儿童特发性和非特发性眼球运动性共济失调的特征和结局。

Characteristics and Outcomes of Idiopathic and Non-idiopathic Ocular Motor Apraxia in Children.

出版信息

J Pediatr Ophthalmol Strabismus. 2022 Sep-Oct;59(5):326-331. doi: 10.3928/01913913-20220106-01. Epub 2022 Feb 22.

DOI:10.3928/01913913-20220106-01

PMID:35192381

原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC9840404/

Abstract

PURPOSE

To systematically compare idiopathic and non-idiopathic ocular motor apraxia (OMA) in children.

METHODS

A retrospective chart review was conducted of all children (< 18 years) diagnosed as having OMA from 2010 to 2020. Demographics, clinical characteristics, and oculomotor outcomes were compared for children with idiopathic and non-idiopathic OMA.

RESULTS

Thirty-seven children were included, 17 (46%) with idiopathic OMA and 20 (54%) with non-idiopathic OMA. Among patients with non-idiopathic OMA, Joubert syndrome was the most frequent underlying diagnosis (30%). Strabismus (45% vs 12%, = .04), nystagmus (30% vs 0%, = .02), and vertical saccade involvement (25% vs 0%, = .049) were significantly more common in non-idiopathic than idiopathic OMA, respectively. Neuroimaging abnormalities (90% vs 18%, < .0001) and developmental delays (100% vs 59%, = .002) were also more frequent in non-idiopathic than idiopathic OMA, respectively. Endocrine disorders (most commonly growth hormone deficiency) were diagnosed in 12% and 20% of children with idiopathic and non-idiopathic OMA, respectively ( = .67). On survival curve analysis, improvement in OMA occurred faster and more frequently in children with idiopathic than non-idiopathic OMA (median time to improvement 56 vs 139 months, respectively, = .034).

CONCLUSIONS

Non-idiopathic OMA is associated with a higher rate of vertical saccade involvement, nystagmus, and developmental delays. These findings should prompt neuroimaging in children with OMA. Additionally, endocrine disorders may be more frequent in children with OMA than the general pediatric population. .

摘要

目的

系统比较儿童特发性和非特发性眼运动性失用症（OMA）。

方法

对 2010 年至 2020 年期间所有诊断为 OMA 的儿童（<18 岁）进行回顾性图表审查。比较特发性和非特发性 OMA 儿童的人口统计学、临床特征和眼动结果。

结果

共纳入 37 例患儿，其中特发性 OMA 17 例（46%），非特发性 OMA 20 例（54%）。在非特发性 OMA 患儿中，Joubert 综合征是最常见的潜在诊断（30%）。斜视（45%比 12%， =.04）、眼球震颤（30%比 0%， =.02）和垂直扫视参与（25%比 0%， =.049）在非特发性 OMA 中明显更常见。神经影像学异常（90%比 18%，<.0001）和发育迟缓（100%比 59%， =.002）在非特发性 OMA 中也更常见。特发性和非特发性 OMA 患儿分别诊断出内分泌疾病（最常见的是生长激素缺乏症）12%和 20%（ =.67）。生存曲线分析显示，特发性 OMA 患儿的 OMA 改善更快且更频繁（中位数改善时间分别为 56 和 139 个月， =.034）。