Finelli P F, Pueschel S M, Padre-Mendoza T, O'Brien M M
J Neurol Neurosurg Psychiatry. 1985 Feb;48(2):150-3. doi: 10.1136/jnnp.48.2.150.
We report two brothers with previously unexplained mental retardation and seizures who had dysmorphic facial features, macro-orchidism, and a fragile site at the X chromosome. This recently described syndrome is the second most common chromosome aberration associated with mental retardation after Down's syndrome. In order to determine the prevalence of seizures and the frequency of specific neurological features, we studied a total of 17 patients with the fragile X syndrome. 41% had grand mal seizures; 41% had extensor plantar responses; 47% had hyperactive behaviour and 65% exhibited stereotypics; 59% had incoordination and 35% had blepharospasm. We emphasise the need for chromosome analysis of patients with unexplained mental retardation, specific phenotypic abnormalities, and large testes.
我们报告了两兄弟,他们患有此前原因不明的智力迟钝和癫痫,面部特征畸形、巨睾症,且X染色体存在脆性位点。这种最近被描述的综合征是继唐氏综合征后与智力迟钝相关的第二常见染色体畸变。为了确定癫痫的患病率和特定神经学特征的频率,我们共研究了17例脆性X综合征患者。41%有癫痫大发作;41%有跖伸反应;47%有多动行为,65%表现出刻板动作;59%有共济失调,35%有睑痉挛。我们强调,对于原因不明的智力迟钝、特定表型异常和睾丸肿大的患者,需要进行染色体分析。