Margari Lucia, Ventura Patrizia, Portoghese Claudia, Presicci Anna, Buttiglione Maura, Di Cuonzo Franca
Child Neurological and Psychiatric Service, Department of Neurological and Psychiatric Sciences, University of Bari, Italy.
Pediatr Neurol. 2006 Jun;34(6):467-73. doi: 10.1016/j.pediatrneurol.2005.10.014.
This report presents clinical, laboratory, and neuroimaging findings in a 7-year-old male with Sydenham's chorea associated with attention-deficit hyperactivity disorder. Western immunoblotting revealed serum anti-human basal ganglia tissue antibodies. Magnetic resonance imaging results were normal. Proton magnetic resonance spectroscopic imaging disclosed increased choline/creatine ratio in basal ganglia, frontal, and parieto-occipital areas, and decreased N-acetyl aspartate/creatine ratio in both basal ganglia and frontal areas. Moreover magnetic resonance spectroscopy revealed a peak between 3.6-4.2 ppm of unclear significance. The findings of this study are compared with the previous magnetic resonance spectroscopic studies reported on Sydenham's chorea and attention-deficit hyperactivity disorder. Magnetic spectroscopic imaging suggests an autoimmune basal ganglia damage in the pathogenesis of Sydenham's chorea and fronto-striatal impairment in attention-deficit hyperactivity disorder. In the present case, the previous history of an attention-deficit hyperactivity disorder suggests that this neurobehavioral disorder could be a risk factor for Sydenham's chorea in children with rheumatic fever.
本报告介绍了一名7岁男性患与注意力缺陷多动障碍相关的 Sydenham 舞蹈病的临床、实验室及神经影像学检查结果。蛋白质免疫印迹法显示血清中存在抗人基底节组织抗体。磁共振成像结果正常。质子磁共振波谱成像显示基底节、额叶及顶枕叶区域胆碱/肌酸比值升高,基底节和额叶区域 N-乙酰天门冬氨酸/肌酸比值降低。此外,磁共振波谱显示在3.6 - 4.2 ppm 之间有一个意义不明的峰。本研究结果与先前报道的关于 Sydenham 舞蹈病和注意力缺陷多动障碍的磁共振波谱研究结果进行了比较。磁共振波谱成像提示在 Sydenham 舞蹈病的发病机制中存在自身免疫性基底节损害,在注意力缺陷多动障碍中存在额纹状体损害。在本病例中,既往注意力缺陷多动障碍病史提示这种神经行为障碍可能是风湿热患儿患 Sydenham 舞蹈病的一个危险因素。