Reduced pituitary volume with relative T1 shortening correlates with behavior in Prader-Willi syndrome

Reduced pituitary volume with relative T1 shortening correlates with behavior in Prader-Willi syndrome
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垂体体积减少和相对 T1 缩短与普瑞德威利综合征的行为相关

DOI:
10.1016/j.bionps.2021.100039
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发表时间:
2021
影响因子:
--
通讯作者:
Watanabe M
Watanabe M
中科院分区:
--
文献类型:
--
作者:
Yamada K;Suzuki K;Watanabe M

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praper - willi综合征是一种复杂的内分泌和发育障碍,其特征为贪食、自闭症和强迫行为,被认为主要源于大脑下丘脑-垂体轴系统的改变。虽然脑垂体已被证明与神经肽(如精氨酸-加压素)相关的行为表型有关,但Prader-Willi综合征的相关改变尚不清楚。本研究旨在探讨垂体结构的发育异常,并确定结构异常是否与Prader-Willi综合征的行为特征有关。总共包括21名日本普瑞德-威利综合征患者和31名典型发育的健康对照者。与对照组相比,Prader-Willi综合征组显示垂体前、后容积比/总颅内容积降低,t1相对缩短,且与年龄相关。此外,改变的体积比和信号强度与贪食症和自闭症问卷得分呈负相关,而与强迫得分呈正相关。研究结果表明,结构和功能的改变,部分是由于下丘脑-垂体功能的改变,可能导致普瑞德-威利综合征的行为。脑垂体的成像-行为相关性和体内神经化学可视化可能是Prader-Willi综合征和其他神经发育障碍行为表型的潜在内在生物标志物。
Prader-Willi syndrome is a complex endocrinological and developmental disorder characterized by hyperphagic, autistic, and obsessive behaviors, which have been considered to primarily originate from hypothalamus-pituitary axis system alterations in the brain. While the pituitary gland has been demonstrated to contribute to behavioral phenotypes associated with neuropeptide, e.g., arginine-vasopressin, the relevant alterations in Prader-Willi syndrome remain unknown. This study aimed to investigate developmental abnormalities in the pituitary gland structures and determine whether the structural abnormalities are associated with behavioral characteristics in Prader-Willi syndrome. In total, 21 Japanese individuals with Prader-Willi syndrome and 31 healthy controls with typical development were included. Compared with the control group, the Prader-Willi syndrome group showed reduced anterior and posterior pituitary volume ratios per total intracranial volume with relative T1shortening in an age-associated manner. Moreover, altered volume ratios and signal intensities were negatively correlated with hyperphagia and autistic questionnaire scores but positively correlated with obsessive scores. The findings suggest that structural and functional alterations, in part due to altered hypothalamus-pituitary function, may contribute to the behavior in Prader-Willi syndrome. The imaging-behavior correlates and in vivo neurochemical visualization of the pituitary gland might be potential intrinsic biomarkers for behavioral phenotypes in Prader-Willi syndrome and other neurodevelopmental disorders.
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