Optic nerve head drusen (ONHD) are typically benign hyaline and calcified concretions located within the optic nerve. Initially asymptomatic, they can increase slowly in size and number over time, and they can cause mechanical stress at the axons of the optic nerve and progressively lead to a loss of the visual field and papillary vascular changes. Here, we reported a rare case of a young patient with bilateral ONHD complicated by a nonarteritic anterior ischemic optic neuropathy (NAION) in the right eye. The aim of this case was to evaluate, using optical coherence tomography angiography (OCTA), the vascular network of the papillary region in both eyes in the presence of this ischemic event associated to ONHD. At OCTA examination, the whole papillary region revealed a general rarefaction of the vascular network, but it turned out to be greater in the right eye affected by nonarteritic anterior ischemic optic neuropathy than in fellow eye. Therefore, these findings demonstrated that ONHD may determine significant ischemic events of the optic nerve and OCTA represents a valid and noninvasive tool in the diagnosis and in the pathogenesis of these rare cases of NAION associated to ONHD in young patients.

视神经头玻璃膜疣（ONHD）通常是良性透明质酸和位于视神经内的钙化结石。最初无症状，随着时间的推移，它们的大小和数量会逐渐增加，并且会在视神经轴突处引起机械应力，并逐渐导致视野丧失和乳头状血管变化。在这里，我们报道了一名罕见的双眼ONHD并伴右眼非动脉性前部缺血性视神经病变（NAION）的年轻患者。该病例的目的是使用光学相干断层扫描血管造影术（OCTA）评估在存在与ONHD相关的缺血事件的情况下两只眼睛的乳头状区域的血管网络。在OCTA检查中，整个乳头状区域都显示出血管网络的普遍缺乏，但事实证明，受非动脉性前部缺血性视神经病变影响的右眼比另一只眼更大。因此，这些发现表明，ONHD可能决定了视神经的重大缺血事件，而OCTA代表了这些与ONHD相关的少见性NAION在年轻患者中的诊断和发病机制，是一种有效的无创工具。