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HMG-CoA裂解酶缺乏症中的磁共振成像(MRI)和磁共振波谱分析(MRS)

MRI and MRS in HMG-CoA lyase deficiency.

作者信息

Yalçinkaya C, Dinçer A, Gündüz E, Fiçicioğlu C, Koçer N, Aydin A

机构信息

Department of Neurology, Istanbul University, Cerrahpaşa Medical Faculty, Turkey.

出版信息

Pediatr Neurol. 1999 May;20(5):375-80. doi: 10.1016/s0887-8994(99)00013-2.

Abstract

3-Hydroxy-3-Methylglutaryl coenzyme A lyase (HMG-CoA) deficiency is a rare inborn error of leucine catabolism. The disease is characterized by recurrent episodes of metabolic acidosis, hyperammonemia without ketosis, hypoglycemia, lethargy, hepatomegaly, and seizures. This study has evaluated the magnetic resonance imaging (MRI) and magnetic resonance spectroscopy (MRS) findings of three patients with HMG-CoA deficiency. The common findings on all of the MRI scans were multiple, coalescent, marked lesions in periventricular white matter and arcuate fibers, most prominently in frontal or periatrial regions that were superimposed on diffuse, slightly hyperintense subcortical white matter signal. Involvement of the caudate nucleus and the dentate nucleus were observed in the reported patients. MRS studies by both STEAM and PRESS spectra of all patients revealed a decrease in N-acetylaspartate and elevation in both myoinositol and choline. A pathologic peak at 1.33 ppm, which is compatible with lactate, and a particular peak at 2.42 ppm in all patients were also found. The combination of both MRI and MRS findings could be considered as being specific in patients with HMG-CoA lyase deficiency.

摘要

3-羟基-3-甲基戊二酰辅酶A裂解酶(HMG-CoA)缺乏症是一种罕见的亮氨酸分解代谢先天性缺陷疾病。该疾病的特征为代谢性酸中毒反复发作、无酮血症的高氨血症、低血糖、嗜睡、肝肿大和癫痫发作。本研究评估了3例HMG-CoA缺乏症患者的磁共振成像(MRI)和磁共振波谱(MRS)表现。所有MRI扫描的常见表现为脑室周围白质和弓形纤维多发、融合、明显的病变,最显著位于额叶或心房周围区域,叠加在弥漫性、轻度高信号的皮质下白质信号上。在所报道的患者中观察到尾状核和齿状核受累。所有患者通过单体素点分辨波谱(PRESS)和刺激回波采集模式(STEAM)波谱进行的MRS研究均显示N-乙酰天门冬氨酸减少,肌醇和胆碱升高。在所有患者中还发现了与乳酸相符的1.33 ppm处的病理性峰以及2.42 ppm处的特定峰。MRI和MRS表现的组合可被认为对HMG-CoA裂解酶缺乏症患者具有特异性。

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