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A) Tinción con hematoxilina-eosina: músculo esquelético con arquitectura conservada sin engrosamiento conectivo endomisial ni infiltración por tejido adiposo. Se identifican frecuentes fibras regenerativas, así como necróticas con fenómenos de miofagocitosis. B) ATPasa 9,4: muestra reagrupamiento por tipos de fibras, con presencia de grupos de fibras tipo 1 y tipo 2, compatible con reinervación.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "A. Chavarría-Miranda, A. Hernández Lain, O. Toldos González, M.I. Pedraza Hueso" "autores" => array:4 [ 0 => array:2 [ "nombre" => "A." "apellidos" => "Chavarría-Miranda" ] 1 => array:2 [ "nombre" => "A." "apellidos" => "Hernández Lain" ] 2 => array:2 [ "nombre" => "O." "apellidos" => "Toldos González" ] 3 => array:2 [ "nombre" => "M.I." 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B) <span class="elsevierStyleSup">18</span>F-FDG PET study revealing a hypermetabolic mass in the right lung, with nodules in the left lower lobe (stars) and diffuse bone metastases (arrows). C) Round cell tumour proliferation. The histological study revealed cells with ill-defined cytoplasm and hyperchromatic nuclei (although most cells were non-nucleated), and abnormal mitosis. D) The immunohistochemical study revealed positivity for anti-CD99 antibodies.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "A. Zegmout, H. Asri, I.A. Rhorfi, A. Abid" "autores" => array:4 [ 0 => array:2 [ "nombre" => "A." "apellidos" => "Zegmout" ] 1 => array:2 [ "nombre" => "H." "apellidos" => "Asri" ] 2 => array:2 [ "nombre" => "I.A." "apellidos" => "Rhorfi" ] 3 => array:2 [ "nombre" => "A." 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A and B) Coronal and axial T2-weighted sequences, showing partial fusion of the cerebellar hemispheres and continuity of the folia (solid red arrow) and fissures (dashed yellow arrow) across the midline. C) Sagittal T1-weighted sequence showing absence of the primary (solid blue arrow) and prepyramidal fissures (dashed green arrow). D) Axial SWI sequence showing proximity of the dentate nuclei to the midline (red arrows). E) Coronal T2-weighted sequence showing left-sided incomplete hippocampal inversion (dashed red arrow), associated with vertical orientation of the collateral sulcus (solid yellow arrow) and dilation of the ipsilateral temporal horn (asterisk). F) Axial SWI sequence showing capillary telangiectasia in the right middle cerebellar peduncle.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "J.M. Sánchez-Villalobos, A.M. Torres-Perales, L. Serrano-Velasco, J.A. Pérez-Vicente" "autores" => array:4 [ 0 => array:2 [ "nombre" => "J.M." 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Chavarría-Miranda, A. Hernández Lain, O. Toldos González, M.I. Pedraza Hueso" "autores" => array:4 [ 0 => array:4 [ "nombre" => "A." "apellidos" => "Chavarría-Miranda" "email" => array:1 [ 0 => "alba-chavarria@hotmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "A." "apellidos" => "Hernández Lain" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "O." "apellidos" => "Toldos González" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "M.I." "apellidos" => "Pedraza Hueso" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Neurología, Hospital Clínico Universitario de Valladolid, Valladolid, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Anatomía Patológica, Hospital Universitario Doce de Octubre, Madrid, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Miopatía necrotizante inmunomediada tras tratamiento con adalimumab en paciente con espondilitis anquilosante HLA-B27" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1085 "Ancho" => 2625 "Tamanyo" => 298339 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Haematoxylin-eosin staining and immunohistochemical study of a muscle specimen. A) Haematoxylin-eosin staining: skeletal muscle with normal architecture, without thickening of endomysial connective tissue or adipose tissue infiltration. The image shows fibre regeneration, as well as fibre necrosis, with signs of myophagocytosis. B) ATPase pH 9.4 stain: rearrangement by fibre type, with groups of type 1 and type 2 fibres, a finding suggestive of reinnervation.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Autoimmune myopathies are rare, with a prevalence of 9-14 cases per 100 000 population.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Immune-mediated necrotising myopathy (IMNM) is characterised by proximal muscle weakness, elevated creatine kinase (CK) levels, muscle fibre necrosis with signs of regeneration, and minimal or no inflammatory infiltration in muscle biopsy.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Development of IMNM has been associated with statin use,<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> certain connective tissue disorders, and HIV infection.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,4</span></a> According to published series,<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> some patients test positive for anti-HMGCR (33%) or anti-SRP antibodies (24%), although up to 20%-30% of patients are seronegative.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> We present a case of immune-mediated necrotising myopathy following treatment with adalimumab in a patient with HLA-B27 ankylosing spondylitis.</p><p id="par0010" class="elsevierStylePara elsevierViewall">Our patient was a 55-year-old man with history of psoriasis and HLA-B27 ankylosing spondylitis of 20 years’ progression, who had been under treatment with adalimumab for the previous 2 years. He consulted due to predominantly proximal muscle pain in the limbs, starting 6 months after onset of adalimumab treatment. The patient reported greater difficulty walking and climbing up and down stairs, and loss of muscle mass in the arms and legs. The neurological examination revealed bilateral paresis (Medical Research Council grade 4/5) in the psoas muscle and quadriceps, and muscle atrophy in the deltoid and quadriceps muscles. We detected no fasciculations or any other spontaneous muscle activity, and the patient did not present scapular winging. Deep tendon reflexes were present and gait was normal.</p><p id="par0015" class="elsevierStylePara elsevierViewall">Laboratory analysis revealed elevated CK levels (385 IU/L) at 7 months after treatment onset; results from a complete blood count, erythrocyte sedimentation rate, aldolase level determination, liver profile, kidney profile, and serology testing were all within normal ranges. Due to suspicion of myopathy, we performed an MRI scan of the lower limbs, which showed no alterations, and an electromyography study, finding no signs of myopathy. CK levels were determined periodically, with a maximum level of 598 IU/L. A biopsy of the quadriceps muscle revealed signs compatible with IMNM (<a class="elsevierStyleCrossRefs" href="#fig0005">Figs. 1 and 2</a>). Tests for antinuclear, anti-ENA, anti-HMGCR, and anti-SRP antibodies and a myositis autoantibody panel yielded negative results. A chest-abdomen-pelvis CT scan ruled out underlying neoplasia.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">In view of the anatomical pathology findings, we suspended adalimumab and started corticosteroid therapy, initially with intravenous methylprednisolone at 500 mg for 3 days, followed by oral prednisone in decreasing doses until reaching a maintenance dose of 20 mg/day. At 6 months, CK levels had decreased to 120-130 IU/L and motor deficits had improved slightly, although muscle pain persisted. Azathioprine 50 mg/12 h slightly improved pain.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Tumour necrosis factor-α (TNF-α) inhibitors are increasingly used for the treatment of autoimmune diseases. Paradoxically, due to their immunomodulatory effects, cases have been reported of immune disorders associated with use of TNF-α inhibitors, including vasculitis, lupus-like syndrome, and interstitial lung disease.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> Case series have been published of patients receiving treatment with TNF-α inhibitors and presenting onset of such immune-mediated myopathies as myositis and dermatomyositis.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">7,8</span></a> Despite this potential association, no cases have previously been described of IMNM associated with adalimumab use. In our patient, the short time between onset of treatment with adalimumab and onset of symptoms and the improvement in CK levels and muscle pain after adalimumab discontinuation suggest that this drug played a role in the development of myopathy.</p><p id="par0030" class="elsevierStylePara elsevierViewall">The recommended treatment for IMNM is induction therapy with oral or intravenous corticosteroids; an immunosuppressant may be added at onset of corticosteroid therapy or during the following month, depending on symptom severity and initial response to treatment. Acceptable response has been reported for such immunosuppressants as methotrexate, azathioprine, ciclosporin, and mycophenolate mofetil.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> Combination therapy with several immunosuppressants may be necessary in 50% of cases; intravenous immunoglobulins or rituximab constitute an alternative for refractory cases, or may even be administered during induction therapy.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> In conclusion, TNF-α inhibitors have been associated with the development of immune-mediated myopathy; identification of the causal factor and early discontinuation of the drug are essential for disease prognosis.</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: Chavarría-Miranda A, Hernández Lain A, Toldos González O, Pedraza Hueso MI. Miopatía necrosante inmunomediada tras tratamiento con adalimumab en paciente con espondilitis anquilosante HLA-B27. Neurología. 2021;36:631–632.</p>" ] ] "multimedia" => array:2 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1085 "Ancho" => 2625 "Tamanyo" => 298339 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Haematoxylin-eosin staining and immunohistochemical study of a muscle specimen. A) Haematoxylin-eosin staining: skeletal muscle with normal architecture, without thickening of endomysial connective tissue or adipose tissue infiltration. The image shows fibre regeneration, as well as fibre necrosis, with signs of myophagocytosis. B) ATPase pH 9.4 stain: rearrangement by fibre type, with groups of type 1 and type 2 fibres, a finding suggestive of reinnervation.</p>" ] ] 1 => array:8 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1267 "Ancho" => 1583 "Tamanyo" => 206679 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Immunohistochemical study of the membrane attack complex: C5b9 deposition in endomysial capillaries.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:9 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Immune-mediated necrotizing myopathy" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "Pinal-Fernández" 1 => "L. 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2024 November | 9 | 0 | 9 |
2024 October | 54 | 5 | 59 |
2024 September | 52 | 16 | 68 |
2024 August | 71 | 21 | 92 |
2024 July | 54 | 14 | 68 |
2024 June | 61 | 12 | 73 |
2024 May | 28 | 7 | 35 |
2024 April | 41 | 8 | 49 |
2024 March | 63 | 6 | 69 |
2024 February | 74 | 2 | 76 |
2024 January | 90 | 10 | 100 |
2023 December | 69 | 16 | 85 |
2023 November | 90 | 7 | 97 |
2023 October | 71 | 5 | 76 |
2023 September | 52 | 6 | 58 |
2023 August | 49 | 7 | 56 |
2023 July | 41 | 7 | 48 |
2023 June | 52 | 8 | 60 |
2023 May | 111 | 4 | 115 |
2023 April | 69 | 3 | 72 |
2023 March | 45 | 11 | 56 |
2023 February | 33 | 8 | 41 |
2023 January | 46 | 14 | 60 |
2022 December | 43 | 8 | 51 |
2022 November | 72 | 10 | 82 |
2022 October | 78 | 17 | 95 |
2022 September | 40 | 12 | 52 |
2022 August | 90 | 14 | 104 |
2022 July | 68 | 7 | 75 |
2022 June | 55 | 10 | 65 |
2022 May | 48 | 4 | 52 |
2022 April | 46 | 12 | 58 |
2022 March | 58 | 12 | 70 |
2022 February | 60 | 8 | 68 |
2022 January | 71 | 10 | 81 |
2021 December | 53 | 11 | 64 |
2021 November | 47 | 19 | 66 |
2021 October | 34 | 13 | 47 |
2021 September | 0 | 6 | 6 |
2021 August | 0 | 5 | 5 |
2021 July | 0 | 6 | 6 |