Hu Zhengping, Cao Jian, Choi Eun Young, Li Yun
Schepens Eye Research Institute, Mass. Eye and Ear, Boston, MA, USA.
Harvard Medical School, Boston, MA, USA.
BMC Ophthalmol. 2021 Jan 22;21(1):56. doi: 10.1186/s12886-021-01815-8.
Sturge-Weber syndrome is a disorder marked by a distinctive facial capillary malformation, neurological abnormalities, and ocular abnormalities such as glaucoma and choroidal hemangioma.
We report a case of progressively formed retinal vessel malformation in a premature male infant with Sturge-Weber syndrome and retinopathy of prematurity, after treatment with intravitreal anti-vascular endothelial growth factor (VEGF). The baby was born at 30 weeks gestation with a nevus flammeus involving his left eyelids and maxillary area. On postmenstrual age week 39, he received intravitreal anti-VEGF. Diffuse choroidal hemangioma became evident at 40 weeks, with the classic "tomato catsup fundus" appearance. These clinical findings characterized Sturge-weber syndrome. He presented with posterior retinal vessel tortuosity and vein-to-vein anastomoses at 44 weeks.
This is a rare case of documented progression of retinal vessel malformations in a patient with Sturge-Weber syndrome and retinopathy of prematurity.
斯-韦综合征是一种以独特的面部毛细血管畸形、神经异常以及眼部异常（如青光眼和脉络膜血管瘤）为特征的疾病。
我们报告了一例早产男婴，患有斯-韦综合征和早产儿视网膜病变，在接受玻璃体内抗血管内皮生长因子（VEGF）治疗后，出现了逐渐形成的视网膜血管畸形。该婴儿在妊娠30周时出生，左侧眼睑和上颌区域有葡萄酒色斑。在月经龄第39周时，他接受了玻璃体内抗VEGF治疗。在40周时，弥漫性脉络膜血管瘤变得明显，呈现出典型的“番茄酱眼底”外观。这些临床表现符合斯-韦综合征。在44周时，他出现了视网膜后血管迂曲和静脉间吻合。
这是一例罕见的记录在案的斯-韦综合征和早产儿视网膜病变患者视网膜血管畸形进展的病例。
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