Infants with Congenital Adrenal Hyperplasia Exhibit Thalamic Discrepancies in Early Brain Structure.
Infants with Congenital Adrenal Hyperplasia Exhibit Thalamic Discrepancies in Early Brain Structure.
复制标题
患有先天性肾上腺增生的婴儿在早期大脑结构中表现出丘脑差异。
DOI：
10.1159/000529403
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发表时间：
2023
影响因子：
3.2
通讯作者：
Kim,MimiS
中科院分区：
文献类型：
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作者：
Rajagopalan,Vidya;Overholtzer,LloydN;Kim,WilliamS;Wisnowski,JessicaL;Pickering,TrevorA;Fraga,NicoleR;Javier,Joyce;Mackintosh,Liza;Mirzaian,Christine;Geffner,MitchellE;Kim,MimiS
简介经典先天性肾上腺增生症 (CAH) 患者存在产前和产后激素失衡。为了描述已报告的 CAH 较大儿童大脑和行为变化的个体发育特征，我们旨在研究与健康对照组相比的 CAH 婴儿的大脑结构。方法我们对 16 名因 21-羟化酶缺乏而患有经典 CAH 的婴儿（8 名男性，胎龄 38.2±1.7 周，受孕后年龄 [PCA] 42.2±3.0 周）和 14 名对照婴儿（9 名）进行了神经影像学检查。 男性，胎龄 38.5±1.8 周，PCA 42.5±2.4 周），利用 3-Tesla 磁共振成像。根据 PCA 和性别对区域脑体积进行调整，并对总脑体积 (TBV) 进行额外调整，通过回归分析进行组比较（平均值，95% 置信区间 [CI]）。通过 relaimpo 分析检查 CAH 和对照婴儿之间每个大脑区域的分化程度，并调整所有其他大脑区域、PCA 和性别。结果与年龄匹配的对照婴儿 (9,215 mm 3, 95% CI) 相比，患有 CAH 的婴儿的丘脑体积显着较小 (8,606 mm 3, 95% CI [8,209, 9,002]) [8,783, 9,647]； β=-609； p= 0.02），在进一步调整 TBV 后仍然较小。进一步调整 TBV 后，与对照婴儿 (65,616 mm 3，CI [64,680, 66,551]；β= 1,202，p= 0.03) 相比，CAH 婴儿的颞叶更大 (66,817 mm 3，CI [65,957, 67,677])。 CAH 与对照组之间差异最大的大脑区域是丘脑 (22%) 和顶叶 (10%)。 结论 患有 CAH 的婴儿在生命早期就表现出较小的丘脑区域，表明产前对 CAH 大脑发育的影响。丘脑在 8-14 周时出现，使得该区域特别容易受到宫内环境变化的影响，这对以后成熟的大脑区域具有潜在影响。这些变化可能需要一段时间才能显现出来，值得对 CAH 的青春期进行纵向研究。
IntroductionPatients with classical congenital adrenal hyperplasia (CAH) have prenatal and postnatal hormonal imbalances. To characterize the ontogeny of reported brain and behavior changes in older children with CAH, we aimed to study the brain structure in infants with CAH compared to healthy controls.MethodsWe performed neuroimaging in 16 infants with classical CAH due to 21-hydroxylase deficiency (8 males, gestational age 38.2±1.7 weeks, post-conceptional age [PCA] 42.2±3.0 weeks) and 14 control infants (9 males, gestational age 38.5±1.8 weeks, PCA 42.5±2.4 weeks) utilizing 3-Tesla magnetic resonance imaging. Regional brain volumes were adjusted for PCA and sex, along with an additional adjustment for total brain volume (TBV), for group comparisons by regression analyses (mean, 95% confidence interval [CI]). The degree to which each brain region was differentiated between CAH and control infants was examined by relaimpo analyses, adjusting for all other brain regions, PCA, and sex.ResultsInfants with CAH had significantly smaller thalamic volumes (8,606 mm 3, 95% CI [8,209, 9,002]) compared to age-matched control infants (9,215 mm 3, 95% CI [8,783, 9,647]; β=− 609; p= 0.02) which remained smaller after further adjustment for TBV. Upon further adjustment for TBV, the temporal lobe was larger in infants with CAH (66,817 mm 3, CI [65,957, 67,677]) compared to controls (65,616 mm 3, CI [64,680, 66,551]; β= 1,202, p= 0.03). The brain regions most differentiated between CAH versus controls were the thalamus (22%) and parietal lobe (10%).ConclusionsInfants with CAH exhibit smaller thalamic regions from early life, suggesting a prenatal influence on brain development in CAH. Thalamic emergence at 8–14 weeks makes the region particularly vulnerable to changes in the intrauterine environment, with potential implications for later maturing brain regions. These changes may take time to manifest, meriting longitudinal study through adolescence in CAH.