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Scientific Letter
Severe Eosinophilic Asthma and Severe Mixed Polyneuropathy: A Case of Eosinophilic Granulomatous Vasculitis With Polyangiitis and New Therapeutic Perspectives
Asma eosinofílica grave y polineuropatía mixta severa. Un nuevo caso de vasculitis granulomatosa eosinofílica con poliangeítis (GEPA). Nuevas perspectivas terapéuticas
María del Carmen Lorenzo Martíneza, Ángela Hidalgo Herranza, Ana María Ochoa Ruiza, Bárbara Gutierrez Ruanob, Raúl Ruiz Estebanc, Sergio Campos Télleza, José Javier Jareño Estebana,
Corresponding author
jjjarenoesteban@yahoo.es

Corresponding author.
a Respiratory Medicine Department, Hospital Central de la Defensa Gómez Ulla, Madrid, Spain
b Neurology Department, Hospital Central de la Defensa Gómez Ulla, Madrid, Spain
c Internal Medicine Department, Hospital Central de la Defensa Gómez Ulla, Madrid, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">We report the case of a 46-year-old woman&#44; non-smoker with a history of severe uncontrolled asthma&#46; Since the age of 30&#44; she has presented symptoms of arthralgia and myalgia&#44; and 2 years ago she developed distal paraesthesia&#46; Physical examination revealed diffuse bilateral wheezing on pulmonary auscultation&#46; Neurological findings included right-sided peripheral hypoesthesia&#46; Laboratory values of note included haemoglobin 11&#46;3<span class="elsevierStyleHsp" style=""></span>g&#47;dl&#44; haematocrit 37&#46;2&#37;&#44; and eosinophils 485 &#40;6&#46;9&#37;&#41;&#46; Kidney function&#44; ionogram and thyroid hormones were normal&#46; Antineutrophil cytoplasmic antibody &#40;ANCA&#41; testing&#44; an immunological study for connective tissue diseases and serologies were all negative&#46; Chest computed tomography showed pulmonary opacities in the left upper lobe&#46; Sural nerve biopsy led to a diagnosis of eosinophilic granulomatosis with polyangiitis &#40;EGPA&#41;&#46; The Birmingham activity score was 10 points&#46; Treatment began with corticosteroids and cyclophosphamide that&#44; due to adverse effects&#44; were switched to azathioprine&#46; At follow-up&#44; paraesthesia had progressed so we changed treatment to rituximab and continued with prednisone&#46; In the following months&#44; worsening asthma control &#40;Asthma Control Test<span class="elsevierStyleHsp" style=""></span>&#60;<span class="elsevierStyleHsp" style=""></span>15&#44; reduced forced expiratory volume in 1 second&#44; elevated fractional exhaled nitric oxide&#41; and progression of neurological symptoms were observed&#46; In view of this situation&#44; inhaled therapy and systemic corticosteroids were optimised&#46; We also started mepolizumab at 100<span class="elsevierStyleHsp" style=""></span>mg that was progressively increased to 300<span class="elsevierStyleHsp" style=""></span>mg to achieve greater control of EGPA activity&#44; asthma&#44; and improve pulmonary function and neurological symptoms &#40;<a class="elsevierStyleCrossRef" href="#tbl0005">Table 1</a>&#41;&#46; Subsequently&#44; we were able to reduce corticosteroid dosage&#46;</p><elsevierMultimedia ident="tbl0005"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">EGPA is characterised by eosinophilic tissue infiltration associated with vasculitis and the presence of granulomas&#46; Clinical manifestations are varied&#44; and diagnostic criteria include asthma&#44; rhinosinusitis&#44; peripheral blood eosinophilia&#44; extravascular eosinophilia&#44; evanescent pulmonary infiltrates&#44; and peripheral neuropathy&#46; Only 40&#37; of patients are ANCA-positive&#46; The Birmingham activity score &#40;&#8805;4&#41; is currently used to assess the degree of activity and remission&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> The survival of patients with vasculitis has increased in recent decades due to the use of immunomodulators and corticosteroids&#44; but these treatments are not exempt from toxicity or risk of infections&#46; In recent years&#44; new treatments such as rituximab and mepolizumab have appeared and have demonstrated their efficacy in inducing remission&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">Rituximab is an anti-CD20 monoclonal antibody that depletes B-lymphocytes and has shown efficacy in microscopic polyangiitis &#40;MPA&#41; and granulomatosis with polyangiitis &#40;GPA&#41;&#46; Experience in EGPA is scant but growing&#44; with the recent publication of several series in EGPA patients in which rituximab demonstrated efficacy in controlling disease activity and possibly reducing maintenance doses of corticosteroids&#46;<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">2&#44;3</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">In our patient&#44; refractory vasculitis and mononeuritis multiplex improved partially with rituximab treatment&#46; However&#44; persistent neurological symptoms and poor asthma control prompted the initiation of mepolizumab associated with corticosteroids as an alternative to other treatments &#40;methotrexate&#44; azathioprine and mycophenolate mofetil&#44; etc&#46;&#41;&#46; Recent multicentre studies in Europe have confirmed the efficacy and safety of this combination&#46; It is well tolerated and is associated few adverse effects and a low risk of infections&#44; allowing a progressive reduction of the corticosteroid dose&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a> Several European studies show that a 100<span class="elsevierStyleHsp" style=""></span>mg dose of mepolizumab is as effective as the 300<span class="elsevierStyleHsp" style=""></span>mg dose recommended by the US Food and Drug Administration&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a></p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Informed consent</span><p id="par0025" class="elsevierStylePara elsevierViewall">The authors have obtained the informed consent of the patient and&#47;or subjects referred to in the article&#46; This document is held by the corresponding author&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Funding</span><p id="par0030" class="elsevierStylePara elsevierViewall">No external financing was needed for this study&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Authors&#8217; contributions</span><p id="par0035" class="elsevierStylePara elsevierViewall">All authors have contributed substantially to each of the following aspects&#58; &#40;1&#41; data acquisition&#44; data analysis and interpretation&#44; &#40;2&#41; drafting of the article&#44; and &#40;3&#41; final approval of the version presented&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Conflicts of interests</span><p id="par0040" class="elsevierStylePara elsevierViewall">The authors state that they have no conflict of interests&#46;</p></span></span>"
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                  \t\t\t\t">6 per year&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t">0&#46;42&nbsp;\t\t\t\t\t\t\n
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Article information
ISSN: 26596636
Original language: English
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