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subclinical myocardial abnormalities are frequent and are found in more than 50&#37; of post-mortem examinations&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Symptomatic cardiomyopathy carries a poor prognosis&#44; accounting for about 50&#37; of deaths&#44; and is thus the major cause of mortality in CSS&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> It can present with eosinophilic vasculitis&#44; myocarditis&#44; pericarditis&#44; pericardial effusion&#44; fibrosis&#44; valvular heart disease&#44; conduction disorders&#44; intracavitary thrombi&#44; and cardiomyopathy&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#8211;6</span></a> Death is usually due to myocardial infarction&#44; heart failure&#44; malignant ventricular arrhythmias&#44; and&#47;or cardiac tamponade&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We describe the case of a patient with newly diagnosed CCS presenting with pronounced eosinophilic myocarditis and extensive focal vasculopathy on coronary angiography consistent with vasculitis&#46; After appropriate and timely pharmacologic therapeutic intervention the symptoms resolved completely&#44; as did the coronary lesions and myocardial infiltrates&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Case report</span><p id="par0015" class="elsevierStylePara elsevierViewall">A 22-year-old woman with a history of allergic rhinitis&#44; asthma and repeated upper respiratory tract infections in the previous two years developed progressive asthenia&#44; dizziness and left leg paresthesias&#46; One month after the beginning of these non-specific symptoms she was hospitalized because of pleuritic chest pain&#46; She had had no recent flu-like symptoms&#44; either of the upper respiratory or gastrointestinal tract&#44; or other symptoms suggestive of a previous infectious disease&#46; Physical examination was unremarkable&#59; the electrocardiogram &#40;ECG&#41; revealed poor anteroseptal R-wave progression and diffuse T-wave inversion &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Figure 1</a>A&#41;&#46; Laboratory tests showed eosinophilia &#40;3&#46;83<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>10<span class="elsevierStyleSup">9</span>&#47;l&#41;&#44; elevated erythrocyte sedimentation rate &#40;43 mm&#47;h&#41; and elevated biochemical markers of myocardial injury &#40;peak troponin I 155&#46;6 ng&#47;ml&#41;&#46; Serologic and PCR tests were negative for cardiotropic viruses&#44; <span class="elsevierStyleItalic">Aspergillus</span>&#44; <span class="elsevierStyleItalic">Toxoplasma</span>&#44; <span class="elsevierStyleItalic">Chlamydia psittaci</span> and <span class="elsevierStyleItalic">Mycoplasma pneumonia</span>&#46; Specific study for parasites was also negative&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Transthoracic echocardiography revealed a small pericardial effusion with no other abnormalities&#46; A diagnosis of myopericarditis was assumed and anti-inflammatory therapy with ibuprofen was initiated&#46; However&#44; the patient&#39;s recurrent chest pain persisted&#44; associated with dynamic ECG abnormalities &#40;transient ST-segment elevation in inferior leads&#41; &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Figure 1</a>B&#41; and new wall motion abnormalities on echocardiography &#40;inferior wall hypokinesia&#41;&#46; Given the unfavorable clinical evolution associated with a significant rise in plasma troponin I concentration and new ECG and echocardiographic abnormalities&#44; despite anti-inflammatory therapy&#44; cardiac catheterization was performed on the fourth day after admission&#46; Coronary arteriography demonstrated irregularity of the larger arteries with long diffuse stenotic lesions in the left anterior descending and right coronary arteries &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Figure 2</a>A and B&#44; arrowed&#41;&#44; consistent with diffuse vasculitis&#46; A myocardial biopsy showed eosinophilic myocarditis &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Figure 3</a>A&#41; and a concomitant nasal biopsy revealed eosinophilic necrotizing granulomatous vasculitis&#46; Immunologic study&#44; including ANCA antibodies&#44; was negative&#44; while electromyography revealed left saphenous nerve mononeuropathy&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">Churg&#8211;Strauss syndrome was then diagnosed as four out of six criteria were present in this patient&#58; &#40;1&#41; asthma&#59; &#40;2&#41; eosinophilia&#59; &#40;3&#41; mononeuropathy&#59; &#40;4&#41; extravascular eosinophils&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> Pulse intravenous &#40;i&#46;v&#46;&#41; corticosteroid treatment &#40;methylprednisolone 1 g i&#46;v&#46; daily for three days&#41; was started&#44; just after the biopsy results&#44; followed by oral glucocorticoid therapy &#40;prednisone 1&#46;5 mg&#47;kg daily&#41; associated with cyclophosphamide &#40;0&#46;6 g&#47;m<span class="elsevierStyleSup">2</span> intravenously once a month&#41;&#46; On day one of treatment&#44; the patient&#39;s symptoms improved significantly and levels of blood eosinophils fell to 0&#46;02<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>10<span class="elsevierStyleSup">9</span>&#47;l&#46; Echocardiography performed after 18 days of treatment showed resolution of the pericardial effusion as well as of the left ventricular wall motion abnormalities&#46; Control coronary angiography one year after initiation of therapy showed complete regression of coronary stenotic lesions &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Figure 2</a>C and D&#41; and a myocardial biopsy confirmed resolution of eosinophilic myocarditis &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Figure 3</a>B&#41;&#46; The patient was stable and symptom-free at one year of follow-up under chronic therapy&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0030" class="elsevierStylePara elsevierViewall">We describe the case of a patient with newly diagnosed CSS presenting with extensive myocarditis&#44; which is an unusual clinical manifestation of this disease&#46; According to the American College of Rheumatology&#44; the presence of four or more of the six possible criteria &#8211; &#40;1&#41; asthma&#59; &#40;2&#41; eosinophilia &#40;&#62;10&#37; of leukocytes by differential cell count&#41;&#59; &#40;3&#41; mononeuropathy or polyneuropathy&#59; &#40;4&#41; migratory or transient pulmonary infiltrates&#59; &#40;5&#41; paranasal sinus abnormality&#59; and &#40;6&#41; extravascular eosinophils &#8211; establishes the diagnosis with a sensitivity of 85&#37; and a specificity of 99&#37;&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> In our case&#44; a diagnosis of CSS was established based on the patient&#39;s history of asthma&#44; hypereosinophilia&#44; mononeuropathy and extravascular eosinophils on myocardial and nasal biopsy&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">This case report highlights the possibility of cardiac involvement in patients with CSS and calls attention to this differential diagnosis in the evaluation of myocarditis&#46; This diagnosis is mostly overlooked because major cardiac problems are rarely the presenting manifestations of vasculitis&#46; Involvement of the heart has been described in the third stage of the disease&#44; as observed in our patient&#46; It is usually associated with vasculitic lesions in the myocardium and coronary vessels causing &#40;peri&#41;myocarditis&#44; heart failure&#44; cardiac tamponade&#44; myocardial infarction&#44; or pericardial effusion&#46;<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">8&#8211;11</span></a> Our patient presented with pronounced myocarditis but pericardial effusion and coronary vasculitis were also evident&#46; The myocardial damage is caused by vasculitis leading to coronary arteritis and coronary occlusion&#44; through the release by activated eosinophils of toxic mediators causing direct myocardial damage&#44;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">12</span></a> or by replacement of the myocardium with granulomas and scar tissue&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">13&#44;14</span></a> Patients with cardiac involvement are mainly ANCA-negative&#44; as in this case&#46;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">15</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">In general&#44; the prognosis of CSS is good&#44; with an overall 10-year survival of 81-92&#37;&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">11&#44;16</span></a> However&#44; cardiac involvement is one of the most important predictors of an adverse outcome&#44; causing up to 50&#37; of CSS-related mortality&#46;<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">10&#44;16</span></a> In a study on 96 patients&#44;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">16</span></a> 78-month survival was 90&#37; in the absence of symptomatic cardiac manifestations&#44; compared to 30&#37; in their presence&#46; Of the patients with myocardial involvement&#44; 39&#37; died in the acute stage of the disease&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">16</span></a> Early diagnosis of cardiac involvement and subsequent adjuvant therapy may prevent progression of cardiac disease and improve prognosis in these patients&#46; Patients with acute multiorgan disease should receive intravenous glucocorticoid &#40;eg&#46; methylprednisolone 1 g daily for three days&#41; followed by oral glucocorticoid therapy&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;17</span></a> Prednisone doses of 0&#46;5-1&#46;5 mg&#47;kg per day are typically administered until disease remission is attained and then gradually tapered to the lowest dose required for control of symptoms and signs of active CSS&#46; The higher dose is used for patients with more aggressive disease&#44; including those with cardiac involvement&#46; Clinical remission of isolated pericarditis&#44; without other visceral involvement&#44; can be obtained with corticosteroid therapy alone&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">13</span></a> However&#44; in the case of myocardial involvement the addition of immunosuppressive therapy is recommended&#46; In higher-risk patients&#44; including those with myocardial injury&#44; lower mortality was noted among those who were treated with cyclophosphamide compared to a separate group in which only some patients received cyclophosphamide &#40;7&#37; vs&#46; 26&#37; mortality&#44; respectively&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">18</span></a> Cyclophosphamide may be administered orally every day or intravenously once a month&#44; but insufficient data are available on CSS to make a clear recommendation regarding this choice&#46; There is also disagreement concerning the duration of immunosuppressive therapy&#46; However&#44; in a preliminary study of patients with CSS&#44; those receiving six pulses of cyclophosphamide had more relapses than those receiving 12 pulses &#40;94&#37; vs&#46; 41&#37;&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">19</span></a> Further data are needed to clarify whether the benefits of 12 pulses outweigh the additional risks&#44; especially of bladder toxicity&#46; Our case report clearly demonstrates the possibility of complete reversal of cardiac disease with appropriate steroid and immunosuppressive therapy&#44; which is in agreement with previous studies&#46;<a class="elsevierStyleCrossRefs" href="#bib0095"><span class="elsevierStyleSup">19&#8211;22</span></a> However&#44; this vasculitis can be fulminant if not identified early and may on occasion present with cardiogenic shock with a malignant course requiring urgent transplantation&#44; despite therapy&#46;<a class="elsevierStyleCrossRefs" href="#bib0115"><span class="elsevierStyleSup">23&#44;24</span></a> Reports of heart transplantation in this setting are rare and only limited information is available on feasibility&#44; outcome or relapse after heart transplantation in patients with CSS&#46; While one case reported a good long-term result&#44;<a class="elsevierStyleCrossRef" href="#bib0115"><span class="elsevierStyleSup">23</span></a> recurrent disease after initially successful transplantation was observed in another patient&#46;<a class="elsevierStyleCrossRef" href="#bib0125"><span class="elsevierStyleSup">25</span></a> Cardiac involvement in CSS thus requires immediate therapy&#44; which may allow recovery of cardiac function and reduce the significant cardiac mortality associated with CSS&#44; underlining the need for an aggressive invasive diagnostic approach in CSS patients with heart lesions&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">Recent research with multimodality assessment&#44; including ECG analysis&#44; echocardiography and cardiac magnetic resonance imaging&#44; has also shown a high incidence of cardiac involvement &#40;62&#8211;90&#37;&#41; in patients with full clinical remission&#44; characterized not only by fibrosis&#44; but also by an active inflammatory process&#46;<a class="elsevierStyleCrossRefs" href="#bib0130"><span class="elsevierStyleSup">26&#44;27</span></a> Since cardiac involvement is one of the most important predictors of an adverse outcome&#44;<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">18</span></a> these findings could influence current recommendations for cardiac evaluation and therapeutic decisions in asymptomatic patients&#46; An early diagnosis detecting silent inflammation could be valuable&#44;<a class="elsevierStyleCrossRef" href="#bib0140"><span class="elsevierStyleSup">28</span></a> since appropriate therapy can prevent progression to harmful episodes of cardiac disease and so reduce the high mortality associated with these manifestations&#46; Systematic cardiac evaluation in asymptomatic patients with detailed imaging still lacks validation of its clinical benefit but could be an important procedure in the future&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conclusion</span><p id="par0050" class="elsevierStylePara elsevierViewall">Because of its multiple forms of presentation and multiorgan involvement&#44; diagnosis of CSS can be difficult&#46; As reported here&#44; it may present with severe cardiac disease&#46; Cardiac involvement is a poor prognostic factor&#44; and is a leading cause of mortality in CSS&#46; As shown by this case report&#44; appropriate and aggressive glucocorticoid and cyclophosphamide therapy may lead to complete recovery from potentially fatal cardiac disease&#46; Physicians should thus be alert to the possibility of CSS as a differential diagnosis in patients presenting with myocarditis&#44; whenever the clinical setting is appropriate&#46; Prompt diagnosis and therapy can change the poor prognosis associated with cardiac involvement in CSS&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Ethical disclosures</span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Protection of human and animal subjects</span><p id="par0055" class="elsevierStylePara elsevierViewall">The authors declare that no experiments were performed on humans or animals for this study&#46;</p></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0055">Confidentiality of data</span><p id="par0060" class="elsevierStylePara elsevierViewall">The authors declare that they have followed the protocols of their work center on the publication of patient data and that all the patients included in the study received sufficient information and gave their written informed consent to participate in the study&#46;</p></span><span id="sec0040" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0060">Right to privacy and informed consent</span><p id="par0065" 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="par0070" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare&#46;</p></span></span>"
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            2 => "Miocardite eosinof&#237;lica"
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        "resumen" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Churg&#8211;Strauss syndrome &#40;CSS&#41; is an unusual disease that presents as systemic vasculitis and peripheral eosinophilia in patients with an atopic constitution&#46; Cardiac involvement is unusual and often not prominent on initial presentation&#44; but is an important cause of morbidity and mortality in patients with CSS&#46; We report the case of a young woman with severe acute myocarditis&#46; Coronary arteriography demonstrated extensive focal vasculopathy&#44; consistent with coronary vasculitis&#44; and myocardial biopsy showed eosinophilic myocarditis&#46; This presentation led to an initial diagnosis of CSS in this patient and appropriate therapy resulted in a spectacular remission of disease activity&#46;</p>"
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        "resumen" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A S&#237;ndrome de Churg-Strauss &#233; uma doen&#231;a rara que se caracteriza por vasculite sist&#233;mica associada a eosinofilia perif&#233;rica&#44; tipicamente em doentes com constitui&#231;&#227;o at&#243;pica&#46; O envolvimento card&#237;aco &#233; incomum e geralmente discreto na apresenta&#231;&#227;o inicial mas constitui uma importante causa de morbilidade e mortalidade nestes doentes&#46; Descrevemos o caso de uma mulher jovem admitida por miocardite aguda&#46; A angiografia coron&#225;ria mostrou les&#245;es coron&#225;rias difusas sugestivas de vasculite coron&#225;ria e a bi&#243;psia mioc&#225;rdica mostrou a presen&#231;a de miocardite eosinof&#237;lica&#46; Esta apresenta&#231;&#227;o cl&#237;nica permitiu o diagn&#243;stico inaugural de s&#237;ndrome de Churg-Strauss nesta doente e a institui&#231;&#227;o de terap&#234;utica adequada resultou na remiss&#227;o completa da atividade da doen&#231;a&#46;</p>"
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          "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Electrocardiography at presentation with diffuse T-wave inversion &#40;A&#41; and after recurrent chest pain with transient ST-segment elevation in inferior leads &#40;B&#41;&#46;</p>"
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          "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Coronary angiography at presentation showing diffuse vessel wall irregularities in the left anterior descending artery &#40;A&#41; and right coronary artery &#40;B&#41;&#46; Coronary angiography after one year of immunosuppressive therapy showing resolution of vasculopathy in the anterior descending artery &#40;C&#41; and right coronary artery &#40;D&#41;&#46;</p>"
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          "en" => "<p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Hematoxylin and eosin cardiac biopsy specimen at presentation showing eosinophilic infiltration &#40;A&#41;&#46; Myocardial biopsy&#44; after glucocorticoid and immunosuppressive therapy&#44; showing complete resolution of eosinophilic myocarditis &#40;B&#41;&#46;</p>"
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                      "titulo" => "The American College of Rheumatology 1990 criteria for the classification of Churg&#8211;Strauss syndrome &#40;allergic granulomatosis and angiitis&#41;"
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                        0 => array:2 [
                          "etal" => true
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Case report
Churg–Strauss syndrome presenting with eosinophilic myocarditis: A diagnostic challenge
Síndrome de Churg Strauss complicada com miocardite eosinofílica: um desafio diagnóstico
Ana Sofia Correiaa,
Autor para correspondência
sofiakorreia@gmail.com

Corresponding author.
, Alexandra Gonçalvesa, Vítor Araújoa, João Almeida e Silvab, José Manuel Pereirac, Pedro Rodrigues Pereirad, Manuel Pizarroe, João Carlos Silvaa, Maria Júlia Maciela
a Cardiology Department, Centro Hospitalar de São João, Porto, Portugal
b Cardiology Department, Instituto Português de Oncologia (IPO), Porto, Portugal
c Intensive Care Unit, Centro Hospitalar de São João, Porto, Portugal
d Pathological Anatomy Department, Centro Hospitalar de São João, Porto, Portugal
e Internal Medicine Department, Centro Hospitalar de São João, Porto, Portugal
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subclinical myocardial abnormalities are frequent and are found in more than 50&#37; of post-mortem examinations&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Symptomatic cardiomyopathy carries a poor prognosis&#44; accounting for about 50&#37; of deaths&#44; and is thus the major cause of mortality in CSS&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> It can present with eosinophilic vasculitis&#44; myocarditis&#44; pericarditis&#44; pericardial effusion&#44; fibrosis&#44; valvular heart disease&#44; conduction disorders&#44; intracavitary thrombi&#44; and cardiomyopathy&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#8211;6</span></a> Death is usually due to myocardial infarction&#44; heart failure&#44; malignant ventricular arrhythmias&#44; and&#47;or cardiac tamponade&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We describe the case of a patient with newly diagnosed CCS presenting with pronounced eosinophilic myocarditis and extensive focal vasculopathy on coronary angiography consistent with vasculitis&#46; After appropriate and timely pharmacologic therapeutic intervention the symptoms resolved completely&#44; as did the coronary lesions and myocardial infiltrates&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Case report</span><p id="par0015" class="elsevierStylePara elsevierViewall">A 22-year-old woman with a history of allergic rhinitis&#44; asthma and repeated upper respiratory tract infections in the previous two years developed progressive asthenia&#44; dizziness and left leg paresthesias&#46; One month after the beginning of these non-specific symptoms she was hospitalized because of pleuritic chest pain&#46; She had had no recent flu-like symptoms&#44; either of the upper respiratory or gastrointestinal tract&#44; or other symptoms suggestive of a previous infectious disease&#46; Physical examination was unremarkable&#59; the electrocardiogram &#40;ECG&#41; revealed poor anteroseptal R-wave progression and diffuse T-wave inversion &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Figure 1</a>A&#41;&#46; Laboratory tests showed eosinophilia &#40;3&#46;83<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>10<span class="elsevierStyleSup">9</span>&#47;l&#41;&#44; elevated erythrocyte sedimentation rate &#40;43 mm&#47;h&#41; and elevated biochemical markers of myocardial injury &#40;peak troponin I 155&#46;6 ng&#47;ml&#41;&#46; Serologic and PCR tests were negative for cardiotropic viruses&#44; <span class="elsevierStyleItalic">Aspergillus</span>&#44; <span class="elsevierStyleItalic">Toxoplasma</span>&#44; <span class="elsevierStyleItalic">Chlamydia psittaci</span> and <span class="elsevierStyleItalic">Mycoplasma pneumonia</span>&#46; Specific study for parasites was also negative&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Transthoracic echocardiography revealed a small pericardial effusion with no other abnormalities&#46; A diagnosis of myopericarditis was assumed and anti-inflammatory therapy with ibuprofen was initiated&#46; However&#44; the patient&#39;s recurrent chest pain persisted&#44; associated with dynamic ECG abnormalities &#40;transient ST-segment elevation in inferior leads&#41; &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Figure 1</a>B&#41; and new wall motion abnormalities on echocardiography &#40;inferior wall hypokinesia&#41;&#46; Given the unfavorable clinical evolution associated with a significant rise in plasma troponin I concentration and new ECG and echocardiographic abnormalities&#44; despite anti-inflammatory therapy&#44; cardiac catheterization was performed on the fourth day after admission&#46; Coronary arteriography demonstrated irregularity of the larger arteries with long diffuse stenotic lesions in the left anterior descending and right coronary arteries &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Figure 2</a>A and B&#44; arrowed&#41;&#44; consistent with diffuse vasculitis&#46; A myocardial biopsy showed eosinophilic myocarditis &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Figure 3</a>A&#41; and a concomitant nasal biopsy revealed eosinophilic necrotizing granulomatous vasculitis&#46; Immunologic study&#44; including ANCA antibodies&#44; was negative&#44; while electromyography revealed left saphenous nerve mononeuropathy&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">Churg&#8211;Strauss syndrome was then diagnosed as four out of six criteria were present in this patient&#58; &#40;1&#41; asthma&#59; &#40;2&#41; eosinophilia&#59; &#40;3&#41; mononeuropathy&#59; &#40;4&#41; extravascular eosinophils&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> Pulse intravenous &#40;i&#46;v&#46;&#41; corticosteroid treatment &#40;methylprednisolone 1 g i&#46;v&#46; daily for three days&#41; was started&#44; just after the biopsy results&#44; followed by oral glucocorticoid therapy &#40;prednisone 1&#46;5 mg&#47;kg daily&#41; associated with cyclophosphamide &#40;0&#46;6 g&#47;m<span class="elsevierStyleSup">2</span> intravenously once a month&#41;&#46; On day one of treatment&#44; the patient&#39;s symptoms improved significantly and levels of blood eosinophils fell to 0&#46;02<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>10<span class="elsevierStyleSup">9</span>&#47;l&#46; Echocardiography performed after 18 days of treatment showed resolution of the pericardial effusion as well as of the left ventricular wall motion abnormalities&#46; Control coronary angiography one year after initiation of therapy showed complete regression of coronary stenotic lesions &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Figure 2</a>C and D&#41; and a myocardial biopsy confirmed resolution of eosinophilic myocarditis &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Figure 3</a>B&#41;&#46; The patient was stable and symptom-free at one year of follow-up under chronic therapy&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0030" class="elsevierStylePara elsevierViewall">We describe the case of a patient with newly diagnosed CSS presenting with extensive myocarditis&#44; which is an unusual clinical manifestation of this disease&#46; According to the American College of Rheumatology&#44; the presence of four or more of the six possible criteria &#8211; &#40;1&#41; asthma&#59; &#40;2&#41; eosinophilia &#40;&#62;10&#37; of leukocytes by differential cell count&#41;&#59; &#40;3&#41; mononeuropathy or polyneuropathy&#59; &#40;4&#41; migratory or transient pulmonary infiltrates&#59; &#40;5&#41; paranasal sinus abnormality&#59; and &#40;6&#41; extravascular eosinophils &#8211; establishes the diagnosis with a sensitivity of 85&#37; and a specificity of 99&#37;&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> In our case&#44; a diagnosis of CSS was established based on the patient&#39;s history of asthma&#44; hypereosinophilia&#44; mononeuropathy and extravascular eosinophils on myocardial and nasal biopsy&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">This case report highlights the possibility of cardiac involvement in patients with CSS and calls attention to this differential diagnosis in the evaluation of myocarditis&#46; This diagnosis is mostly overlooked because major cardiac problems are rarely the presenting manifestations of vasculitis&#46; Involvement of the heart has been described in the third stage of the disease&#44; as observed in our patient&#46; It is usually associated with vasculitic lesions in the myocardium and coronary vessels causing &#40;peri&#41;myocarditis&#44; heart failure&#44; cardiac tamponade&#44; myocardial infarction&#44; or pericardial effusion&#46;<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">8&#8211;11</span></a> Our patient presented with pronounced myocarditis but pericardial effusion and coronary vasculitis were also evident&#46; The myocardial damage is caused by vasculitis leading to coronary arteritis and coronary occlusion&#44; through the release by activated eosinophils of toxic mediators causing direct myocardial damage&#44;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">12</span></a> or by replacement of the myocardium with granulomas and scar tissue&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">13&#44;14</span></a> Patients with cardiac involvement are mainly ANCA-negative&#44; as in this case&#46;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">15</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">In general&#44; the prognosis of CSS is good&#44; with an overall 10-year survival of 81-92&#37;&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">11&#44;16</span></a> However&#44; cardiac involvement is one of the most important predictors of an adverse outcome&#44; causing up to 50&#37; of CSS-related mortality&#46;<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">10&#44;16</span></a> In a study on 96 patients&#44;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">16</span></a> 78-month survival was 90&#37; in the absence of symptomatic cardiac manifestations&#44; compared to 30&#37; in their presence&#46; Of the patients with myocardial involvement&#44; 39&#37; died in the acute stage of the disease&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">16</span></a> Early diagnosis of cardiac involvement and subsequent adjuvant therapy may prevent progression of cardiac disease and improve prognosis in these patients&#46; Patients with acute multiorgan disease should receive intravenous glucocorticoid &#40;eg&#46; methylprednisolone 1 g daily for three days&#41; followed by oral glucocorticoid therapy&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;17</span></a> Prednisone doses of 0&#46;5-1&#46;5 mg&#47;kg per day are typically administered until disease remission is attained and then gradually tapered to the lowest dose required for control of symptoms and signs of active CSS&#46; The higher dose is used for patients with more aggressive disease&#44; including those with cardiac involvement&#46; Clinical remission of isolated pericarditis&#44; without other visceral involvement&#44; can be obtained with corticosteroid therapy alone&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">13</span></a> However&#44; in the case of myocardial involvement the addition of immunosuppressive therapy is recommended&#46; In higher-risk patients&#44; including those with myocardial injury&#44; lower mortality was noted among those who were treated with cyclophosphamide compared to a separate group in which only some patients received cyclophosphamide &#40;7&#37; vs&#46; 26&#37; mortality&#44; respectively&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">18</span></a> Cyclophosphamide may be administered orally every day or intravenously once a month&#44; but insufficient data are available on CSS to make a clear recommendation regarding this choice&#46; There is also disagreement concerning the duration of immunosuppressive therapy&#46; However&#44; in a preliminary study of patients with CSS&#44; those receiving six pulses of cyclophosphamide had more relapses than those receiving 12 pulses &#40;94&#37; vs&#46; 41&#37;&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">19</span></a> Further data are needed to clarify whether the benefits of 12 pulses outweigh the additional risks&#44; especially of bladder toxicity&#46; Our case report clearly demonstrates the possibility of complete reversal of cardiac disease with appropriate steroid and immunosuppressive therapy&#44; which is in agreement with previous studies&#46;<a class="elsevierStyleCrossRefs" href="#bib0095"><span class="elsevierStyleSup">19&#8211;22</span></a> However&#44; this vasculitis can be fulminant if not identified early and may on occasion present with cardiogenic shock with a malignant course requiring urgent transplantation&#44; despite therapy&#46;<a class="elsevierStyleCrossRefs" href="#bib0115"><span class="elsevierStyleSup">23&#44;24</span></a> Reports of heart transplantation in this setting are rare and only limited information is available on feasibility&#44; outcome or relapse after heart transplantation in patients with CSS&#46; While one case reported a good long-term result&#44;<a class="elsevierStyleCrossRef" href="#bib0115"><span class="elsevierStyleSup">23</span></a> recurrent disease after initially successful transplantation was observed in another patient&#46;<a class="elsevierStyleCrossRef" href="#bib0125"><span class="elsevierStyleSup">25</span></a> Cardiac involvement in CSS thus requires immediate therapy&#44; which may allow recovery of cardiac function and reduce the significant cardiac mortality associated with CSS&#44; underlining the need for an aggressive invasive diagnostic approach in CSS patients with heart lesions&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">Recent research with multimodality assessment&#44; including ECG analysis&#44; echocardiography and cardiac magnetic resonance imaging&#44; has also shown a high incidence of cardiac involvement &#40;62&#8211;90&#37;&#41; in patients with full clinical remission&#44; characterized not only by fibrosis&#44; but also by an active inflammatory process&#46;<a class="elsevierStyleCrossRefs" href="#bib0130"><span class="elsevierStyleSup">26&#44;27</span></a> Since cardiac involvement is one of the most important predictors of an adverse outcome&#44;<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">18</span></a> these findings could influence current recommendations for cardiac evaluation and therapeutic decisions in asymptomatic patients&#46; An early diagnosis detecting silent inflammation could be valuable&#44;<a class="elsevierStyleCrossRef" href="#bib0140"><span class="elsevierStyleSup">28</span></a> since appropriate therapy can prevent progression to harmful episodes of cardiac disease and so reduce the high mortality associated with these manifestations&#46; Systematic cardiac evaluation in asymptomatic patients with detailed imaging still lacks validation of its clinical benefit but could be an important procedure in the future&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conclusion</span><p id="par0050" class="elsevierStylePara elsevierViewall">Because of its multiple forms of presentation and multiorgan involvement&#44; diagnosis of CSS can be difficult&#46; As reported here&#44; it may present with severe cardiac disease&#46; Cardiac involvement is a poor prognostic factor&#44; and is a leading cause of mortality in CSS&#46; As shown by this case report&#44; appropriate and aggressive glucocorticoid and cyclophosphamide therapy may lead to complete recovery from potentially fatal cardiac disease&#46; Physicians should thus be alert to the possibility of CSS as a differential diagnosis in patients presenting with myocarditis&#44; whenever the clinical setting is appropriate&#46; Prompt diagnosis and therapy can change the poor prognosis associated with cardiac involvement in CSS&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Ethical disclosures</span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Protection of human and animal subjects</span><p id="par0055" class="elsevierStylePara elsevierViewall">The authors declare that no experiments were performed on humans or animals for this study&#46;</p></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0055">Confidentiality of data</span><p id="par0060" class="elsevierStylePara elsevierViewall">The authors declare that they have followed the protocols of their work center on the publication of patient data and that all the patients included in the study received sufficient information and gave their written informed consent to participate in the study&#46;</p></span><span id="sec0040" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0060">Right to privacy and informed consent</span><p id="par0065" 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="par0070" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare&#46;</p></span></span>"
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        "resumen" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Churg&#8211;Strauss syndrome &#40;CSS&#41; is an unusual disease that presents as systemic vasculitis and peripheral eosinophilia in patients with an atopic constitution&#46; Cardiac involvement is unusual and often not prominent on initial presentation&#44; but is an important cause of morbidity and mortality in patients with CSS&#46; We report the case of a young woman with severe acute myocarditis&#46; Coronary arteriography demonstrated extensive focal vasculopathy&#44; consistent with coronary vasculitis&#44; and myocardial biopsy showed eosinophilic myocarditis&#46; This presentation led to an initial diagnosis of CSS in this patient and appropriate therapy resulted in a spectacular remission of disease activity&#46;</p>"
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        "resumen" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A S&#237;ndrome de Churg-Strauss &#233; uma doen&#231;a rara que se caracteriza por vasculite sist&#233;mica associada a eosinofilia perif&#233;rica&#44; tipicamente em doentes com constitui&#231;&#227;o at&#243;pica&#46; O envolvimento card&#237;aco &#233; incomum e geralmente discreto na apresenta&#231;&#227;o inicial mas constitui uma importante causa de morbilidade e mortalidade nestes doentes&#46; Descrevemos o caso de uma mulher jovem admitida por miocardite aguda&#46; A angiografia coron&#225;ria mostrou les&#245;es coron&#225;rias difusas sugestivas de vasculite coron&#225;ria e a bi&#243;psia mioc&#225;rdica mostrou a presen&#231;a de miocardite eosinof&#237;lica&#46; Esta apresenta&#231;&#227;o cl&#237;nica permitiu o diagn&#243;stico inaugural de s&#237;ndrome de Churg-Strauss nesta doente e a institui&#231;&#227;o de terap&#234;utica adequada resultou na remiss&#227;o completa da atividade da doen&#231;a&#46;</p>"
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          "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Electrocardiography at presentation with diffuse T-wave inversion &#40;A&#41; and after recurrent chest pain with transient ST-segment elevation in inferior leads &#40;B&#41;&#46;</p>"
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          "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Coronary angiography at presentation showing diffuse vessel wall irregularities in the left anterior descending artery &#40;A&#41; and right coronary artery &#40;B&#41;&#46; Coronary angiography after one year of immunosuppressive therapy showing resolution of vasculopathy in the anterior descending artery &#40;C&#41; and right coronary artery &#40;D&#41;&#46;</p>"
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                      "titulo" => "Allergic granulomatosis&#46; Allergic angiitis and periarteritis nodosa"
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                          "etal" => false
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                            0 => "J&#46; Churg"
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                0 => array:2 [
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                    0 => array:2 [
                      "titulo" => "Cardiac involvement in the Churg&#8211;Strauss syndrome"
                      "autores" => array:1 [
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Informação do artigo
ISSN: 08702551
Idioma original: Inglês
Dados atualizados diariamente
Ano/Mês Html Pdf Total
2024 Novembro 19 6 25
2024 Outubro 74 26 100
2024 Setembro 84 24 108
2024 Agosto 92 29 121
2024 Julho 56 33 89
2024 Junho 41 20 61
2024 Maio 56 16 72
2024 Abril 54 25 79
2024 Maro 56 25 81
2024 Fevereiro 42 20 62
2024 Janeiro 44 30 74
2023 Dezembro 48 19 67
2023 Novembro 46 27 73
2023 Outubro 36 19 55
2023 Setembro 54 32 86
2023 Agosto 37 16 53
2023 Julho 40 7 47
2023 Junho 56 11 67
2023 Maio 62 33 95
2023 Abril 52 8 60
2023 Maro 53 24 77
2023 Fevereiro 63 16 79
2023 Janeiro 46 12 58
2022 Dezembro 52 16 68
2022 Novembro 66 27 93
2022 Outubro 56 20 76
2022 Setembro 53 22 75
2022 Agosto 80 29 109
2022 Julho 59 36 95
2022 Junho 53 25 78
2022 Maio 44 30 74
2022 Abril 75 15 90
2022 Maro 83 35 118
2022 Fevereiro 90 20 110
2022 Janeiro 72 37 109
2021 Dezembro 52 34 86
2021 Novembro 56 38 94
2021 Outubro 89 39 128
2021 Setembro 73 31 104
2021 Agosto 63 35 98
2021 Julho 59 27 86
2021 Junho 70 23 93
2021 Maio 54 30 84
2021 Abril 124 75 199
2021 Maro 130 17 147
2021 Fevereiro 117 11 128
2021 Janeiro 70 13 83
2020 Dezembro 59 5 64
2020 Novembro 63 12 75
2020 Outubro 89 9 98
2020 Setembro 60 8 68
2020 Agosto 56 8 64
2020 Julho 50 6 56
2020 Junho 65 6 71
2020 Maio 56 2 58
2020 Abril 46 11 57
2020 Maro 76 13 89
2020 Fevereiro 132 29 161
2020 Janeiro 36 3 39
2019 Dezembro 73 8 81
2019 Novembro 34 2 36
2019 Outubro 27 7 34
2019 Setembro 82 7 89
2019 Agosto 39 9 48
2019 Julho 47 10 57
2019 Junho 62 6 68
2019 Maio 60 10 70
2019 Abril 36 15 51
2019 Maro 103 18 121
2019 Fevereiro 111 12 123
2019 Janeiro 127 5 132
2018 Dezembro 96 15 111
2018 Novembro 143 10 153
2018 Outubro 311 23 334
2018 Setembro 41 10 51
2018 Agosto 48 17 65
2018 Julho 39 10 49
2018 Junho 54 6 60
2018 Maio 77 8 85
2018 Abril 63 4 67
2018 Maro 98 8 106
2018 Fevereiro 72 3 75
2018 Janeiro 106 6 112
2017 Dezembro 144 13 157
2017 Novembro 70 11 81
2017 Outubro 49 11 60
2017 Setembro 53 11 64
2017 Agosto 73 14 87
2017 Julho 49 18 67
2017 Junho 48 10 58
2017 Maio 81 13 94
2017 Abril 49 4 53
2017 Maro 93 8 101
2017 Fevereiro 127 16 143
2017 Janeiro 46 11 57
2016 Dezembro 51 10 61
2016 Novembro 53 4 57
2016 Outubro 84 16 100
2016 Setembro 179 16 195
2016 Agosto 84 14 98
2016 Julho 34 13 47
2016 Junho 2 0 2
2016 Maio 27 14 41
2016 Abril 70 2 72
2016 Maro 127 23 150
2016 Fevereiro 126 33 159
2016 Janeiro 102 21 123
2015 Dezembro 146 25 171
2015 Novembro 127 16 143
2015 Outubro 121 25 146
2015 Setembro 128 18 146
2015 Agosto 119 20 139
2015 Julho 159 14 173
2015 Junho 107 5 112
2015 Maio 129 11 140
2015 Abril 101 18 119
2015 Maro 79 14 93
2015 Fevereiro 117 10 127
2015 Janeiro 141 10 151
2014 Dezembro 121 13 134
2014 Novembro 96 21 117
2014 Outubro 118 19 137
2014 Setembro 98 29 127
2014 Agosto 98 11 109
2014 Julho 118 15 133
2014 Junho 68 14 82
2014 Maio 79 17 96
2014 Abril 77 21 98
2014 Maro 101 17 118
2014 Fevereiro 106 23 129
2014 Janeiro 88 32 120
2013 Dezembro 87 20 107
2013 Novembro 112 49 161
2013 Outubro 93 59 152
2013 Setembro 10 8 18
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