Misawa Manami, Tampo Hironobu, Makino Shinji
Department of Ophthalmology, Jichi Medical University, Tochigi, Shimotsuke, Japan.
Case Rep Ophthalmol Med. 2022 Sep 5;2022:8136115. doi: 10.1155/2022/8136115. eCollection 2022.
This study is aimed at reporting a rare and unusual focal choroidal excavation with a macular hole in a patient with Alagille syndrome (AGS). A 21-year-old woman with an established early-life AGS diagnosis was referred to our hospital prior to liver transplantation. Examination revealed best-corrected visual acuity of 16/20 and 20/20 in the right and left eye, respectively. Slit-lamp examination was positive for posterior embryotoxon in both eyes. Fundoscopy revealed diffuse choroidal hypopigmentation with increased visibility of the choroidal blood vessels and circumferential chorioretinal atrophy in the mid-peripheral and peripheral retina in both eyes. A full-thickness macular hole with underlying focal choroidal excavation was observed in the right eye. Optical coherence tomography through the macula confirmed choroidal excavation with a full-thickness macular hole in the right eye. To our knowledge, this is the first report describing focal choroidal excavation with a macular hole in an AGS patient.
本研究旨在报告1例患有阿拉吉耶综合征(AGS)的患者出现的罕见且特殊的伴有黄斑裂孔的局灶性脉络膜凹陷。一名早年确诊为AGS的21岁女性在肝移植前被转诊至我院。检查发现右眼最佳矫正视力为16/20,左眼为20/20。裂隙灯检查显示双眼均有后胚胎环阳性。眼底镜检查发现双眼脉络膜弥漫性色素减退,脉络膜血管可见度增加,中周部和周边视网膜有环形脉络膜视网膜萎缩。右眼观察到一个伴有下方局灶性脉络膜凹陷的全层黄斑裂孔。通过黄斑的光学相干断层扫描证实右眼存在伴有全层黄斑裂孔的脉络膜凹陷。据我们所知,这是首次报道AGS患者出现伴有黄斑裂孔的局灶性脉络膜凹陷。