Letter to the Editor
Hemorrhagic myositis as a complication of cancer-associated dermatomyositis
Miositis hemorrágica como complicación de una dermatomiositis asociada a neoplasia
a Servicio de Medicina Interna, Hospital Valle del Nalón, Langreo, Asturias, Spain
b Servicio de Neurología, Hospital Universitario Central de Asturias, Oviedo, Asturias, Spain
c Servicio de Medicina Interna, Unidad de Enfermedades Autoinmunes Sistémicas, Instituto de Investigación Sanitaria del Principado de Asturias (ISPA), Hospital Universitario Central de Asturias, Oviedo, Spain
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(D and E) Marked epidermal thickening and purplish erythema of the eyelid.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Idiopathic inflammatory myopathies (IIM) may be accompanied by cutaneous, articular or pulmonary manifestations and may occur in the context of neoplasms.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Their association with specific autoantibodies has led to their being called autoimmune inflammatory myopathies.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Diagnosis is based on symptoms, electromyographic findings, elevated muscle enzymes, imaging tests and muscle biopsy. They are sometimes a diagnostic challenge, such as the patient reported here with extensive skin and muscle bruising as a form of presentation.</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 77-year-old male patient, hypertensive and diabetic, with a biological prosthetic cardiac valve and aortocoronary <span class="elsevierStyleItalic">bypass</span>, for which he was being treated with 100 mg/day of aspirin. In 2018, a multifocal BCLC-B stage liver cancer was diagnosed, probably secondary to alcoholic liver disease, and underwent transarterial chemoembolization and radiofrequency ablation, with disease control.</p><p id="par0015" class="elsevierStylePara elsevierViewall">In February 2019, while he was asymptomatic and after undergoing a liver magnetic resonance imaging (MRI) scan, where a diagnosis of recurrence of the neoplasm was made, he went to the emergency department due to oedema and redness of the face, neck, arms and upper thoracic region. A contrast-induced phototoxic rash was suspected and was treated with 20 mg/day of prednisone. Despite this, he developed pain and muscle weakness and was hospitalised. The examination showed pain in the proximal muscle masses and decreased strength in the shoulder and pelvic girdle. The most striking features were heliotrope erythema, lesions on the hands characteristic of dermatomyositis and ecchymosis on the trunk and limbs (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A–E). Laboratory tests: creatine kinase 1661 U/L (n: 20–200), troponin T 107 ng/L (n < 14), creatinine 1.34 mg/dL, with a normal blood count and basic coagulation study. Electromyogram without myopathy symptomatology, and a chest-abdominal computed tomography (CT), without pulmonary lesions or other neoplasms. ANA 1/320 speckled pattern, without specificities, with negative myositis-specific autoantibodies (anti-Jo-1, PL-7, PL-12, EJ, SRP, HMGCR, Mi-2, MDA-5, TIF1-γ, SAE, NXP-2 and SSA/Ro52). Complement, cryoglobulins and ANCA also negative. A muscle biopsy on the right biceps showed no muscle swelling. Despite this, and with the suspicion of paraneoplastic dermatomyositis (DM), treatment was started with 40 methylprednisolone mg/day and aspirin was discontinued. Progression was poor, with the development of oedema, deterioration of renal function and new spontaneous hematomas. An ultrasound and an MRI of the upper limbs identified muscle oedema, and a PET/CT scan detected an increase in carbohydrate metabolism in the upper limb muscles. A new muscle biopsy guided by imaging tests demonstrated a predominantly perimysial inflammatory myopathy with MCH-1 expression. The patient received three boluses of 125 mg methylprednisolone, immunoglobulins and prednisone 0.4 mg/kg/day in a tapering oral regimen, which reduced oedema, muscle pain and bruising, improved myopathy and restored autonomy. After the initial improvement, he suffered dysphagia, decompensation of his liver disease, and died six months later.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Our patient presented with muscle haemorrhage as a form of presentation of DM, following recurrence of his liver cancer, a previously unreported event. Cases have been described in which muscle bleeds of different characteristics occur within an inflammatory myopathy, which are defined in the literature as “haemorrhagic myositis” (HM), given the atypical and rare nature of this complication. Orrell et al.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> reported the first two cases in 1998, one of them with hematomas in the rectus muscle of the thighs in a patient with calcinosis in that area, and another in an 11-year-old girl with a retroperitoneal hematoma. In both cases, the diagnosis of DM was prior to the onset of bleeding, and they had not received anticoagulants or antiplatelet agents. Eleven cases have been described, between 1998 and 2020, which were collected by Chandler et al.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> They had a mean age of 50 years and a similar distribution by sex, in IIM already diagnosed and receiving immunosuppressive treatment. Haematomas appear in large muscle groups, such as those of the abdomen or the psoas, but also in the limbs, such as the deltoid muscles. Most received low molecular weight heparins (LMWH) or antiplatelet agents, which has been suggested to be a risk factor for this complication.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> However, at least three cases have been described where HM occurred in the absence of such treatments. Two cases had autoantibodies, anti-Ro52 and anti-Tif1γ, without correlation between their presence and the occurrence of hemorrhages.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> The pathophysiology of MH has been attributed to possible capillary vasculitis at the muscular level, and tissue fragility induced by the steroids they often receive.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">Our patient was receiving antiplatelet therapy and prophylactic LMWH, which were subsequently discontinued, with further progression of the haematomas. Treatment of HM is based on hemorrhagic control (embolization, transfusions)<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> and its prognosis is usually poor, with mortality in 50% of cases.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> The improvement with steroids and immunoglobulins that we observed could support the inflammatory origin of HMs, and the use of immunosuppressants or immunoglobulins to control bleeding (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>).</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflict of interests</span><p id="par0030" class="elsevierStylePara elsevierViewall">The authors declare no conflict of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:2 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Conflict of interests" ] 1 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Suárez-Díaz S, Riesco-Pérez NP, Caminal-Montero L. Miositis hemorrágica como complicación de una dermatomiositis asociada a neoplasia. Med Clin (Barc). 2021;157:e319–e320.</p>" ] ] "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" => 1559 "Ancho" => 2500 "Tamanyo" => 335319 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A–C) Hematomas on arms, trunk/back of arms and legs respectively. (D and E) Marked epidermal thickening and purplish erythema of the eyelid.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Classification and management of adult inflammatory myopathies" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "A. Selva-O’Callaghan" 1 => "I. Pinal-Fernández" 2 => "E. Trallero-Araguás" 3 => "J.C. Milsenda" 4 => "J.M. Grau-Junyent" 5 => "A. 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