Kuo Hung-Chou, Lee Ming-Jen, Chuang Wen-Li, Huang Chin-Chang
Department of Neurology, Chang Gung Memorial Hospital and College of Medicine, University, Taipei, Taiwan.
J Neurol Sci. 2007 Sep 15;260(1-2):231-5. doi: 10.1016/j.jns.2007.03.018. Epub 2007 Apr 24.
We report a patient with acute intermittent porphyria who presented with progressive motor neuropathy, particularly in the upper limbs. The electrophysiological studies showed an asymmetric motor neuropathy with a prominent involvement of both the radial and left peroneal nerves. During the 1-year follow-up period, 6 courses of hematin infusion, with 150 mg daily for 4 consecutive days every month, were administrated. The motor neuropathy showed a steady and gradual improvement following the hematin treatment. Molecular analysis of the porphobilinogen deaminase gene revealed a short segment deletion (1008-1019delCAGCCTGGCCAA) resulting in a truncated protein. The findings suggest that early hematin treatment is temporally associated with interval improvement of the patient's porphyric motor neuropathy.
我们报告了一名急性间歇性卟啉病患者,该患者出现进行性运动神经病,尤其是在上肢。电生理研究显示为不对称性运动神经病,桡神经和左侧腓总神经明显受累。在1年的随访期内,每月连续4天每天给予150mg的血红素输注,共进行了6个疗程。血红素治疗后,运动神经病呈稳定且逐渐改善。对胆色素原脱氨酶基因的分子分析显示有一个短片段缺失(1008 - 1019delCAGCCTGGCCAA),导致产生截短蛋白。这些发现表明早期血红素治疗与患者卟啉性运动神经病的间歇性改善在时间上相关。