Churg–Strauss syndrome presenting with eosinophilic myocarditis: A diagnostic challenge | Revista Portuguesa de Cardiologia (English edition)

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Department, Centro Hospitalar de São João, Porto, Portugal" "etiqueta" => "e" "identificador" => "aff0025" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "pt" => array:1 [ "titulo" => "Síndrome de Churg Strauss complicada com miocardite eosinofílica: um desafio diagnóstico" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 1454 "Ancho" => 2334 "Tamanyo" => 668683 ] ] "descripcion" => array:1 [ "en" => "Hematoxylin and eosin cardiac biopsy specimen at presentation showing eosinophilic infiltration (A). Myocardial biopsy, after glucocorticoid and immunosuppressive therapy, showing complete resolution of eosinophilic myocarditis (B)." ] ] ] "textoCompleto" => "IntroductionChurg–Strauss syndrome (CSS) is a rare primary systemic vasculitis characterized by peripheral eosinophilia in patients with an atopic condition, typically with a previous history of asthma or allergy.<a class="elsevierStyleCrossRef" href="#bib0005">1</a> It affects both small and medium-sized blood vessels of nearly all organ systems. The pathophysiology of this syndrome can be divided into three stages: first, a prodromal stage characterized by asthma and allergic manifestations; second, eosinophilic infiltration into tissues, predominantly the lungs and myocardium; and finally, a systemic stage, associated with the development of necrotizing vasculitis.<a class="elsevierStyleCrossRefs" href="#bib0010">2,3</a> Although cardiac involvement is unusual and often not prominent on initial presentation, subclinical myocardial abnormalities are frequent and are found in more than 50% of post-mortem examinations.<a class="elsevierStyleCrossRef" href="#bib0005">1</a> Symptomatic cardiomyopathy carries a poor prognosis, accounting for about 50% of deaths, and is thus the major cause of mortality in CSS.<a class="elsevierStyleCrossRef" href="#bib0010">2</a> It can present with eosinophilic vasculitis, myocarditis, pericarditis, pericardial effusion, fibrosis, valvular heart disease, conduction disorders, intracavitary thrombi, and cardiomyopathy.<a class="elsevierStyleCrossRefs" href="#bib0020">4–6</a> Death is usually due to myocardial infarction, heart failure, malignant ventricular arrhythmias, and/or cardiac tamponade.<a class="elsevierStyleCrossRef" href="#bib0020">4</a>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. After appropriate and timely pharmacologic therapeutic intervention the symptoms resolved completely, as did the coronary lesions and myocardial infiltrates.Case reportA 22-year-old woman with a history of allergic rhinitis, asthma and repeated upper respiratory tract infections in the previous two years developed progressive asthenia, dizziness and left leg paresthesias. One month after the beginning of these non-specific symptoms she was hospitalized because of pleuritic chest pain. She had had no recent flu-like symptoms, either of the upper respiratory or gastrointestinal tract, or other symptoms suggestive of a previous infectious disease. Physical examination was unremarkable; the electrocardiogram (ECG) revealed poor anteroseptal R-wave progression and diffuse T-wave inversion (<a class="elsevierStyleCrossRef" href="#fig0005">Figure 1</a>A). Laboratory tests showed eosinophilia (3.83×109/l), elevated erythrocyte sedimentation rate (43 mm/h) and elevated biochemical markers of myocardial injury (peak troponin I 155.6 ng/ml). Serologic and PCR tests were negative for cardiotropic viruses, Aspergillus, Toxoplasma, Chlamydia psittaci and Mycoplasma pneumonia. Specific study for parasites was also negative.<elsevierMultimedia ident="fig0005"></elsevierMultimedia>Transthoracic echocardiography revealed a small pericardial effusion with no other abnormalities. A diagnosis of myopericarditis was assumed and anti-inflammatory therapy with ibuprofen was initiated. However, the patient's recurrent chest pain persisted, associated with dynamic ECG abnormalities (transient ST-segment elevation in inferior leads) (<a class="elsevierStyleCrossRef" href="#fig0005">Figure 1</a>B) and new wall motion abnormalities on echocardiography (inferior wall hypokinesia). Given the unfavorable clinical evolution associated with a significant rise in plasma troponin I concentration and new ECG and echocardiographic abnormalities, despite anti-inflammatory therapy, cardiac catheterization was performed on the fourth day after admission. Coronary arteriography demonstrated irregularity of the larger arteries with long diffuse stenotic lesions in the left anterior descending and right coronary arteries (<a class="elsevierStyleCrossRef" href="#fig0010">Figure 2</a>A and B, arrowed), consistent with diffuse vasculitis. A myocardial biopsy showed eosinophilic myocarditis (<a class="elsevierStyleCrossRef" href="#fig0015">Figure 3</a>A) and a concomitant nasal biopsy revealed eosinophilic necrotizing granulomatous vasculitis. Immunologic study, including ANCA antibodies, was negative, while electromyography revealed left saphenous nerve mononeuropathy.<elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia>Churg–Strauss syndrome was then diagnosed as four out of six criteria were present in this patient: (1) asthma; (2) eosinophilia; (3) mononeuropathy; (4) extravascular eosinophils.<a class="elsevierStyleCrossRef" href="#bib0035">7</a> Pulse intravenous (i.v.) corticosteroid treatment (methylprednisolone 1 g i.v. daily for three days) was started, just after the biopsy results, followed by oral glucocorticoid therapy (prednisone 1.5 mg/kg daily) associated with cyclophosphamide (0.6 g/m2 intravenously once a month). On day one of treatment, the patient's symptoms improved significantly and levels of blood eosinophils fell to 0.02×109/l. Echocardiography performed after 18 days of treatment showed resolution of the pericardial effusion as well as of the left ventricular wall motion abnormalities. Control coronary angiography one year after initiation of therapy showed complete regression of coronary stenotic lesions (<a class="elsevierStyleCrossRef" href="#fig0010">Figure 2</a>C and D) and a myocardial biopsy confirmed resolution of eosinophilic myocarditis (<a class="elsevierStyleCrossRef" href="#fig0015">Figure 3</a>B). The patient was stable and symptom-free at one year of follow-up under chronic therapy.DiscussionWe describe the case of a patient with newly diagnosed CSS presenting with extensive myocarditis, which is an unusual clinical manifestation of this disease. According to the American College of Rheumatology, the presence of four or more of the six possible criteria – (1) asthma; (2) eosinophilia (>10% of leukocytes by differential cell count); (3) mononeuropathy or polyneuropathy; (4) migratory or transient pulmonary infiltrates; (5) paranasal sinus abnormality; and (6) extravascular eosinophils – establishes the diagnosis with a sensitivity of 85% and a specificity of 99%.<a class="elsevierStyleCrossRef" href="#bib0035">7</a> In our case, a diagnosis of CSS was established based on the patient's history of asthma, hypereosinophilia, mononeuropathy and extravascular eosinophils on myocardial and nasal biopsy.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. This diagnosis is mostly overlooked because major cardiac problems are rarely the presenting manifestations of vasculitis. Involvement of the heart has been described in the third stage of the disease, as observed in our patient. It is usually associated with vasculitic lesions in the myocardium and coronary vessels causing (peri)myocarditis, heart failure, cardiac tamponade, myocardial infarction, or pericardial effusion.<a class="elsevierStyleCrossRefs" href="#bib0040">8–11</a> Our patient presented with pronounced myocarditis but pericardial effusion and coronary vasculitis were also evident. The myocardial damage is caused by vasculitis leading to coronary arteritis and coronary occlusion, through the release by activated eosinophils of toxic mediators causing direct myocardial damage,<a class="elsevierStyleCrossRef" href="#bib0060">12</a> or by replacement of the myocardium with granulomas and scar tissue.<a class="elsevierStyleCrossRefs" href="#bib0065">13,14</a> Patients with cardiac involvement are mainly ANCA-negative, as in this case.<a class="elsevierStyleCrossRef" href="#bib0075">15</a>In general, the prognosis of CSS is good, with an overall 10-year survival of 81-92%.<a class="elsevierStyleCrossRefs" href="#bib0055">11,16</a> However, cardiac involvement is one of the most important predictors of an adverse outcome, causing up to 50% of CSS-related mortality.<a class="elsevierStyleCrossRefs" href="#bib0050">10,16</a> In a study on 96 patients,<a class="elsevierStyleCrossRef" href="#bib0080">16</a> 78-month survival was 90% in the absence of symptomatic cardiac manifestations, compared to 30% in their presence. Of the patients with myocardial involvement, 39% died in the acute stage of the disease.<a class="elsevierStyleCrossRef" href="#bib0080">16</a> Early diagnosis of cardiac involvement and subsequent adjuvant therapy may prevent progression of cardiac disease and improve prognosis in these patients. Patients with acute multiorgan disease should receive intravenous glucocorticoid (eg. methylprednisolone 1 g daily for three days) followed by oral glucocorticoid therapy.<a class="elsevierStyleCrossRefs" href="#bib0010">2,17</a> Prednisone doses of 0.5-1.5 mg/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. The higher dose is used for patients with more aggressive disease, including those with cardiac involvement. Clinical remission of isolated pericarditis, without other visceral involvement, can be obtained with corticosteroid therapy alone.<a class="elsevierStyleCrossRef" href="#bib0065">13</a> However, in the case of myocardial involvement the addition of immunosuppressive therapy is recommended. In higher-risk patients, including those with myocardial injury, lower mortality was noted among those who were treated with cyclophosphamide compared to a separate group in which only some patients received cyclophosphamide (7% vs. 26% mortality, respectively).<a class="elsevierStyleCrossRef" href="#bib0090">18</a> Cyclophosphamide may be administered orally every day or intravenously once a month, but insufficient data are available on CSS to make a clear recommendation regarding this choice. There is also disagreement concerning the duration of immunosuppressive therapy. However, in a preliminary study of patients with CSS, those receiving six pulses of cyclophosphamide had more relapses than those receiving 12 pulses (94% vs. 41%).<a class="elsevierStyleCrossRef" href="#bib0095">19</a> Further data are needed to clarify whether the benefits of 12 pulses outweigh the additional risks, especially of bladder toxicity. Our case report clearly demonstrates the possibility of complete reversal of cardiac disease with appropriate steroid and immunosuppressive therapy, which is in agreement with previous studies.<a class="elsevierStyleCrossRefs" href="#bib0095">19–22</a> However, this vasculitis can be fulminant if not identified early and may on occasion present with cardiogenic shock with a malignant course requiring urgent transplantation, despite therapy.<a class="elsevierStyleCrossRefs" href="#bib0115">23,24</a> Reports of heart transplantation in this setting are rare and only limited information is available on feasibility, outcome or relapse after heart transplantation in patients with CSS. While one case reported a good long-term result,<a class="elsevierStyleCrossRef" href="#bib0115">23</a> recurrent disease after initially successful transplantation was observed in another patient.<a class="elsevierStyleCrossRef" href="#bib0125">25</a> Cardiac involvement in CSS thus requires immediate therapy, which may allow recovery of cardiac function and reduce the significant cardiac mortality associated with CSS, underlining the need for an aggressive invasive diagnostic approach in CSS patients with heart lesions.Recent research with multimodality assessment, including ECG analysis, echocardiography and cardiac magnetic resonance imaging, has also shown a high incidence of cardiac involvement (62–90%) in patients with full clinical remission, characterized not only by fibrosis, but also by an active inflammatory process.<a class="elsevierStyleCrossRefs" href="#bib0130">26,27</a> Since cardiac involvement is one of the most important predictors of an adverse outcome,<a class="elsevierStyleCrossRef" href="#bib0090">18</a> these findings could influence current recommendations for cardiac evaluation and therapeutic decisions in asymptomatic patients. An early diagnosis detecting silent inflammation could be valuable,<a class="elsevierStyleCrossRef" href="#bib0140">28</a> since appropriate therapy can prevent progression to harmful episodes of cardiac disease and so reduce the high mortality associated with these manifestations. 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.ConclusionBecause of its multiple forms of presentation and multiorgan involvement, diagnosis of CSS can be difficult. As reported here, it may present with severe cardiac disease. Cardiac involvement is a poor prognostic factor, and is a leading cause of mortality in CSS. As shown by this case report, appropriate and aggressive glucocorticoid and cyclophosphamide therapy may lead to complete recovery from potentially fatal cardiac disease. Physicians should thus be alert to the possibility of CSS as a differential diagnosis in patients presenting with myocarditis, whenever the clinical setting is appropriate. Prompt diagnosis and therapy can change the poor prognosis associated with cardiac involvement in CSS.Ethical disclosuresProtection of human and animal subjectsThe authors declare that no experiments were performed on humans or animals for this study.Confidentiality of dataThe 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.Right to privacy and informed consentThe authors declare that no patient data appear in this article.Conflicts of interestThe authors have no conflicts of interest to declare." 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Cardiac involvement is unusual and often not prominent on initial presentation, but is an important cause of morbidity and mortality in patients with CSS. We report the case of a young woman with severe acute myocarditis. Coronary arteriography demonstrated extensive focal vasculopathy, consistent with coronary vasculitis, and myocardial biopsy showed eosinophilic myocarditis. This presentation led to an initial diagnosis of CSS in this patient and appropriate therapy resulted in a spectacular remission of disease activity." ] "pt" => array:2 [ "titulo" => "Resumo" "resumen" => "A Síndrome de Churg-Strauss é uma doença rara que se caracteriza por vasculite sistémica associada a eosinofilia periférica, tipicamente em doentes com constituição atópica. O envolvimento cardíaco é incomum e geralmente discreto na apresentação inicial mas constitui uma importante causa de morbilidade e mortalidade nestes doentes. Descrevemos o caso de uma mulher jovem admitida por miocardite aguda. A angiografia coronária mostrou lesões coronárias difusas sugestivas de vasculite coronária e a biópsia miocárdica mostrou a presença de miocardite eosinofílica. Esta apresentação clínica permitiu o diagnóstico inaugural de síndrome de Churg-Strauss nesta doente e a instituição de terapêutica adequada resultou na remissão completa da atividade da doença." ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "Please cite this article as: Correia AS, Gonçalves A, Araújo V, Almeida e Silva J, Pereira JM, Rodrigues Pereira P, et al. Síndrome de Churg Strauss complicada com miocardite eosinofílica: um desafio diagnóstico 2012. http://dx.doi.org/." ] ] "multimedia" => array:3 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 707 "Ancho" => 2995 "Tamanyo" => 513481 ] ] "descripcion" => array:1 [ "en" => "Electrocardiography at presentation with diffuse T-wave inversion (A) and after recurrent chest pain with transient ST-segment elevation in inferior leads (B)." ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1654 "Ancho" => 1667 "Tamanyo" => 229652 ] ] "descripcion" => array:1 [ "en" => "Coronary angiography at presentation showing diffuse vessel wall irregularities in the left anterior descending artery (A) and right coronary artery (B). Coronary angiography after one year of immunosuppressive therapy showing resolution of vasculopathy in the anterior descending artery (C) and right coronary artery (D)." ] ] 2 => array:7 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 1454 "Ancho" => 2334 "Tamanyo" => 668683 ] ] "descripcion" => array:1 [ "en" => "Hematoxylin and eosin cardiac biopsy specimen at presentation showing eosinophilic infiltration (A). 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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,

Corresponding author

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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    "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Churg&#8211;Strauss syndrome &#40;CSS&#41; is a rare primary systemic vasculitis characterized by peripheral eosinophilia in patients with an atopic condition&#44; typically with a previous history of asthma or allergy&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> It affects both small and medium-sized blood vessels of nearly all organ systems&#46; The pathophysiology of this syndrome can be divided into three stages&#58; first&#44; a prodromal stage characterized by asthma and allergic manifestations&#59; second&#44; eosinophilic infiltration into tissues&#44; predominantly the lungs and myocardium&#59; and finally&#44; a systemic stage&#44; associated with the development of necrotizing vasculitis&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;3</span></a> Although cardiac involvement is unusual and often not prominent on initial presentation&#44; 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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        "titulo" => "Abstract"
        "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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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Correia AS&#44; Gon&#231;alves A&#44; Ara&#250;jo V&#44; Almeida e Silva J&#44; Pereira JM&#44; Rodrigues Pereira P&#44; et al&#46; S&#237;ndrome de Churg Strauss complicada com miocardite eosinof&#237;lica&#58; um desafio diagn&#243;stico 2012&#46; http&#58;&#47;&#47;dx&#46;doi&#46;org&#47;&#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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Article information

ISSN: 21742049
Original language: English

DOI:10.1016/j.repce.2012.10.012

The statistics are updated each day

Year/Month	Html	Pdf	Total

2024 November	14	3	17
2024 October	211	26	237
2024 September	145	27	172
2024 August	229	47	276
2024 July	151	25	176
2024 June	105	20	125
2024 May	161	22	183
2024 April	163	31	194
2024 March	182	31	213
2024 February	144	22	166
2024 January	141	40	181
2023 December	120	30	150
2023 November	122	34	156
2023 October	73	17	90
2023 September	61	30	91
2023 August	54	19	73
2023 July	42	13	55
2023 June	51	13	64
2023 May	90	39	129
2023 April	28	8	36
2023 March	43	21	64
2023 February	49	23	72
2023 January	41	18	59
2022 December	35	18	53
2022 November	58	26	84
2022 October	38	19	57
2022 September	23	34	57
2022 August	30	42	72
2022 July	49	48	97
2022 June	35	14	49
2022 May	37	30	67
2022 April	38	20	58
2022 March	50	27	77
2022 February	46	26	72
2022 January	35	39	74
2021 December	32	23	55
2021 November	39	42	81
2021 October	55	46	101
2021 September	40	26	66
2021 August	31	24	55
2021 July	24	26	50
2021 June	31	14	45
2021 May	34	33	67
2021 April	80	51	131
2021 March	81	16	97
2021 February	65	18	83
2021 January	45	12	57
2020 December	46	10	56
2020 November	63	9	72
2020 October	36	15	51
2020 September	56	11	67
2020 August	48	7	55
2020 July	50	5	55
2020 June	44	8	52
2020 May	65	6	71
2020 April	51	9	60
2020 March	59	13	72
2020 February	121	18	139
2020 January	52	8	60
2019 December	103	11	114
2019 November	86	6	92
2019 October	163	16	179
2019 September	60	15	75
2019 August	43	1	44
2019 July	57	18	75
2019 June	41	7	48
2019 May	40	8	48
2019 April	49	19	68
2019 March	112	28	140
2019 February	87	15	102
2019 January	125	11	136
2018 December	117	13	130
2018 November	117	4	121
2018 October	182	20	202
2018 September	71	15	86
2018 August	69	8	77
2018 July	47	6	53
2018 June	60	6	66
2018 May	71	7	78
2018 April	65	5	70
2018 March	62	7	69
2018 February	45	4	49
2018 January	42	7	49
2017 December	69	7	76
2017 November	39	6	45
2017 October	40	6	46
2017 September	49	3	52
2017 August	47	8	55
2017 July	43	10	53
2017 June	40	4	44
2017 May	46	8	54
2017 April	23	2	25
2017 March	37	12	49
2017 February	40	3	43
2017 January	45	4	49
2016 December	35	11	46
2016 November	23	3	26
2016 October	39	5	44
2016 September	15	11	26
2016 August	4	3	7
2016 July	7	3	10
2016 June	5	4	9
2016 May	12	6	18
2016 April	21	7	28
2016 March	29	12	41
2016 February	54	19	73
2016 January	36	11	47
2015 December	36	10	46
2015 November	30	15	45
2015 October	29	9	38
2015 September	32	9	41
2015 August	33	10	43
2015 July	24	8	32
2015 June	10	1	11
2015 May	28	7	35
2015 April	24	3	27
2015 March	16	3	19
2015 February	22	2	24
2015 January	23	3	26
2014 December	27	8	35
2014 November	16	4	20
2014 October	35	10	45
2014 September	31	2	33
2014 August	29	3	32
2014 July	41	8	49
2014 June	29	2	31
2014 May	33	9	42
2014 April	36	7	43
2014 March	63	14	77
2014 February	52	4	56
2014 January	45	14	59
2013 December	30	8	38

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