METHODS: Among 2681 children recruited for the Collaboration on Fetal Alcohol Spectrum Disorders Prevalence (CoFASP) study, 1726 had an FASD or sufficient evidence of PAE having occurred or not in their pregnancy. Children were then categorized into groups using the modified Hoyme diagnostic criteria (FAS (n = 24), pFAS (n = 99) and ARND (n = 87), and No FASD (n = 1516), including those with No FASD and a history of PAE (No FASD/PAE, n = 498) and those with No FASD and no history of PAE (No FASD/No PAE, n = 1018). The frequencies of 26 secondary dysmorphic features were compared among these groups, both individually and combined in non-weighted and weighted dysmorphic scores. Correlations of the total dysmorphic scores with an index of overall cognitive ability were also compared by group status.
RESULTS: Several of these features were significantly more frequent in children with FAS than in those with No FASD diagnosis with or without PAE but not in comparison to those with ARND. The number of features was also significantly higher in the FAS group as compared to all other groups for both weighted and unweighted dysmorphology scores but were not higher in the group with ARND when compared to the groups with No FASD either in the presence or absence of PAE. Although not diagnostic, higher total dysmorphology scores were predictive of lower general cognitive abilities in the group with ARND, suggesting severity of alcohol-related dysmorphology is predictive of severity of alcohol-related neurobehavioral impairment.
CONCLUSIONS: Secondary physical features were not more frequent in children with ARND compared to children without an FASD diagnosis but were a marker for lower cognitive function. The use of secondary physical features to support a diagnosis of ARND was not supported in this sample.
方法:在入选胎儿酒精谱系障碍患病率(CoFASP)研究的2681名儿童中,1726有FASD或足够的证据表明他们在怀孕期间发生或没有发生PAE。然后使用改良的Hoyme诊断标准将儿童分组(FAS(n=24),pFAS(n=99)和ARND(n=87),无FASD(n=1516),包括那些没有FASD和PAE历史的人(没有FASD/PAE,n=498)和无FASD且无PAE病史(无FASD/无PAE,n=1018)。在这些组中比较了26个次级异形特征的频率,无论是单独的还是组合的非加权和加权的异形分数。还按小组状态比较了总变形得分与总体认知能力指数的相关性。
结果:这些特征中的一些在FAS患儿中的频率明显高于那些没有FASD诊断且有或没有PAE的患儿,但与ARND患儿相比没有。与所有其他组相比,FAS组的加权和未加权形态学评分的特征数量也显着较高,但与存在或不存在PAE的无FASD组相比,ARND组的特征数量均不高。虽然不是诊断,较高的总形态学评分预示着ARND组的一般认知能力较低,提示酒精相关形态学的严重程度可预测酒精相关神经行为障碍的严重程度。
结论:与没有FASD诊断的儿童相比,ARND儿童的次要身体特征并不常见,但却是认知功能较低的标志。本样本不支持使用次要物理特征来支持ARND的诊断。