Ovarian smooth muscle tumours are rare. Notable myxoid change in smooth muscle tumours is uncommon, and raises diagnostic issues that need to be considered on evaluating a spindle cell lesion with notable myxoid change. There is only one case of myxoid leiomyoma of the ovary previously reported. We here report a case of ovarian leiomyoma with areas of myxoid stroma and discuss the relevant differential diagnosis and histological features to be assessed in such a lesion.