Department of Neurology, Boston Children's Hospital, Harvard Medical School, Boston, Massachusetts, USA.
Department of Radiology, Massachusetts General Hospital, Harvard Medical School, Boston, Massachusetts, USA.
Am J Med Genet A. 2024 Dec;194(12):e63825. doi: 10.1002/ajmg.a.63825. Epub 2024 Jul 26.
Pyruvate dehydrogenase complex deficiency (PDCD) is a mitochondrial disorder of carbohydrate oxidation characterized by lactic acidosis and central nervous system involvement. Knowledge of the affected metabolic pathways and clinical observations suggest that early initiation of the ketogenic diet may ameliorate the metabolic and neurologic course of the disease. We present a case in which first trimester ultrasound identified structural brain abnormalities prompting a prenatal molecular diagnosis of PDCD. Ketogenic diet, thiamine, and N-acetylcysteine were initiated in the perinatal period with good response, including sustained developmental progress. This case highlights the importance of a robust neurometabolic differential diagnosis for prenatally diagnosed structural anomalies and the use of prenatal molecular testing to facilitate rapid, genetically tailored intervention.
丙酮酸脱氢酶复合体缺陷症(PDCD)是一种糖氧化的线粒体疾病,其特征为乳酸酸中毒和中枢神经系统受累。对受影响的代谢途径的了解和临床观察表明,早期启动生酮饮食可能改善疾病的代谢和神经学过程。我们报告了一个病例,该病例的早孕期超声检查发现了结构性脑异常,提示产前分子诊断为 PDCD。在围产期开始使用生酮饮食、硫胺素和 N-乙酰半胱氨酸,反应良好,包括持续的发育进展。这个病例强调了为产前诊断的结构性异常建立强大的神经代谢鉴别诊断的重要性,以及利用产前分子检测来促进快速、基因定制的干预。