Dabrowski Edward, Smathers Sarah A, Ralstrom Curt S, Nigro Michael A, Leleszi Jimmie P
Division of Physical Medicine and Rehabilitation, Children's Hospital of Michigan, 3901 Beaubien Boulevard, Detroit, MI 48201, USA.
Dev Med Child Neurol. 2005 Sep;47(9):636-9.
Lesch-Nyhan syndrome (LNS) is an X-linked recessive disorder resulting from a deficiency of the metabolic enzyme hypozanthine-guanine phosphoribosyltransferase (HPRT). This syndrome presents with abnormal metabolic and neurological manifestations including hyperuricemia, mental retardation*, spastic cerebral palsy (CP), dystonia, and self-mutilation. The mechanism behind the severe self-mutilating behavior exhibited by patients with LNS is unknown and remains one of the greatest obstacles in providing care to these patients. This report describes a 10-year-old male child with confirmed LNS who was treated for self-mutilation of his hands, tongue, and lips with repeated botulinum toxin A (BTX-A) injections into the bilateral masseters. Our findings suggest that treatment with BTX-A affects both the central and peripheral nervous systems, resulting in reduced self-abusive behavior in this patient.
莱施-奈恩综合征(LNS)是一种X连锁隐性疾病,由代谢酶次黄嘌呤-鸟嘌呤磷酸核糖转移酶(HPRT)缺乏引起。该综合征表现出异常的代谢和神经学表现,包括高尿酸血症、智力发育迟缓*、痉挛性脑瘫(CP)、肌张力障碍和自残行为。LNS患者表现出的严重自残行为背后的机制尚不清楚,仍然是为这些患者提供护理的最大障碍之一。本报告描述了一名确诊为LNS的10岁男童,他因双手、舌头和嘴唇自残而接受治疗,通过向双侧咬肌反复注射肉毒杆菌毒素A(BTX-A)。我们的研究结果表明,BTX-A治疗会影响中枢神经系统和外周神经系统,从而减少该患者的自我伤害行为。