NIH, National Institute of Child Health and Human Development, Bethesda, MD 20892, United States of America.
Children's National Medical Center, Division of Neurogenetics and Neurodevelopmental Pediatrics, Washington, DC 20010, United States of America.
Mol Genet Metab. 2020 Mar;129(3):207-212. doi: 10.1016/j.ymgme.2019.12.014. Epub 2020 Jan 10.
Hyperammonia due to ornithine transcarbamylase deficiency (OTCD) can cause a range of deficiencies in domains of executive function and working memory. Only a few fMRI studies have focused on neuroimaging data in a population with OTCD. Yet, there is a need for monitoring the disease progression and neurocognitive function in this population. In this study, we used a non-invasive neuroimaging technique, functional Near Infrared Spectroscopy (fNIRS), to examine the hemodynamics of prefrontal cortex (PFC) based on neural activation in an OTCD population. Using fNIRS, we measured the activation in PFC of the participants while performing the Stroop task. Behavioral assessment such as reaction time and correct response were recorded. We investigated the difference in behavioral measures as well as brain activation in left and right PFC in patients with OTCD and controls. Results revealed a distinction in left PFC activation between controls and patients with OTCD, where control subjects showed higher task related activation increase. Subjects with OTCD also exhibited bilateral increase in PFC activation. There was no significant difference in response time or correct response between the two groups. Our findings suggest the alterations in neurocognitive function of PFC in OTCD compared to the controls despite the behavioral profiles exhibiting no such differences. This is a first study using fNIRS to examine a neurocognitive function in OTCD population and can provide a novel insight into the screening of OTCD progression and examining neurocognitive changes.
鸟氨酸氨甲酰基转移酶缺乏症(OTCD)引起的高氨血症可导致执行功能和工作记忆领域的多种缺陷。只有少数 fMRI 研究关注 OTCD 人群的神经影像学数据。然而,需要监测该人群的疾病进展和神经认知功能。在这项研究中,我们使用了一种非侵入性的神经影像学技术——功能近红外光谱(fNIRS),根据 OTCD 人群的神经激活来检查前额叶皮层(PFC)的血液动力学。使用 fNIRS,我们在参与者执行 Stroop 任务时测量了 PFC 的激活。记录了行为评估,如反应时间和正确反应。我们研究了 OTCD 患者和对照组之间行为测量和左右 PFC 脑激活的差异。结果表明,OTCD 患者与对照组之间左 PFC 激活存在差异,对照组的任务相关激活增加更高。OTCD 患者的 PFC 激活也呈双侧增加。两组之间的反应时间或正确反应没有显著差异。我们的发现表明,OTCD 患者的 PFC 神经认知功能发生了改变,尽管行为特征没有显示出这种差异。这是首次使用 fNIRS 研究 OTCD 人群的神经认知功能,可以为 OTCD 进展的筛查和神经认知变化的检查提供新的见解。