Zimmermann Caroline Maria, Singh Shonar, Cardakli Nur, Kraus Courtney Lynn
Department of Ophthalmology and Visual Sciences, University of Maryland School of Medicine, Baltimore, Maryland, USA.
Wilmer Eye Institute, Johns Hopkins University School of Medicine, Baltimore, Maryland, USA.
Neuroophthalmology. 2024 Feb 12;48(1):19-26. doi: 10.1080/01658107.2023.2276189. eCollection 2024.
The purpose of this study was to evaluate the ophthalmologic findings in children with neurofibromatosis type 1 (NF1) and compare these findings in eyes with and without optic pathway gliomas (OPGs). We carried out a retrospective chart review of children with NF1. We recorded demographic characteristics, clinical manifestations of disease, and ophthalmologic findings including visual acuity, intraocular pressure, cup-to-disc ratio, visual field testing, and optical coherence tomography findings. Ophthalmologic findings were examined for the cohort for initial and final appointments. These findings were also compared between eyes with and without OPGs. The study included 119 participants with 238 total eyes. The most common clinical manifestations of NF1 in this cohort were café au lait macules (98%), axillary or inguinal freckling (91%), Lisch nodules (66%), and cutaneous neurofibromas (57%). Thirty-seven participants had imaging that allowed evaluation for choroidal abnormalities, and 28 (76%) had choroidal lesions. Twenty-seven participants (23%) had OPGs, and 44 eyes were affected. On initial assessment, eyes with OPGs had worse visual acuity. On final examination, eyes with OPGs were more likely to have a worse visual acuity and a thinner generalised retinal nerve fibre layer (RNFL) thickness, inferior RNFL thickness, and temporal RNFL thickness. This study provides longitudinal follow-up of children affected by NF1 with and without OPGs. Eyes with OPGs were found to be associated with worse visual acuity and thinner RNFLs overall on final testing.
本研究的目的是评估1型神经纤维瘤病(NF1)患儿的眼科检查结果,并比较有无视神经通路胶质瘤(OPG)的眼睛的这些检查结果。我们对NF1患儿进行了回顾性病历审查。我们记录了人口统计学特征、疾病临床表现以及眼科检查结果,包括视力、眼压、杯盘比、视野检查和光学相干断层扫描结果。对该队列患儿初诊和复诊时的眼科检查结果进行了检查。还比较了有无OPG的眼睛的这些检查结果。该研究纳入了119名参与者,共238只眼睛。该队列中NF1最常见的临床表现为牛奶咖啡斑(98%)、腋窝或腹股沟雀斑(91%)、Lisch结节(66%)和皮肤神经纤维瘤(57%)。37名参与者的影像学检查允许评估脉络膜异常,其中28名(76%)有脉络膜病变。27名参与者(23%)患有OPG,44只眼睛受影响。在初次评估时,患有OPG的眼睛视力较差。在最终检查时,患有OPG的眼睛更有可能视力较差,且总体视网膜神经纤维层(RNFL)厚度、下方RNFL厚度和颞侧RNFL厚度更薄。本研究对患有和未患有OPG的NF1患儿进行了纵向随访。研究发现,在最终检查时,患有OPG的眼睛总体上与较差的视力和较薄的RNFL相关。