Department of Ophthalmology, Edward S. Harkness Eye Institute, Columbia University Irving Medical Center, 635 West 165th Street, Box 212, New York, NY, 10032, USA.
Jonas Children's Vision Care, New York, NY, USA.
Sci Rep. 2021 Jul 12;11(1):14300. doi: 10.1038/s41598-021-93316-3.
Characterization of vascular impairment in Best vitelliform macular dystrophy (BVMD) is essential for the development of treatment modalities and therapy trials. As such, we seek to characterize the choriocapillaris (CC) at each stage of the disease process in 22 patients (44 eyes) with a diagnosis of BVMD confirmed by genetic sequencing. We utilize optical coherence tomography angiography (OCTA) images to characterize the CC and correlate our findings to the status of the retinal pigment epithelium (RPE) as observed on short-wavelength fundus autofluorescence (SW-AF) images. We observed that in the vitelliruptive stage, the CC appeared as bright and granular in the area where the vitelliform lesion was present. In the atrophic stage, varying degrees of CC atrophy were observed within the lesion area, with the regions of CC atrophy appearing as hypoautofluorescent on SW-AF images. Our results suggest that the CC impairment observed in the vitelliruptive stage of BVMD progressively culminates in the CC atrophy observed at the atrophic stage. As such, OCTA imaging can be used to characterize CC impairment in BVMD patients as part of diagnosis and tracking of disease progression. Our findings suggest that the best window of opportunity for therapeutic approaches is before the atrophic stage, as it is during this stage that CC atrophy is observed.
对 Best 型卵黄样黄斑营养不良症 (BVMD) 血管损伤的特征分析,对于治疗方法和疗法试验的开发至关重要。因此,我们旨在通过基因测序确诊为 BVMD 的 22 名患者(44 只眼)的 OCTA 图像来分析脉络膜毛细血管(CC)在疾病进程的各个阶段的特征,并将我们的发现与短波长眼底自发荧光(SW-AF)图像上观察到的视网膜色素上皮(RPE)状态相关联。我们观察到,在玻璃膜疣期,CC 在存在卵黄样病变的区域呈现出明亮的颗粒状。在萎缩期,病变区域内观察到不同程度的 CC 萎缩,CC 萎缩区域在 SW-AF 图像上呈现出低自发荧光。我们的研究结果表明,BVMD 玻璃膜疣期观察到的 CC 损伤逐渐进展为萎缩期观察到的 CC 萎缩。因此,OCTA 成像可用于对 BVMD 患者的 CC 损伤进行特征分析,作为诊断和疾病进展跟踪的一部分。我们的研究结果表明,治疗方法的最佳机会窗口是在萎缩期之前,因为正是在这个阶段观察到 CC 萎缩。