Letter to the Editor
Tako-tsubo syndrome in patients with ANCA vasculitis
Síndrome de tako-tsubo en pacientes con vasculitis ANCA
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"documento" => "simple-article" "crossmark" => 1 "subdocumento" => "crp" "cita" => "Med Clin. 2024;162:250-1" "abierto" => array:3 [ "ES" => false "ES2" => false "LATM" => false ] "gratuito" => false "lecturas" => array:1 [ "total" => 0 ] "en" => array:10 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Scientific letter</span>" "titulo" => "Clinical characteristics and outcomes of COVID-19/HIV co-infected patients at referral unit in Recife, Brazil" "tienePdf" => "en" "tieneTextoCompleto" => "en" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "250" "paginaFinal" => "251" ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Características clínicas y resultados de pacientes coinfectados con COVID-19/VIH en una unidad de referencia en Recife, Brasil" ] ] "contieneTextoCompleto" => array:1 [ "en" => true ] "contienePdf" => array:1 [ "en" => true ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Maria Júnia Lira e Silva, Bruno Barbosa Pacífico, Polyana Monteiro" "autores" => array:3 [ 0 => array:2 [ "nombre" => "Maria Júnia" "apellidos" => "Lira e Silva" ] 1 => array:2 [ "nombre" => "Bruno Barbosa" "apellidos" => "Pacífico" ] 2 => array:2 [ "nombre" => "Polyana" "apellidos" => "Monteiro" ] ] ] ] ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2387020624000561?idApp=UINPBA00004N" "url" => "/23870206/0000016200000005/v1_202403010948/S2387020624000561/v1_202403010948/en/main.assets" ] "en" => array:13 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Letter to the Editor</span>" "titulo" => "Tako-tsubo syndrome in patients with ANCA vasculitis" "tieneTextoCompleto" => true "saludo" => "<span class="elsevierStyleItalic">Dear Editor,</span>" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "252" "paginaFinal" => "253" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "María Martínez-Urbistondo, Sonia García-Prieto, Víctor Moreno-Torres" "autores" => array:3 [ 0 => array:4 [ "nombre" => "María" "apellidos" => "Martínez-Urbistondo" "email" => array:1 [ 0 => "mmurbistondo@gmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "Sonia" "apellidos" => "García-Prieto" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "Víctor" "apellidos" => "Moreno-Torres" "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Hospital Universitario Puerta de Hierro Majadahonda, Majadahonda, Madrid, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "UNIR Health Sciences School and Medical Center, 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" => "Síndrome de tako-tsubo en pacientes con vasculitis ANCA" ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Tako-tsubo syndrome is a transient cardiac dysfunction whofse association with systemic autoimmune diseases is rare. We report 2 cases of vasculitis associated with MPO anti-neutrophil cytoplasmic antibodies (ANCA) in this context.</p><p id="par0010" class="elsevierStylePara elsevierViewall">First, a 67-year-old woman diagnosed with high blood pressure. The patient went to the emergency department complaining of a 2-month history of fever and weight loss. On physical examination, she was haemodynamically stable and had bilateral pitting oedema. The baseline electrocardiogram (ECG) and chest X-ray were unremarkable. Laboratory tests showed creatinine 2.15<span class="elsevierStyleHsp" style=""></span>mg/dl, urea 104<span class="elsevierStyleHsp" style=""></span>mg/dl, C-reactive protein 80.1<span class="elsevierStyleHsp" style=""></span>mg/l, microcytic anaemia (haemoglobin 6.2<span class="elsevierStyleHsp" style=""></span>mg/dl; mean corpuscular volume [MCV] 72.4<span class="elsevierStyleHsp" style=""></span>fl; 400. 000 reticulocytes/μl; 763,000 platelets/μl and complement consumption (C3 82<span class="elsevierStyleHsp" style=""></span>mg/dl, C4 25<span class="elsevierStyleHsp" style=""></span>mg/dl). Prior to admission, 2 units of packed red blood cells were transfused.</p><p id="par0015" class="elsevierStylePara elsevierViewall">After 36<span class="elsevierStyleHsp" style=""></span>h, she developed acute dyspnoea, desaturation and oppressive chest pain. The ECG documented T-wave inversion in the inferior and anterolateral aspect, and the echocardiogram detected a left ventricular ejection fraction (LVEF) of 30%, with septal, mid anterior and apical akinesia, as well as hypercontractility of the basal segments and B-lines in all lung fields. Troponin levels were 673<span class="elsevierStyleHsp" style=""></span>ng/l. Diuretic treatment was started with good response and catheterisation ruled out lesions. An MRI scan was not possible due to the clinical situation.</p><p id="par0020" class="elsevierStylePara elsevierViewall">The MPO-specific ANCA test was positive (127<span class="elsevierStyleHsp" style=""></span>U; reference value<span class="elsevierStyleHsp" style=""></span>><span class="elsevierStyleHsp" style=""></span>5<span class="elsevierStyleHsp" style=""></span>U) and a renal biopsy was performed, compatible with pauci-immune extracapillary proliferative glomerulonephritis. She received treatment with glucocorticoids in combination with rituximab, with a favourable clinical, laboratory and echocardiographic outcome.</p><p id="par0025" class="elsevierStylePara elsevierViewall">The second case was a 72-year-old woman with previous diagnoses of hypercholesterolemia and primary hypothyroidism. She came to the emergency department with a 2-month history of fever, despite antibiotic treatment, and a weight loss of 5<span class="elsevierStyleHsp" style=""></span>kg.</p><p id="par0030" class="elsevierStylePara elsevierViewall">On physical examination, vital signs were normal and bilateral dry crackles were heard. Laboratory tests showed a C-reactive protein >250<span class="elsevierStyleHsp" style=""></span>mg/l, a glomerular sedimentation rate of 94<span class="elsevierStyleHsp" style=""></span>mm and a leucocyte count of 20,430/μl with 17,600/μl neutrophils, and bibasilar interstitial infiltrates on radiological examination. She was admitted with broad-spectrum antibiotic treatment, persisted febrile without microbiological isolations and developed acute renal failure (maximum creatinine of 2.28<span class="elsevierStyleHsp" style=""></span>mg/dl), normocytic anaemia (haemoglobin of 7.9<span class="elsevierStyleHsp" style=""></span>mg/dl, MCV of 84.3<span class="elsevierStyleHsp" style=""></span>fl) and progressive thrombocytosis (maximum of 832,000/μl). The laboratory study showed positive MPO-ANCA results at >134<span class="elsevierStyleHsp" style=""></span>U titres. In addition, chest CT scan showed areas of ground glass over a fibrosing pattern, bronchoalveolar lavage showed inflammatory features (predominantly macrophages) and positron emission tomography showed extensive bilateral pulmonary and renal pathological uptake. Renal biopsy revealed evidence of necrotising vasculitis with glomerular involvement and tubulointerstitial nephritis. She was initially treated with glucocorticoids. Rituximab was later added.</p><p id="par0035" class="elsevierStylePara elsevierViewall">One month later, she presented with a sudden onset of dyspnoea with radiological findings suggestive of acute pulmonary oedema. The echocardiogram showed acute mitral regurgitation and severe systolic dysfunction (LVEF 31%), compatible with reverse tako-tsubo syndrome, with no abnormalities on catheterisation. The maximum troponin levels achieved were 701<span class="elsevierStyleHsp" style=""></span>ng/l. Haemodynamic instability, as well as possible renal toxicity, prevented the performance of an MRI. She required diuretic treatment, with clinical and echocardiographic resolution.</p><p id="par0040" class="elsevierStylePara elsevierViewall">The most common cardiac complications of ANCA vasculitis are coronary arteritis, pericarditis and arrhythmias,<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> the vast majority of which have a poor short-term prognosis.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> However, its association with tako-tsubo syndrome has previously been described in only 3 cases<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,4</span></a> (according to a literature search including all studies registered in the PubMed platform under the key terms "ANCA vasculitis" and "tako-tsubo"), all in women over 65 years of age and with elevated MPO-ANCA titres, following immunosuppressive treatment in the context of a disease flare. Several pathophysiological hypotheses have been proposed: the anabolic steroid effect itself, the presence of severe organ damage at another level or coronary microvascular inflammation.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> The broad differential diagnosis in ANCA vasculitis may explain some under-diagnosis. Its early detection in patients with risk factors would imply an early establishment of therapeutic measures and a better prognosis.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Ethical considerations</span><p id="par0045" class="elsevierStylePara elsevierViewall">All patients included provided informed consent.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Funding</span><p id="par0050" class="elsevierStylePara elsevierViewall">No funding was required for the preparation of this publication.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflict of interest</span><p id="par0055" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflict of interest in connection with this letter to the editor.</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 "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" => "Cardiac involvement in the adult primary vasculitides" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "G. 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