de Sousa C, Piesowicz A T, Brett E M, Leonard J V
Queen Mary's Hospital for Children, Carshalton, Surrey, Great Britain.
Neuropediatrics. 1989 Nov;20(4):199-201. doi: 10.1055/s-2008-1071292.
Neurological abnormalities are common in patients with organic acidaemias and sometimes these are distinctive. Lesions in various parts of the brain are increasingly being recognised in patients with methylmalonic acidaemia. We present the cases of two patients with methylmalonic acidaemia who became acutely ill with marked metabolic acidosis. CT scan showed bilateral areas of low density involving the globi pallidi. One patient developed dystonic posturing of the upper and lower limbs with rigidity and bradykinesia following her recovery. Treatment with benzhexol and with levodopa and carbidopa had no sustained benefit. Her gait slowly improved but her dystonic posturing remained. The other patient developed marked truncal hypotonia and a variable increase in limb tone following the acute illness. These abnormalities have persisted. It is concluded that structural brain changes, and in particular symmetrical lesions in the globi pallidi, may follow acute illness in patients with methylmalonic acidaemia and be accompanied by neurological symptoms.
神经学异常在患有有机酸血症的患者中很常见,有时这些异常很独特。甲基丙二酸血症患者中,大脑各部位的病变越来越受到认可。我们报告了两名患有甲基丙二酸血症的患者,他们因明显的代谢性酸中毒而病情急性加重。CT扫描显示双侧苍白球低密度区。一名患者康复后出现上肢和下肢肌张力障碍姿势,伴有僵硬和运动迟缓。使用苯海索以及左旋多巴和卡比多巴治疗均未获得持续疗效。她的步态逐渐改善,但肌张力障碍姿势仍然存在。另一名患者在急性病后出现明显的躯干肌张力减退,肢体肌张力有不同程度增加。这些异常情况持续存在。得出的结论是,甲基丙二酸血症患者在急性病后可能会出现脑部结构改变,尤其是苍白球对称性病变,并伴有神经学症状。