Okanishi Tohru, Ito Tetsuya, Nakajima Yoko, Ito Koichi, Kakita Hiroki, Yamada Yasumasa, Kobayashi Satoru, Ando Naoki, Togari Hajime
Department of Pediatrics, Neonatology and Congenital Disorders, Nagoya City University Graduate School of Medical Sciences, Mizuho-cho, Mizuho-ku, Nagoya, Aichi, Japan.
Brain Dev. 2010 Aug;32(7):567-70. doi: 10.1016/j.braindev.2009.07.001. Epub 2009 Jul 28.
Urea cycle disorders are congenital metabolic disorders that often cause episodic hyperammonemia. Neuroimaging in episodic hyperammonemia demonstrates several patterns of brain injuries, including focal lesions in the lentiform nucleus, insula, cingulate gyrus, and perirolandic fissure, as well as diffuse cerebral edema. In cases with neonatal onset of hyperammonemia, similar lesions have also been reported. We herein report a boy with severe neonatal hyperammonemia caused by ornithine transcarbamylase deficiency. He presented with parieto-occipital encephalomalacia, which resembles severe neonatal hypoglycemia on magnetic resonance imaging. This radiological finding may indicate parieto-occipital vulnerability not only to hypoglycemia but also to hyperammonemia.
尿素循环障碍是先天性代谢紊乱,常导致发作性高氨血症。发作性高氨血症的神经影像学检查显示出几种脑损伤模式,包括豆状核、岛叶、扣带回和中央沟周围裂的局灶性病变,以及弥漫性脑水肿。在新生儿期发病的高氨血症病例中,也有类似病变的报道。我们在此报告一名因鸟氨酸转氨甲酰酶缺乏导致严重新生儿高氨血症的男孩。他表现为顶枕部脑软化,在磁共振成像上类似于严重的新生儿低血糖。这一影像学发现可能表明顶枕部不仅对低血糖敏感,对高氨血症也敏感。