) valve was implanted (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>G). CSF opening pressure was 25 cm H2O. Postoperative cranial CT scan showed clear improvement of the hydrocephalus (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>D–F). The patient was discharged ambulatory with assistance and required a wheelchair for long transfers.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Our patient had already been diagnosed with POEMS syndrome when we assessed her for a sudden motor and language deterioration. As this was not justified by the previous polyneuropathy, we performed a cranial MRI which showed clear tetraventricular hydrocephalus with transependymal oedema. It was treated with a VPS valve and the patient improved in the first few days. We suggested that the hydrocephalus might be due to the high level of protein observed in the patient's CSF (503.8 mg/dL), which probably caused obstruction of the arachnoid granulations, and thus changes in CSF circulation and reabsorption.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Hydrocephalus could be a possible diagnosis for any neurological worsening in these patients. Although this is an isolated case, we believe that POEMS syndrome may have central neurological involvement more often than previously thought, rather than just peripheral involvement. It is possible that early detection with imaging tests could prevent complications and allow early treatment, which could change clinicians' attitudes.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Ethical considerations</span><p id="par0025" class="elsevierStylePara elsevierViewall">The authors declare that the protocols and procedures of our centre have been followed with regard to the publication of patient data.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Funding</span><p id="par0030" class="elsevierStylePara elsevierViewall">This work has not received any funding.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflict of interest</span><p id="par0035" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Ethical considerations" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Funding" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Conflict of interest" ] 3 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "multimedia" => array:1 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1441 "Ancho" => 3333 "Tamanyo" => 459491 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A–C: preoperative non-contrast cranial MRI showing clear tetraventricular dilatation with transependymal oedema (white arrows), compatible with a diagnosis of communicating hydrocephalus. A: axial FLAIR sequence; B: sagittal T2 sequence; C: axial T2 sequence. Postoperative cranial CT scan without contrast. Shows the normal position of the ventricular catheter in the third ventricle. Reduction of the size of the ventricular system and improvement of the transependymal oedema were observed. D: axial FLAIR sequence. E: sagittal T2 sequence. F: axial T2 sequence. G: 3D reconstruction of a cranial CT scan showing normal positioning of the Medtronic fixed pressure VPS valve reservoir.</p> <p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">CT: computed tomography; FLAIR: fluid attenuated inversion recovery; VPS: ventriculoperitoneal shunt.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:4 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Ventriculoperitoneal shunt placement for POEMS syndrome" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "C.A. Alvarez-Breckenridge" 1 => "M.A. Attiah" 2 => "M. Zachariah" 3 => "A. Gummadavelli" 4 => "J. Yang" 5 => "P.J. 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Journal Information
Vol. 163. Issue 4.
Pages 212-213 (August 2024)
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Vol. 163. Issue 4.
Pages 212-213 (August 2024)
Letter to the Editor
Hydrocephalus associated with POEMS syndrome
Hidrocefalia asociada al síndrome de POEMS
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