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Ankylosing spondylitis (AS) is a chronic inflammatory disease of primarily the joints of the spine. In the literature, AS is known to have cardiac manifestations. Most frequently, this is aortic regurgitation. However, in rare cases also mitral valve (MV) disease is observed in AS patients. The extent and mechanism of this involvement are still unclear. We aim to describe a histologically validated case report to add understanding on this topic.