Cardiac valve lipomatous hamartomas are very rare lesions with preferential localization on the pericardium, followed by the endocardial surfaces. We report a quite unique case of a lipomatous hamartoma-like lesion involving a bicuspid aortic valve. The lesion was incidentally found in a young patient undergoing cardiac surgery for aortic regurgitation, and the diagnosis was done by histology. It cannot be excluded that the lipomatous hamartoma-like lesion might have contributed to aortic valve regurgitation.
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