Descriptive epidemiology of cerebellar hypoplasia in the National Birth Defects Prevention Study.
Descriptive epidemiology of cerebellar hypoplasia in the National Birth Defects Prevention Study.
复制标题
国家出生缺陷预防研究中小脑发育不全的描述性流行病学。
DOI：
10.1002/bdr2.1388
复制
发表时间：
2018
影响因子：
2.1
通讯作者：
NationalBirthDefectsPreventionStudy
中科院分区：
文献类型：
--
作者：
Howley,MeredithM;Keppler-Noreuil,KimM;Cunniff,ChristopherM;Browne,MarilynL;NationalBirthDefectsPreventionStudy
背景小脑发育不全是一种罕见的小脑发育障碍，表现为小脑发育不全、小于正常大小或完全缺失。出生时小脑发育不全的患病率尚不清楚，流行病学危险因素也知之甚少。使用国家出生缺陷预防研究（NBDPS），一项基于人群的病例对照研究的数据，我们分析了非综合征性小脑发育不全的临床特征和潜在危险因素。我们描述了小脑发育不全病例的临床特征。我们通过将病例与非畸形活产对照婴儿进行比较，探讨了小脑发育不全的风险因素（病例特征、人口统计学、妊娠特征、母体健康状况、母体药物使用和母体行为暴露）。我们计算粗比值比（OR）和95%的置信区间，使用logistic回归models.ResultsWe确定了87个合格的小脑发育不全的情况下，55名母亲谁参加了NBDPS。访谈和非访谈病例之间的临床特征无差异。小脑发育不全病例比对照组更可能来自多胎妊娠、早产和低出生体重。小脑发育不全的病例更有可能出生在2005年或之后，而不是NBDPS的早期。我们发现，母亲使用血管活性药物，非西班牙裔黑人母亲，母亲与高血压病史的OR升高，没有统计学意义。ConclusionsAlthough未经调整，我们的研究结果从一个大的，以人口为基础的研究可以有助于新的假设小脑发育不全的病因。
BackgroundCerebellar hypoplasia is a rare disorder of cerebellar formation in which the cerebellum is not completely developed, smaller than it should be, or completely absent. The prevalence of cerebellar hypoplasia at birth is unknown, and little is known about epidemiological risk factors. Using data from the National Birth Defects Prevention Study (NBDPS), a population‐based, case–control study, we analyzed clinical features and potential risk factors for nonsyndromic cerebellar hypoplasia.MethodsThe NBDPS included pregnancies with estimated delivery dates from 1997–2011. We described clinical features of cerebellar hypoplasia cases from the study area. We explored risk factors for cerebellar hypoplasia (case characteristics, demographics, pregnancy characteristics, maternal health conditions, maternal medication use, and maternal behavioral exposures) by comparing cases to non‐malformed live born control infants. We calculated crude odds ratios (ORs) and 95% confidence intervals using logistic regression models.ResultsWe identified 87 eligible cerebellar hypoplasia cases and 55 mothers who participated in the NBDPS. There were no differences in clinical features between interviewed and non‐interviewed cases. Cerebellar hypoplasia cases were more likely than controls to be from a multiple pregnancy, be born preterm, and have low birth weight. Cerebellar hypoplasia cases were more likely to be born in or after 2005, as opposed to earlier in NBDPS. We found elevated ORs that were not statistically significant for maternal use of vasoactive medications, non‐Hispanic black mothers, and mothers with a history of hypertension.ConclusionsAlthough unadjusted, our findings from a large, population‐based study can contribute to new hypotheses regarding the etiology of cerebellar hypoplasia.