Oculomotor Nerve Palsy following Cardiac Tamponade with Churg-Strauss Syndrome: A Case Report

Suganuma, Kazuki; Hashimoto, Takao; Sato, Hiromasa; Suzuki, Tomohiro; Sakurai, Shunpei

doi:10.1159/000334127

Cited by 5 publications

(4 citation statements)

References 10 publications

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“…The CNS vasculitis presented with ischemic brain lesions, 15 , 39 , 55 subarachnoid hemorrhage, 54 seizure, 36 or rarely with oculomotor nerve palsy. 16 In a case reported by Bujak et al, 45 the patient presented with the CNS symptoms due to brain ischemia but it was actually due to embolization from an LV thrombus. Renal involvement was not common in our study population with regard to ANCA positivity (only 9 cases).…”

Section: Resultsmentioning

confidence: 98%

“… 73 The prevalence of pericardial involvement in previous studies is around 20.0%, 66 but severe pericarditis or cardiac tamponade has rarely been reported. 16 Comarmond et al, 74 in a long-term follow-up study of 383 patients suffering from EGPA, reported a 16.4% prevalence rate of cardiomyopathy in the total study group and 19.2% in the ANCA-negative subgroup. In our study, which was confined to patients suffering from EGPA with documented cardiac involvement, we observed cardiomyopathy and pericardial effusion in 71.0% and 37.1% of our cases, respectively.…”

Section: Discussionmentioning

confidence: 95%

“…In an interesting case of EGPA introduced by Yano et al, 38 the patient presented with tamponade, mononeuritis multiplex, skin lesions, and diarrhea, and pericarditis was confirmed by the observation of late gadolinium enhancement in the pericardium in CMR. A unique case by Suganuma et al 16 showed 2 rare manifestations of EGPA: tamponade and subsequently oculomotor nerve palsy. The patient presented with double vision, which developed 18 days after an episode of cardiac tamponade.…”

Section: Resultsmentioning

confidence: 99%

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Cardiac Involvement in Eosinophilic Granulomatosis with Polyangiitis: A Meta-analysis of 62 Case Reports

Pakbaz

2020

JTHC

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Background: Eosinophilic granulomatosis with polyangiitis (EGPA) is a rare multi-systemic vasculitis, with cardiac involvement being one of its most serious manifestations. We aimed to systematically review and analyze the limited case reports of EGPA with cardiac involvement. f EGPA with cardiac involvement. Methods: Based on the Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines, we performed a systematic literature search for the case reports of EGPA with cardiac involvement in the MEDLINE database from 2011 until 2018. For each case, clinical data including sex, age, clinical presentation, electrocardiographic and cardiac imaging findings, the type of cardiac involvement, the available laboratory data (cardiac biomarkers, white blood cell count, eosinophilic count, erythrocyte sedimentation rate, C-reactive protein, and antineutrophil cytoplasmic antibody positivity), therapeutic regimen, and the outcome of the patients were collected and analyzed. Results: A total number of 62 cases were included. The mean age was 48.29±15.60 years, and 51.6% were male. All the cases were in the active disease state. Cardiac symptoms, electrocardiographic abnormalities, abnormal biomarkers, and abnormal echocardiography were detected in 82.3%, 68.5%, 77.4%, and 96.8%, respectively. Cardiac magnetic resonance was done in 46.8% of the patients, and it was abnormal in all. The most common abnormal findings in echocardiography were systolic left ventricular dysfunction (83.9%) and pericardial effusion (37.1%). The most common type of clinical presentation was clinical heart failure (51.6%). Only 6.5% of the patients presented with tamponade. The overall prognosis was good. Conclusion: Any part of the heart could be involved by EGPA. The results emphasize the necessity of in-depth cardiac evaluation in these patients.

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Section: Resultsmentioning

confidence: 98%

Section: Discussionmentioning

confidence: 95%

Section: Resultsmentioning

confidence: 99%

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Cardiac Involvement in Eosinophilic Granulomatosis with Polyangiitis: A Meta-analysis of 62 Case Reports

Pakbaz

2020

JTHC

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“…Our patient had an oculomotor palsy which is very rare and has been observed in only five EGPA patients in the literature. [5][6][7][8][9] Clinical characteristics of these patients are summarized in (Table 1). Even though the number of patient is very low, oculomotor palsy occurs more frequently in ANCA negative patient (80%) and exhibit a striking male predominance (100%).…”

Section: Resultsmentioning

confidence: 99%

A strange oculomotor palsy with eosinophilia

Yıldız

Ould-Nana

Lelotte

et al. 2016

Acta Clinica Belgica

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A 75-year-old man was admitted to the Department of internal medicine because of a 2-month history of neurological deterioration. During the previous year, he complained of recurrent sinusitis, asthma, arthralgias, myalgias and asthenia. Later on, an oculomotor palsy, weakness and disturbance of the sensibility of the right upper limb appeared. Blood sample showed 6510 eosinophils per microlitre. The cerebral magnetic resonance demonstrated bilateral frontal and left parietal subcortical lesions from which the most voluminous presented large haemorrhagic areas. A cerebral biopsy showed small vessel's vasculitis, fibrinoid necrosis and extravascular eosinophilic encroachment. A diagnosis of oculomotor palsy secondary to eosinophilic granulomatosis with polyangeitis was then made, which was successfully treated with corticosteroids and cyclophosphamide.

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Cardiac tamponade leading to the diagnosis of eosinophilic granulomatosis with polyangiitis (Churg−Strauss syndrome): a case report and review of the literature

et al. 2014

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Eosinophilic granulomatosis with polyangiitis (EGPA), which was previously called Churg-Strauss syndrome, is a necrotizing systemic vasculitis of unknown cause accompanied by prominent eosinophilia. Cardiovascular complications, including eosinophilic myocarditis, are a major cause of mortality in this disorder. Acute pericarditis with slight pericardial effusion is a typical manifestation in EGPA, though hemodynamically significant pericardial effusion has been reported in a few cases. We report a case that initially presented with isolated cardiac tamponade, which was followed by systemic manifestations of EGPA over 3 weeks. Including the present case, previous EGPA cases with cardiac tamponade are reviewed to delineate its clinical characteristics.

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Oculomotor Nerve Palsy following Cardiac Tamponade with Churg-Strauss Syndrome: A Case Report

Cited by 5 publications

References 10 publications

Cardiac Involvement in Eosinophilic Granulomatosis with Polyangiitis: A Meta-analysis of 62 Case Reports

Cardiac Involvement in Eosinophilic Granulomatosis with Polyangiitis: A Meta-analysis of 62 Case Reports

A strange oculomotor palsy with eosinophilia

Cardiac tamponade leading to the diagnosis of eosinophilic granulomatosis with polyangiitis (Churg−Strauss syndrome): a case report and review of the literature

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