视神经发育不良和视隔发育不良患儿的危险因素。

Risk factors in children with optic nerve hypoplasia and septo-optic dysplasia.

机构信息

Section of Pediatric Neurology, Department of Pediatrics and Child Health, Max Rady College of Medicine, Rady Faculty of Health Sciences, University of Manitoba, Winnipeg, Manitoba, Canada.

Section of Neonatology, Department of Pediatrics and Child Health, Max Rady College of Medicine, Rady Faculty of Health Sciences, University of Manitoba, Winnipeg, Manitoba, Canada.

出版信息

Dev Med Child Neurol. 2024 Jan;66(1):106-116. doi: 10.1111/dmcn.15678. Epub 2023 Jul 2.

DOI:10.1111/dmcn.15678

PMID:37394738

Abstract

AIM

To identify the risk factors for optic nerve hypoplasia (ONH) and septo-optic dysplasia (SOD).

METHOD

A retrospective, population-based study with case-control design was undertaken using the Population Research Data Repository at the Manitoba Center for Health Policy in Manitoba, Canada. Cases were 111 patients (63 males, 48 females; age range 1-35 years [mean 11 years 6 months, SD 7 years 2 months]) with ONH and SOD diagnosed from 1990 to 2019, matched to 555 unrelated population-based controls (315 males, 240 females; age range 1-35 years [mean 11 years 6 months, SD 7 years 2 months]) on year of birth, sex, and area of residence. Additionally, 75 cases (46 males, 29 females; age range 2-35 years [mean 12 years 6 months, SD 7 years 2 months]) with ONH and SOD were matched one-on-one with sibling controls (40 males, 35 females; age range 0-33 years [mean 11 years 7 months, SD 7 years 10 months], the rest did not have siblings). Several antenatal maternal risk factors associated with ONH and SOD were tested for their association with case and control group membership using adjusted odds ratios (ORs) and 95% confidence intervals (CIs) from a multivariate conditional logistic regression model. The outcome was the risk of developing ONH and SOD.

RESULTS

Maternal age at conception (OR = 0.91, 95% CI = 0.86-0.96), primigravida (OR = 3.39, 95% CI = 1.92-6.01), and smoking (OR = 2.86, 95% CI = 1.61-5.05) were independently associated with ONH and SOD in the cohort matched to unrelated controls (p < 0.001). In the sibling cohort, smoking was an important risk factor (OR = 3.65, 95% CI = 1.2-11.1, p = 0.02).

INTERPRETATION

Unmodifiable and modifiable antenatal maternal risk factors are associated with ONH and SOD. Our investigation suggests that several risk factors reported in previous studies may have been due to confounding bias and that maternal smoking during pregnancy is the main modifiable risk factor associated with ONH and SOD.

WHAT THIS PAPER ADDS

Historically, many antenatal risk factors have been associated with optic nerve hypoplasia (ONH) and septo-optic dysplasia (SOD). Population-based data with matched controls for potential confounding bias are lacking. Young maternal age at conception, primigravida, and smoking during pregnancy are the main risk factors for ONH and SOD using a population-based, case-control design.

摘要

目的

确定视神经发育不全（ONH）和蝶鞍发育不全（SOD）的危险因素。

方法

本研究采用加拿大曼尼托巴省健康政策曼尼托巴中心的人群研究数据资源库，进行了一项回顾性、基于人群的病例对照设计的研究。病例组为 111 名（63 名男性，48 名女性；年龄 1-35 岁[平均 11 年 6 个月，标准差 7 年 2 个月]）从 1990 年至 2019 年确诊为 ONH 和 SOD 的患者，与 555 名无血缘关系的基于人群的对照组（315 名男性，240 名女性；年龄 1-35 岁[平均 11 年 6 个月，标准差 7 年 2 个月]）在出生年份、性别和居住地方面进行匹配。此外，75 名（46 名男性，29 名女性；年龄 2-35 岁[平均 12 年 6 个月，标准差 7 年 2 个月]）患有 ONH 和 SOD 的病例与同胞对照组（40 名男性，35 名女性；年龄 0-33 岁[平均 11 年 7 个月，标准差 7 年 10 个月]）进行一对一匹配，其余病例没有兄弟姐妹。使用多变量条件逻辑回归模型，对与 ONH 和 SOD 相关的几项产前母亲危险因素进行调整后比值比（OR）和 95%置信区间（CI）检验，以确定其与病例组和对照组成员的关联。结局是发生 ONH 和 SOD 的风险。

结果

受孕时母亲的年龄（OR=0.91，95%CI=0.86-0.96）、初产妇（OR=3.39，95%CI=1.92-6.01）和吸烟（OR=2.86，95%CI=1.61-5.05）与无血缘关系对照组的 ONH 和 SOD 独立相关（p<0.001）。在同胞队列中，吸烟是一个重要的危险因素（OR=3.65，95%CI=1.2-11.1，p=0.02）。