Singer H S, Pepple J M, Ramage A L, Butler I J
Ann Neurol. 1978 Jul;4(1):21-5. doi: 10.1002/ana.410040105.
A possible association between the Gilles de la Tourette and Lesch-Nyhan syndromes has recently been postulated. Fourteen patients with Tourette syndrome demonstrated no similarity to Lesch-Nyhan based upon patterns of inheritance, behavioral changes, or alterations of purine metabolism. Despite a strong male predominance, a sex-linked pattern of inheritance could not be confirmed. Self-mutilating behavior was found in 4 male patients but was readily differentiated from that characteristic of the Lesch-Nyhan syndrome. Quantitation of hypoxanthine-guanine phosphoribosyltransferase and isoelectric focusing of its isoenzymes produced results that were indistinguishable from those in controls. We speculate that, pathophysiologically, Tourette syndrome represents an imbalance between the central neurotransmitters dopamine and serotonin rather than an alteration in purine metabolism.
近来有人推测抽动秽语综合征(Gilles de la Tourette syndrome)与莱施-奈恩综合征(Lesch-Nyhan syndrome)之间可能存在关联。基于遗传模式、行为变化或嘌呤代谢改变,14例抽动秽语综合征患者与莱施-奈恩综合征并无相似之处。尽管男性占主导,但无法证实其为性连锁遗传模式。在4例男性患者中发现了自残行为,但很容易与莱施-奈恩综合征的特征相区分。次黄嘌呤-鸟嘌呤磷酸核糖转移酶的定量分析及其同工酶的等电聚焦结果与对照组无异。我们推测,从病理生理学角度来看,抽动秽语综合征代表着中枢神经递质多巴胺和血清素之间的失衡,而非嘌呤代谢的改变。