Soares-Fernandes João P, Teixeira-Gomes Roseli, Cruz Romeu, Ribeiro Manuel, Magalhães Zita, Rocha Jaime F, Leijser Lara M
Department of Neuroradiology, Hospital de S Marcos, Largo Engenheiro Carlos Amarante, Braga 4710-965, Portugal.
Pediatr Radiol. 2008 May;38(5):559-62. doi: 10.1007/s00247-007-0721-9. Epub 2008 Jan 16.
Pyruvate dehydrogenase (PDH) deficiency is one of the most common causes of congenital lactic acidosis. Correlations between the genetic defect and neuroimaging findings are lacking. We present conventional and diffusion-weighted MRI findings in a 7-day-old male neonate with PDH deficiency due to a mosaicism for the R302H mutation in the PDHA1 gene. Corpus callosum dysgenesis, widespread increased diffusion in the white matter, and bilateral subependymal cysts were the main features. Although confirmation of PDH deficiency depends on specialized biochemical analyses, neonatal MRI plays a role in evaluating the pattern and extent of brain damage, and potentially in early diagnosis and clinical decision making.
丙酮酸脱氢酶(PDH)缺乏症是先天性乳酸性酸中毒最常见的病因之一。目前尚缺乏基因缺陷与神经影像学表现之间的相关性研究。我们报告了一名7日龄男性新生儿的常规MRI和弥散加权MRI表现,该新生儿因PDHA1基因R302H突变的嵌合体而患有PDH缺乏症。胼胝体发育不全、白质广泛弥散增加以及双侧室管膜下囊肿是主要特征。虽然PDH缺乏症的确诊依赖于专门的生化分析,但新生儿MRI在评估脑损伤的类型和程度方面发挥着作用,并且可能有助于早期诊断和临床决策。