Bilateral isolated coronary ostial stenosis in a middle-aged premenopausal woman with vasospastic angina: a case report

Background Vasospastic angina (VSA) is uncommon in premenopausal women who have less chronic endothelial injury causing vascular remodelling, considered to play a primary role in the pathogenesis for coronary vasospasms. Furthermore, vasospasms rarely occur in the bilateral coronary ostia. Isolated coronary ostial stenosis (ICOS), which often causes severe effort angina and requires surgical intervention, is more commonly reported in middle-aged women, with causes including fibromuscular dysplasia (FMD) and large-vessel vasculitis. However, ICOS associated with VSA is extremely rare.Case summary A 50-year-old premenopausal Japanese woman presented with a complaint of typical chest pain due to angina during light exertion daily in the early morning hours since 3 years. Coronary angiography (CAG) revealed bilateral mild-to-moderate ICOS in addition to multi-vessel spasms involving the bilateral coronary ostia confirmed by the vasospasm provocation test using intracoronary acetylcholine injection. Tests to determine the cause of ICOS did not identify FMD or any other disease. The angina attacks alleviated after calcium channel blocker (CCB) administration without intervention for bilateral ICOS for 24 years since the first presentation. Moreover, coronary computed tomography angiography (CTA) performed 24 years after the first presentation showed no ICOS.Discussion In our patient with typical and frequent VSA symptoms, CAG revealed both mild-to-moderate ICOS and the vasospasms in the bilateral coronary ostia. Fibromuscular dysplasia or large-vessel vasculitis was ruled out as the causes of ICOS. Vasospastic angina rarely occurred after the prescription of CCB, and coronary CTA 24 years after the first presentation showed no ICOS. Bilateral ICOS in our patient might be VSA related.