Scientific letter
Paraneoplastic myopathy: Diagnostic value of muscle MRI
Miopatía paraneoplásica: Utilidad de la resonancia muscular
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array:21 [ "pii" => "S2667049622000333" "issn" => "26670496" "doi" => "10.1016/j.neurop.2022.05.002" "estado" => "S300" "fechaPublicacion" => "2022-07-01" "aid" => "89" "copyright" => "Sociedad Española de Neurología" "copyrightAnyo" => "2022" "documento" => "article" "crossmark" => 1 "subdocumento" => "crp" "abierto" => array:3 [ "ES" => false "ES2" => false "LATM" => false ] "gratuito" => false "lecturas" => array:1 [ "total" => 0 ] "itemAnterior" => array:17 [ "pii" => "S2667049622000321" "issn" => "26670496" "doi" => "10.1016/j.neurop.2022.03.004" "estado" => "S300" "fechaPublicacion" => "2022-07-01" "aid" => "88" "copyright" => "Sociedad Española de Neurología" "documento" => "article" "crossmark" => 1 "subdocumento" => "crp" "abierto" => array:3 [ "ES" => false "ES2" => false "LATM" => false ] "gratuito" => false "lecturas" => array:1 [ "total" => 0 ] "en" => array:11 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Scientific letter</span>" "titulo" => "Asymptomatic cystic dilatation of a perivascular space after embolisation of a middle cerebral artery aneurysm: A case report" "tienePdf" => "en" "tieneTextoCompleto" => "en" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "187" "paginaFinal" => "189" ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Dilatación quística perivascular asintomática tras la embolización de un aneurisma de arteria cerebral media: A propósito de un caso" ] ] "contieneTextoCompleto" => array:1 [ "en" => true ] "contienePdf" => array:1 [ "en" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "f0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1414 "Ancho" => 1713 "Tamanyo" => 215195 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "al0005" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="sp0005" class="elsevierStyleSimplePara elsevierViewall">Changes in the size of the cystic lesion on T1- (1A, 1C, 1E) and T2-weighted MRI sequences (1B, 1D, 1F) from the time of its diagnosis in 2015 (1A and 1B) to its maximum size in 2017 (1C and 1D) and subsequent reduction in size in 2019 (1E and 1F).</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "J. Martinez-Fernandez, A.J. Mosqueira, M. Blanco-Ulla, J.M. Pumar" "autores" => array:4 [ 0 => array:2 [ "nombre" => "J." "apellidos" => "Martinez-Fernandez" ] 1 => array:2 [ "nombre" => "A.J." "apellidos" => "Mosqueira" ] 2 => array:2 [ "nombre" => "M." "apellidos" => "Blanco-Ulla" ] 3 => array:2 [ "nombre" => "J.M." "apellidos" => "Pumar" ] ] ] ] ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2667049622000321?idApp=UINPBA00004N" "url" => "/26670496/0000000200000003/v2_202312201929/S2667049622000321/v2_202312201929/en/main.assets" ] "en" => array:17 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Scientific letter</span>" "titulo" => "Paraneoplastic myopathy: Diagnostic value of muscle MRI" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "190" "paginaFinal" => "192" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "R. Cuenca Hernández, B. Obispo Portero, J. Herreros Rodríguez, L. Fernandez Vila" "autores" => array:4 [ 0 => array:4 [ "nombre" => "R." "apellidos" => "Cuenca Hernández" "email" => array:2 [ 0 => "rcuencah@gmail.com" 1 => "raquel.cuenca@salud.madrid.org" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "af0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cr0005" ] ] ] 1 => array:3 [ "nombre" => "B." "apellidos" => "Obispo Portero" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "af0010" ] ] ] 2 => array:3 [ "nombre" => "J." "apellidos" => "Herreros Rodríguez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "af0005" ] ] ] 3 => array:3 [ "nombre" => "L." "apellidos" => "Fernandez Vila" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "af0015" ] ] ] ] "afiliaciones" => array:3 [ 0 => array:3 [ "entidad" => "Sección de Neurología, Hospital Infanta Leonor, Madrid, Spain" "etiqueta" => "a" "identificador" => "af0005" ] 1 => array:3 [ "entidad" => "Sección de Oncología, Hospital Infanta Leonor, Madrid, Spain" "etiqueta" => "b" "identificador" => "af0010" ] 2 => array:3 [ "entidad" => "Sección de Radiología, Hospital Infanta Leonor, Madrid, Spain" "etiqueta" => "c" "identificador" => "af0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cr0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "en" => array:1 [ "titulo" => "Miopatía paraneoplásica: Utilidad de la resonancia muscular" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "f0010" "etiqueta" => "Fig. 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 627 "Ancho" => 969 "Tamanyo" => 35339 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "al0010" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="sp0010" class="elsevierStyleSimplePara elsevierViewall">MRI scan of the thighs. T1-weighted sequence, axial plane. The image does not reveal muscle atrophy or fatty infiltration in the affected compartments, which suggests an acute/subacute course.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="s0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="st0005">Introduction</span><p id="p0005" class="elsevierStylePara elsevierViewall">Paraneoplastic myopathies are widely described in the literature; several immune and pathological patterns may predict their association with tumours.<a class="elsevierStyleCrossRef" href="#bb0005"><span class="elsevierStyleSup">1</span></a></p><p id="p0010" class="elsevierStylePara elsevierViewall">Muscle MRI is increasingly used in the context of neuromuscular diseases; this tool is helpful in the diagnosis of immune-mediated myopathies (IMM) due to its high sensitivity.<a class="elsevierStyleCrossRef" href="#bb0010"><span class="elsevierStyleSup">2</span></a><span class="elsevierStyleSup">,</span><a class="elsevierStyleCrossRef" href="#bb0015"><span class="elsevierStyleSup">3</span></a> Few studies have confirmed its usefulness in patients testing negative for biomarkers of IMM.</p><p id="p0015" class="elsevierStylePara elsevierViewall">We present the case of a patient with aggressive myopathy associated with breast cancer, in whom neither creatine phosphokinase (CPK) levels nor electromyography (EMG), muscle biopsy, or antibody test results supported diagnosis; muscle MRI was the most sensitive tool in this case.</p></span><span id="s0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="st0010">Clinical case</span><p id="p0020" class="elsevierStylePara elsevierViewall">Our patient was a 61-year-old woman with no relevant medical history. She visited our hospital's emergency department due to a 2-month history of asthenia and anorexia. In the previous 10 days, she had also experienced difficulty climbing stairs, combing her hair, and swallowing. She was admitted to hospital. The examination revealed symmetric limb-girdle weakness, inability to stand from a seated position and to walk, and hypoactive reflexes. A laboratory analysis revealed anaemia, mild lymphocytic leukocytosis, and an erythrocyte filtration rate of 49 mm/h. A biochemical study detected dissociated cholestasis, a CPK level of 16 U/L, elevated levels of acute-phase reactants, and a carcinoembryonic antigen level of 170.2 ng/mL. An EMG of the proximal muscles detected no signs of myopathy. <a class="elsevierStyleCrossRefs" href="#f0005">Figs. 1 and 2</a> show results from the MRI study of the thigh muscles. The patient tested negative for antibodies against Mi-2, TIF1-γ, MDA5, NXP2, SAE1, SAE2, Jo-1, PL-7, PL-12, EJ, HMGCoAR, and SRP. A whole-body CT scan revealed right breast cancer; axillary, paratracheal, and hilar adenopathy; lymphangitis carcinomatosa; peritoneal carcinomatosis; and bone metastases. A breast biopsy identified invasive ductal carcinoma with positivity for hormonal receptors. A quadriceps muscle biopsy revealed no signs of necrosis or inflammation, with mild atrophy of type II muscle fibres.</p><elsevierMultimedia ident="f0005"></elsevierMultimedia><elsevierMultimedia ident="f0010"></elsevierMultimedia><p id="p0025" class="elsevierStylePara elsevierViewall">Based on the clinical and imaging findings, the patient was diagnosed with paraneoplastic myositis, and received bolus steroid therapy, followed by oral steroids and immunoglobulins, combined with aromatase inhibitors. She displayed excellent progression at 2 months, with 4<span class="elsevierStyleHsp" style=""></span>+/5 strength in the proximal muscles, and was able to walk independently, hop on one leg, and get up from the floor. Carcinoembryonic antigen and acute-phase reactant levels also decreased.</p></span><span id="s0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="st0015">Discussion</span><p id="p0030" class="elsevierStylePara elsevierViewall">The subtypes of IMM associated with cancer include processes that are increasingly well-categorised from an immunological, anatomopathological, and clinical viewpoint. Some examples include dermatomyositis associated with anti-TIF1-γ or anti-NXP2 antibodies<a class="elsevierStyleCrossRef" href="#bb0005"><span class="elsevierStyleSup">1</span></a> and necrotising myopathies, with rapidly progressive, severe limb-girdle weakness.<a class="elsevierStyleCrossRef" href="#bb0005"><span class="elsevierStyleSup">1</span></a><span class="elsevierStyleSup">,</span><a class="elsevierStyleCrossRef" href="#bb0020"><span class="elsevierStyleSup">4</span></a></p><p id="p0035" class="elsevierStylePara elsevierViewall">We present the case of a woman with aggressive limb-girdle weakness coinciding with a diagnosis of extensive breast cancer. Clinical signs of myopathy were not supported by complementary test results: CPK levels were within the normal range, the EMG study detected no alterations, the patient tested negative for specific antibodies, and a biopsy detected no inflammation or necrosis. Muscle MRI, however, revealed muscle inflammation.</p><p id="p0040" class="elsevierStylePara elsevierViewall">Malartre et al.<a class="elsevierStyleCrossRef" href="#bb0010"><span class="elsevierStyleSup">2</span></a> support the use of muscle MRI in the diagnostic workup of IMM, to supplement the information provided by clinical signs, CPK levels, and presence of specific antibodies. The authors describe the technique as a non-invasive, safe, highly sensitive tool enabling morphological analysis and characterisation of the muscle, whose results are immediately available. In a cohort of 36 patients with IMM, Ukichi et al.<a class="elsevierStyleCrossRef" href="#bb0025"><span class="elsevierStyleSup">5</span></a> proposed performing MRI studies of the thigh muscles.</p><p id="p0045" class="elsevierStylePara elsevierViewall">Wasserman et al.<a class="elsevierStyleCrossRef" href="#bb0015"><span class="elsevierStyleSup">3</span></a> used STIR and proton density fat saturation sequences on the axial and coronal planes, as well as T1-weighted sequences. STIR and DIXON sequences are used to detect oedema, while T1-weighted sequences display muscle atrophy and fatty degeneration.<a class="elsevierStyleCrossRef" href="#bb0010"><span class="elsevierStyleSup">2</span></a><span class="elsevierStyleSup">,</span><a class="elsevierStyleCrossRef" href="#bb0015"><span class="elsevierStyleSup">3</span></a><span class="elsevierStyleSup">,</span><a class="elsevierStyleCrossRef" href="#bb0025"><span class="elsevierStyleSup">5</span></a></p><p id="p0050" class="elsevierStylePara elsevierViewall">In the case presented here, a thigh MRI scan yielded conclusive results, and treatment was immediately started. The images revealed active inflammation without fatty infiltration.</p><p id="p0055" class="elsevierStylePara elsevierViewall">Ukichi et al.<a class="elsevierStyleCrossRef" href="#bb0025"><span class="elsevierStyleSup">5</span></a> describe a distinctive MRI finding of muscular dystrophy: signal hyperintensity in the fascia, peripheral areas of the muscle, and subcutaneous adipose tissue (the latter is not observed in such other IMM as polymyositis), with a typical honeycomb pattern.</p><p id="p0060" class="elsevierStylePara elsevierViewall">In our patient, MRI showed a typical pattern of dermatomyositis,<a class="elsevierStyleCrossRef" href="#bb0010"><span class="elsevierStyleSup">2</span></a><span class="elsevierStyleSup">,</span><a class="elsevierStyleCrossRef" href="#bb0015"><span class="elsevierStyleSup">3</span></a> except that oedema was diffuse. The patient did not present skin lesions, probably due to the early onset of immunomodulatory treatment.</p><p id="p0065" class="elsevierStylePara elsevierViewall">Although anatomical pathology studies constitute the gold-standard for diagnosis and characterisation of IMM,<a class="elsevierStyleCrossRef" href="#bb0005"><span class="elsevierStyleSup">1</span></a> this was not the case in our patient; this further supports the diagnostic value of muscle MRI.</p></span><span id="s0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="st0020">Conclusion</span><p id="p0070" class="elsevierStylePara elsevierViewall">Muscle MRI supports the clinical diagnosis of IMM, providing immediate results while determining the stage of the disease. We support the routine use of this technique in the diagnostic workup of neuromuscular disorders, especially in patients with negative biopsy results.</p></span><span id="s0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="st0025">Data protection</span><p id="p0075" class="elsevierStylePara elsevierViewall">The authors observed their centre's protocols for the publication of patient data. The patient gave informed consent for the publication of this case report.</p></span><span id="s0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="st0030">Conflicts of interest</span><p id="p0080" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare.</p></span><span id="s0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="st0035">Financiation</span><p id="p0085" class="elsevierStylePara elsevierViewall">None.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:8 [ 0 => array:2 [ "identificador" => "s0005" "titulo" => "Introduction" ] 1 => array:2 [ "identificador" => "s0010" "titulo" => "Clinical case" ] 2 => array:2 [ "identificador" => "s0015" "titulo" => "Discussion" ] 3 => array:2 [ "identificador" => "s0020" "titulo" => "Conclusion" ] 4 => array:2 [ "identificador" => "s0025" "titulo" => "Data protection" ] 5 => array:2 [ "identificador" => "s0030" "titulo" => "Conflicts of interest" ] 6 => array:2 [ "identificador" => "s0035" "titulo" => "Financiation" ] 7 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2022-03-30" "fechaAceptado" => "2022-05-01" "apendice" => array:1 [ 0 => array:1 [ "seccion" => array:1 [ 0 => array:4 [ "apendice" => "<p id="p0090" class="elsevierStylePara elsevierViewall"><elsevierMultimedia ident="ec0005"></elsevierMultimedia></p>" "etiqueta" => "Appendix A" "titulo" => "Supplementary data" "identificador" => "s0040" ] ] ] ] "multimedia" => array:3 [ 0 => array:8 [ "identificador" => "f0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 707 "Ancho" => 969 "Tamanyo" => 35806 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "al0005" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="sp0005" class="elsevierStyleSimplePara elsevierViewall">MRI scan of the thighs. STIR sequence, axial plane. Bilateral, symmetric myofascial oedema (asterisks) involving the bellies of muscles in the anterior compartment of the thigh. Oedema involving the adjacent subcutaneous tissue and skin thickening (arrow).</p>" ] ] 1 => array:8 [ "identificador" => "f0010" "etiqueta" => "Fig. 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 627 "Ancho" => 969 "Tamanyo" => 35339 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "al0010" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="sp0010" class="elsevierStyleSimplePara elsevierViewall">MRI scan of the thighs. T1-weighted sequence, axial plane. The image does not reveal muscle atrophy or fatty infiltration in the affected compartments, which suggests an acute/subacute course.</p>" ] ] 2 => array:7 [ "identificador" => "ec0005" "tipo" => "MULTIMEDIAECOMPONENTE" "mostrarFloat" => false "mostrarDisplay" => true "detalles" => array:1 [ 0 => array:3 [ "identificador" => "al0015" "detalle" => "Image " "rol" => "short" ] ] "Ecomponente" => array:2 [ "fichero" => "mmc1.pdf" "ficheroTamanyo" => 193382 ] "descripcion" => array:1 [ "en" => "<p id="sp0015" class="elsevierStyleSimplePara elsevierViewall">Supplementary material</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bs0005" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bb0005" "etiqueta" => "1." "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Current classification and management of inflammatory myopathies" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:1 [ 0 => "J. Schmidt" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.3233/jnd-180308" "Revista" => array:6 [ "tituloSerie" => "J Neuromuscul Dis" "fecha" => "2018" "volumen" => "5" "numero" => "2" "paginaInicial" => "109" "paginaFinal" => "129" ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bb0010" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "MRI and muscle imaging for idiopathic inflammatory myopathies" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "S. Malartre" 1 => "D. Bachasson" 2 => "G. Mercy" 3 => "E. Sarkis" 4 => "C. Anquetil" 5 => "O. 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Pinal-Fernandez" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.nmd.2019.09.014" "Revista" => array:7 [ "tituloSerie" => "Neuromuscul Disord" "fecha" => "Nov 2019" "volumen" => "29" "numero" => "11" "paginaInicial" => "819" "paginaFinal" => "825" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/31635909" "web" => "Medline" ] ] ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bb0025" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "MRI of skeletal muscles in patients with idiopathic inflammatory myopathies: characteristic findings and diagnostic performance in dermatomyositis" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "T. Ukichi" 1 => "K. Yoshida" 2 => "S. Matsushima" 3 => "G. Kawakami" 4 => "K. Noda" 5 => "K. Furuya" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1136/rmdopen-2018-000850" "Revista" => array:4 [ "tituloSerie" => "RMD Open" "fecha" => "2019" "volumen" => "5" "numero" => "1" ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/26670496/0000000200000003/v2_202312201929/S2667049622000333/v2_202312201929/en/main.assets" "Apartado" => array:4 [ "identificador" => "92880" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Scientific letter" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/26670496/0000000200000003/v2_202312201929/S2667049622000333/v2_202312201929/en/main.pdf?idApp=UINPBA00004N&text.app=https://www.elsevier.es/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2667049622000333?idApp=UINPBA00004N" ]
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