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神经纤维瘤病 1 型小儿脑瘤的围生期危险因素。

Peri-gestational risk factors for pediatric brain tumors in Neurofibromatosis Type 1.

机构信息

Brown School, Washington University, St. Louis, MO 63130, United States.

Brown School, Washington University, St. Louis, MO 63130, United States.

出版信息

Cancer Epidemiol. 2016 Jun;42:53-9. doi: 10.1016/j.canep.2016.03.005. Epub 2016 Mar 25.

Abstract

BACKGROUND

Individuals with Neurofibromatosis Type 1 (NF1) are strongly predisposed to developing pediatric brain tumors (PBTs), especially optic pathway gliomas (OPGs). Although developmental factors have been implicated in the origins of PBTs in both human and animal studies, associations between early-life factors and PBTs have not been evaluated in individuals with NF1. Our objective was to evaluate associations between peri-gestational characteristics and PBTs in this population.

METHODS

We conducted a cross-sectional study, ascertaining questionnaire and medical record data for 606 individuals<18years old who enrolled in the NF1 Patient Registry Initiative (NPRI) from 6/9/2011-6/29/2015. One hundred eighty-four individuals had reported PBT diagnoses, including 65 who were identified with OPG diagnoses. Cox proportional hazards regression was used to calculate hazard ratios (HRs) and 95% confidence intervals (CIs) for associations between PBT and OPG diagnoses and peri-gestational characteristics (prematurity, birth weight, parental age, plurality, family history of NF1, assisted reproductive technology, maternal vitamin supplementation, and parental smoking).

RESULTS

We observed no significant associations between any of the assessed characteristics and PBTs overall or OPGs with the exception of birth weight. After controlling for potential confounding variables, we observed a significant positive association between birth weight quartile and OPGs with a HR of 3.32 (95% CI 1.39-7.94) for the fourth (≥3915.5g) compared to the first (≤3020g) quartile (p for trend=0.001).

CONCLUSIONS

Consistent with results for PBTs in the general population, these results suggest that higher birth weights increase OPG risk in individuals with NF1.

摘要

背景

患有神经纤维瘤病 1 型(NF1)的个体极易患上小儿脑肿瘤(PBT),尤其是视神经胶质瘤(OPG)。虽然在人类和动物研究中都表明发育因素与 PBT 的起源有关,但 NF1 患者的围生期因素与 PBT 之间的关联尚未得到评估。我们的目的是评估该人群中围生期特征与 PBT 之间的关系。

方法

我们进行了一项横断面研究,通过 NF1 患者登记倡议(NPRI),在 2011 年 6 月 9 日至 2015 年 6 月 29 日期间,对 606 名年龄<18 岁的个体进行问卷调查和病历数据的采集。其中 184 名个体报告了 PBT 诊断,包括 65 名患有 OPG 诊断的个体。使用 Cox 比例风险回归计算 PBT 和 OPG 诊断与围生期特征(早产、出生体重、父母年龄、多胎、NF1 家族史、辅助生殖技术、母亲维生素补充剂和父母吸烟)之间的风险比(HR)和 95%置信区间(CI)。

结果

除了出生体重,我们没有观察到任何评估特征与 PBT 或 OPG 之间的显著关联。在控制潜在混杂因素后,我们观察到出生体重四分位数与 OPG 之间存在显著的正相关,与第一四分位数(≤3020g)相比,第四四分位数(≥3915.5g)的 HR 为 3.32(95%CI 1.39-7.94)(趋势检验 p 值=0.001)。

结论

与一般人群中 PBT 的结果一致,这些结果表明,较高的出生体重会增加 NF1 个体中 OPG 的风险。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7b09/4899111/769cda4d24e3/nihms-768502-f0001.jpg

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