Saffren Brooke D, Yassin Shaden H, Geddie Brooke E, de Faber Jan Tjeerd H N, Blieden Lauren S, Bhate Manjushree, Gamio Susana, Rutar Tina, Levin Alex V
Philadelphia College of Osteopathic Medicine (BDS), Philadelphia, Pennsylvania; Pediatric Ophthalmology and Ocular Genetics (BDS, SHY), Wills Eye Hospital, Philadelphia, Pennsylvania; Pediatric Ophthalmology (BEG), Helen DeVos Children's Hospital, Grand Rapids, Michigan; The Rotterdam Eye Hospital (JTHNF), Rotterdam, the Netherlands ; Cullen Eye Institute (LSB), Baylor College of Medicine, Houston, Texas; Department of Pediatric Ophthalmology (MB), LV Prasad Eye Institute, Hyderabad, India ; Ophthalmology Unit (SG), Ricardo Gutiérrez Children Hospital, Buenos Aires, Argentina ; Cataract and Laser Institute of Southern Oregon PC (TR), Medford, Oregon; Flaum Eye Institute and Golisano Children's Hospital (AVL), University of Rochester, New York, New York.
J Neuroophthalmol. 2022 Mar 1;42(1):e140-e146. doi: 10.1097/WNO.0000000000001246. Epub 2021 Apr 14.
Optic nerve aplasia (ONA) is a rare ocular anomaly. We report ophthalmologic, systemic, and genetic findings in ONA.
Patients were identified through an International Pediatric Ophthalmology listserv and from the practice of the senior author. Participating Listserv physicians completed a data collection sheet. Children of all ages were included. Neuroimaging findings were also recorded.
Nine cases of ONA are reported. Patients' ages ranged from 10 days to 2 years (median 9 months). Seven cases were bilateral. All patients had absence of the optic nerve and retinal vessels in the affected eye or eyes. Ophthalmologic findings included glaucoma, microcornea, persistent pupillary membrane, iris coloboma, aniridia, retinal dysplasia, retinal atrophy, chorioretinal coloboma, and persistent fetal vasculature. Systemic findings included facial dysmorphism, cardiac, genitourinary, skeletal, and developmental defects. A BCOR mutation was found in one patient. One patient had rudimentary optic nerves and chiasm on imaging.
ONA is a unilateral or bilateral condition that may be associated with anomalies of the anterior or posterior segment with or without systemic findings. Rudimentary optic nerve on neuroimaging in one case suggests that ONA is on the continuum of optic nerve hypoplasia.
视神经发育不全(ONA)是一种罕见的眼部异常。我们报告了ONA的眼科、全身及遗传学检查结果。
通过国际小儿眼科邮件列表以及资深作者的临床实践来确定患者。参与邮件列表的医生填写了一份数据收集表。纳入了各年龄段的儿童。还记录了神经影像学检查结果。
报告了9例ONA病例。患者年龄从10天至2岁不等(中位数为9个月)。7例为双侧病变。所有患者患眼均无视神经及视网膜血管。眼科检查结果包括青光眼、小角膜、永存瞳孔膜、虹膜缺损、无虹膜、视网膜发育异常、视网膜萎缩、脉络膜视网膜缺损以及永存原始玻璃体增生症。全身检查结果包括面部畸形、心脏、泌尿生殖系统、骨骼及发育缺陷。1例患者发现有BCOR基因突变。1例患者影像学检查显示有发育不全的视神经及视交叉。
ONA可为单侧或双侧病变,可能伴有前段或后段异常,有或无全身检查结果。1例患者神经影像学检查显示有发育不全的视神经,提示ONA处于视神经发育不良的连续谱系中。