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Ixekizumab-Induced Cardiac Sarcoidosis: A Case Report.

ABSTRACT: A 58-year-old man with a history of hypertension and psoriasis presented with acute-onset heart failure with an ejection fraction of 25%-30%. During the work-up, cardiac magnetic resonance imaging showed a pattern of inflammation consistent with sarcoidosis, which was confirmed with (18)F-fluorodeoxyglucose positron emission tomography . The patient was recently initiated on ixekizumab for psoriasis, which was then discontinued. This discontinuation resulted in complete resolution of cardiac sarcoidosis, with establishment of normal ejection fraction. This result suggests a potential causal association of ixekizumab-induced cardiac sarcoidosis, which is a rare phenomenon. Elucidation of the mechanism behind the effect of ixekizumab may provide insights into the possible mechanism(s) behind cardiac sarcoidosis.

SUBMITTER: Hatipoglu D

PROVIDER: S-EPMC7801220 | biostudies-literature | 2021 Jan

REPOSITORIES: biostudies-literature

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Ixekizumab-Induced Cardiac Sarcoidosis: A Case Report.

Hatipoglu Dilara D Tanski Christopher C Hachamovitch Rory R Legha Sapna S Moudgil Rohit R

CJC open 20200903 1

A 58-year-old man with a history of hypertension and psoriasis presented with acute-onset heart failure with an ejection fraction of 25%-30%. During the work-up, cardiac magnetic resonance imaging showed a pattern of inflammation consistent with sarcoidosis, which was confirmed with (18)F-fluorodeoxyglucose positron emission tomography . The patient was recently initiated on ixekizumab for psoriasis, which was then discontinued. This discontinuation resulted in complete resolution of cardiac sar ...[more]

PMID: 33458639

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Project description:Background Cardiac sarcoidosis is a chronic, inflammatory disease that can affect the heart and often results in heart failure and lethal arrhythmias. A multimodality imaging approach without endomyocardial biopsy allows for the diagnosis of isolated cardiac sarcoidosis. Coronary vasomotion abnormalities are highly prevalent in various cardiovascular and inflammatory diseases. It remains unknown whether active myocardial inflammation due to cardiac sarcoidosis is associated with coronary vasomotion abnormalities.Case summary A 68-year-old man without a past medical history experienced an out-of-hospital cardiac arrest due to ventricular fibrillation and was successfully resuscitated without neurologic sequelae. Coronary angiography showed normal coronary arteries; however, intracoronary acetylcholine provocation testing demonstrated both epicardial coronary and coronary microvascular spasm. He was diagnosed with isolated cardiac sarcoidosis by fulfilling the diagnostic criteria proposed by the Japanese Circulation Society 2016 diagnostic guidelines, including fatal ventricular arrhythmia, focal left ventricular wall asynergy, increased myocardial fluorodeoxyglucose uptake by positron emission tomography, and late gadolinium enhancement by cardiac magnetic resonance in the heart. He was treated with calcium-channel blocker for coronary artery spasm and prednisolone for cardiac sarcoidosis and underwent implantation of an implantable cardioverter-defibrillator for secondary prevention. Following the treatment, the severity of coronary artery spasm was reduced along with regression of the myocardial inflammation.Discussion Epicardial coronary artery and coronary microvascular spasm can be accompanied by active myocardial inflammation of isolated cardiac sarcoidosis, and the treatment with corticosteroid and calcium-channel blocker may be effective for relieving the severity of coronary artery spasm in association with regression of myocardial inflammation of the disease.

Project description:BackgroundSarcoidosis is a rare multiorgan inflammatory disorder of unknown aetiology, characterized by the formation of non-caseating granulomas in the affected organs. Cardiac involvement is underrecognized and observed in up to 25% of cases in autopsy studies, and is associated with a high mortality rate, especially due to sudden cardiac death due to ventricular arrhythmias.Case summaryA 41-year-old man well known to our hospital because of his father's diagnosis of cardiac amyloidosis, and carrier of transthyretin (TTR) gene mutation, was hospitalized following a resuscitated cardiac arrest. The patient was hospitalized a month before for a syncopal episode with demonstration of preserved left ventricular ejection fraction (LVEF) with akinetic basal septum at heart ultrasound and normal coronary. Chest computed tomography, performed in the emergency department, was significant for hilar lymphadenopathies and pulmonary nodules highly suggestive of sarcoidosis. A subsequent 18-fluorodeoxyglucose-positron emission tomography (FDG-PET) showed multiorgan phlogistic involvement, including the myocardium. After the diagnosis of cardiac sarcoidosis, the patient was started on steroids therapy and underwent ICD implantation. A follow-up 18-FDG-PET showed a reduction of organs glucose uptake and a follow-up echo an improvement in LVEF. Despite that, he occurs occasional recording of repetitive ventricular arrhythmias and one appropriate ICD shock during the next 12 months.DiscussionCardiac sarcoidosis is an insidious disease. Its diagnosis can be challenging, with no specific finding in echocardiography. The best strategy would be multi-modality imaging involving both magnetic resonance imaging with late gadolinium enhancement and 18-FDG-PET, followed by biopsy to confirm the diagnosis. Multi-modality imaging should be further used to evaluate the response to treatment and assess prognosis. Since the patient was a known carrier of the TTR gene mutation, many efforts were made in order to come up with the correct diagnosis considering that both cardiac amyloidosis and cardiac sarcoidosis are non-ischaemic cardiomyopathy with systemic involvement.

Dataset Information

Ixekizumab-Induced Cardiac Sarcoidosis: A Case Report.

Publications

Ixekizumab-Induced Cardiac Sarcoidosis: A Case Report.

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