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the use of which is standard in other connective tissue disorders&#46;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> A series of published cases of NAM exist which were treated with rituximab&#44; with varying clinical responses&#46;<a class="elsevierStyleCrossRefs" href="#bib0080"><span class="elsevierStyleSup">6&#8211;10</span></a></p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Clinical Case</span><p id="par0020" class="elsevierStylePara elsevierViewall">A female aged 30 was diagnosed with NAM as a result of the presence of proximal myopathy&#44; dysphagia to liquids and dysphonia of 2-month onset&#44; elevation of muscular enzymes &#40;CK 5&#46;922<span class="elsevierStyleHsp" style=""></span>UI&#47;l&#44; aldolase 84<span class="elsevierStyleHsp" style=""></span>UI&#47;l&#41;&#44; electromyography with myopathic pattern&#44; signs of intramuscular oedema on the magnetic resonance in shoulder and pelvic girdles &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>a&#41; and anatomical&#8211;pathological findings of necrotic myopathy which had defied standard treatment &#40;corticoids&#44; inmunoglobulins&#44; azathioprine&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">As a result of histological findings&#44; anti-SRP antibodies were requested which resulted positive &#40;IFI 1&#58;1&#46;280 technique&#41;&#46; Treatment was initiated with methotrexate &#40;up to 30<span class="elsevierStyleHsp" style=""></span>mg per week&#41;&#46; Clinical symptoms improved after 3 months with the exception of the iliacus-psoas musculature &#40;4&#47;5&#41;&#46; Progressive reduction of corticoids resulted in clinical and analytical deterioration&#46; Concomitant treatment was subsequently started with rituximab &#40;two 1-g doses&#41;&#46; Five months later&#44; the patient&#39;s condition was improved&#46; After 6 months&#44; a new cycle of rituximab was begun to maintain therapy response&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">Magnetic resonance of the muscles after 2 years showed an absence of any signs of inflammatory activity &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>b&#41;&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">As methotrexate was lowered below 22&#46;5<span class="elsevierStyleHsp" style=""></span>mg per week&#44; the patient again began to be clinically and analytically compromised&#44; and the dose was therefore increased to regain clinical control in the last year &#40;with the exception of iliacus-psoas musculature weakness&#41;&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0040" class="elsevierStylePara elsevierViewall">To our knowledge&#44; less than 30 cases of anti-SRP NAMs treated with rituximab have been published&#46;<a class="elsevierStyleCrossRefs" href="#bib0080"><span class="elsevierStyleSup">6&#8211;10</span></a> Pinal-Fernandez et al&#46;<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a> describe 17 patients treated with rituximab&#44; which was apparently effective in 13 of them&#46; The duration of the biological effect of the rituximab was variable &#40;from 6 to 9 months to over more than 2 years&#41;&#46; In our patient&#44; it has not been administered on more occasions since mid-2012&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">Two refractory cases were also published which improved with the combination of corticoids&#44; plasmapheresis and rituximab&#44;<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a> as well as another two cases treated with rituximab with favourable outcomes&#46;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">However&#44; a British series<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a> of two patients treated with rituximab presented a poor clinical response&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">In our case&#44; two cycles of rituximab were administered&#44; leading to an exacerbation on reduction of the dose of methotexate&#44; with favourable subsequent control when the dose was increased&#46; The CK values of the patient were lower than that published in other series and this may be one of the reasons why a good therapeutic outcome was achieved&#46; The imaging findings were also of note&#44; where greater compromise of the posterior thigh compartment was noted when there were no apparent clinical signs of this&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conclusion</span><p id="par0060" class="elsevierStylePara elsevierViewall">NAMs should be suspected in patients with atypical findings&#44; of MII&#44; both histopathologically &#40;presence of necrotic muscular fibres&#44; near absence of inflammatory cell infiltrate&#41; and clinically &#40;poor response to immunosuppressants&#41;&#46;</p><p id="par0065" class="elsevierStylePara elsevierViewall">Rituximab could be a good therapeutic option for those cases where standard treatment has failed&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Ethical Liabilities</span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Protection of people and animals</span><p id="par0070" class="elsevierStylePara elsevierViewall">The authors declare that no experiments using human beings or animals have been carried out for this research study&#46;</p></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0055">Data confidentiality</span><p id="par0075" class="elsevierStylePara elsevierViewall">The authors declare that no patient data appear in this article&#46;</p></span><span id="sec0040" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0060">Right to privacy and informed consent</span><p id="par0080" class="elsevierStylePara elsevierViewall">The authors declare that no patient data appear in this article&#46;</p></span></span><span id="sec0045" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0065">Conflicts of Interest</span><p id="par0085" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare&#46;</p></span></span>"
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    "fechaRecibido" => "2016-09-10"
    "fechaAceptado" => "2017-02-24"
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            0 => "Necrotizing autoimmune myopathy"
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          "clase" => "keyword"
          "titulo" => "Palabras clave"
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          "palabras" => array:3 [
            0 => "Miopat&#237;as necrosantes autoinmunes"
            1 => "Part&#237;cula de reconocimiento de la se&#241;al"
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        "titulo" => "Abstract"
        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Necrotizing autoimmune myopathy &#40;NAM&#41; is a rare and emerging entity of idiopathic inflammatory myopathy &#40;IIM&#41;&#46;</p><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">They have been associated with connective tissue disorders&#44; viral infections&#44; malignancy&#44; anti-signal recognition particle &#40;SRP&#41; and anti-3-hydroxy-3-methylglutaryl-coenzyme A reductase protein &#40;with or without the use of statins&#41;&#46;</p><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Anti-SRP-associated NAM has different clinical and histological characteristics that differentiate them from other IIMs&#44; resulting in a poor prognosis&#46; Very few cases treated with rituximab have been published&#44; with varying clinical response&#46;</p><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Here we describe a case of anti-SRP-associated NAM refractory to conventional immunosuppressants and its successful long-term management with the combination of rituximab&#44; corticosteroids and methotrexate&#46;</p></span>"
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        "titulo" => "Resumen"
        "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Las miopat&#237;as necrosantes autoinmunes &#40;NAM&#41; son unas entidades raras y emergentes de las miopat&#237;as inflamatorias idiop&#225;ticas &#40;MII&#41;&#46;</p><p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">Su presencia se ha asociado a conectivopat&#237;as&#44; infecciones virales&#44; neoplasias&#44; anticuerpos frente a la part&#237;cula de reconocimiento de la se&#241;al &#40;anti-SRP&#41; y a anticuerpos frente a la prote&#237;na 3-hidroxi-3-metilglutaril-coenzima A reductasa &#40;HMG-CR&#41; &#40;asociada al empleo o no de estatinas&#41;&#46;</p><p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">Las NAM asociadas al anti-SRP presentan unas caracter&#237;sticas cl&#237;nicas y anatomopatol&#243;gicas que las diferencian del resto de las MII&#44; resultando en un peor pron&#243;stico&#46; Hasta el momento han sido muy pocos los casos publicados tratados con rituximab y con diferente respuesta cl&#237;nica&#46;</p><p id="spar0040" class="elsevierStyleSimplePara elsevierViewall">Presentamos el caso de una mujer de 30 a&#241;os con NAM asociada al anti-SRP refractaria a tratamiento convencional inmunosupresor y que ha sido controlada a largo plazo con la combinaci&#243;n de rituximab&#44; metotrexato y corticoides&#46;</p></span>"
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      0 => array:2 [
        "etiqueta" => "&#9734;"
        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; N&#243;voa Medina FJ&#44; Guti&#233;rrez Mart&#237;nez J&#44; Gonz&#225;lez Gonz&#225;lez Y&#44; Romero D&#237;az B&#44; Mach&#237;n Garc&#237;a S&#44; Rosas Romero A&#46; Tratamiento con rituximab para las miopat&#237;as necrosantes autoinmunes asociadas al anti-SRP&#58; caso cl&#237;nico y revisi&#243;n de la evidencia&#46; Reumatol Clin&#46; 2018&#59;14&#58;379&#8211;381&#46;</p>"
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          "en" => "<p id="spar0045" class="elsevierStyleSimplePara elsevierViewall">&#40;a&#41; Baseline MRI &#40;T1 sequence&#41;&#46; There is minimal oedema in the distal end of both the sartorius and gracilis muscles&#46; Compromise of both compartments&#44; with the lateral mass more affected in the anterior compartment and the middle masses more affected in the posterior compartment&#46; In general&#44; there is greater posterior compromise &#40;arrow&#41;&#46; &#40;b&#41; MRI after treatment with rituximab &#40;T1 sequence&#41;&#46; Bilateral and symmetrical compromise is noted of both compartments with muscular atrophy observed with diffuse fatty deposits&#46; The lateral and middle masses are more affected in the anterior compartment and the hamstring muscles are more affected in the posterior compartment&#44; with predominance of compromise in the posterior compartment &#40;arrow&#41;&#46;</p>"
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                            0 => "P&#46;J&#46; Utz"
                            1 => "M&#46; Hottelet"
                            2 => "T&#46;M&#46; Le"
                            3 => "S&#46;J&#46; Kim"
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                      "titulo" => "Inhibitors of MAPK pathway ERK1&#47;2 or p38 prevent the IL-1&#946;-induced Up-regulation of SRP72 autoantigen in Jurkat cells"
                      "autores" => array:1 [
                        0 => array:2 [
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                            0 => "V&#46;E&#46; Arana-Arg&#225;ez"
                            1 => "V&#46; Delgado-Rizo"
                            2 => "O&#46;E&#46; Pizano-Mart&#237;nez"
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                          ]
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                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Rituximab in autoimmune diseases"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
                          "autores" => array:1 [
                            0 => "K&#46;L&#46; Randall"
                          ]
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              "etiqueta" => "6"
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                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Rituximab therapy for myopathy associated with anti-signal recognition particle antibodies&#58; a case series"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
                          "autores" => array:5 [
                            0 => "R&#46; Valiyil"
                            1 => "L&#46; Casciola-Rosen"
                            2 => "G&#46; Hong"
                            3 => "A&#46; Mammen"
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                          ]
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                  "contribucion" => array:1 [
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                      "titulo" => "Longitudinal course of disease in a large cohort of myositis patients with autoantibodies recognizing the signal recognition particle"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => true
                          "autores" => array:6 [
                            0 => "I&#46; Pinal-Fernandez"
                            1 => "C&#46; Parks"
                            2 => "J&#46;L&#46; Werner"
                            3 => "J&#46; Akbayda"
                            4 => "J&#46; Paik"
                            5 => "S&#46;K&#46; Danoff"
                          ]
                        ]
                      ]
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                  ]
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                    0 => array:1 [
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              "etiqueta" => "8"
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                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Marked efficacy of a therapeutic strategy associating prednisone and plasma exchange followed by rituximab in two patients with refractory myopathy associated with antibodies to the signal recognition particle &#40;SRP&#41;"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => true
                          "autores" => array:6 [
                            0 => "J&#46;B&#46; Arlet"
                            1 => "D&#46; Dimitri"
                            2 => "C&#46; Pagnoux"
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                          ]
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                          ]
                        ]
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                  ]
                ]
              ]
            ]
            8 => array:3 [
              "identificador" => "bib0095"
              "etiqueta" => "9"
              "referencia" => array:1 [
                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Rituximab therapy for myopathy associated with anti-signal recognition particle antibodies&#58; a case series"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => true
                          "autores" => array:6 [
                            0 => "C&#46; Deligny"
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