PurposeTo present the unique case of numerous, recurring retinal arterial macroaneurysms (RAMs) in a hypothyroid patient with hypertension.Methods67-year-old woman’s clinical findings, laboratory results, fundus fluorescein angiography, optical coherence tomography (OCT), and optical coherence tomography angiography (OCTA) are given. Over a two-year period, the patient was monitored.ResultsA 67-year-old woman presented to the outpatient clinic with vitreous and dense subretinal hemorrhages in her right eye. RAM rupture was discovered along the suprotemporal retinal arteriole. A diagnosis of systemic arterial hypertension was made. Two months later, the vitreous hemorrhage spontaneously resolved and the patient’s vision improved. After nine months of initial presentation, the patient developed another RAM. Meanwhile the patient developed ichthyosis caused by hypothyroidism. Because fundus fluorescein angiography revealed that the first RAM was still active, an intravitreal injection of anti-VEGF was administered six months afterwards. More proximal RAM with macular edema developed after another six months necessitating laser photocoagulation. However macular edema didn’t resolve and a second injection of intravitreal anti-VEGF was given.ConclusionsPatients with RAMs, particularly if multiple or recurring, should be thoroughly investigated and assessed, particularly for secondary causes of hypertension. OCT and OCTA are useful tools for RAM confirmation and follow-up. It is important to look into how RAM behavior interacts with coexisting macular edema, and other variables affecting hemodynamic status.Supplementary InformationThe online version contains supplementary material available at 10.1186/s12886-023-02953-x.