Herbst D S
Am J Med Genet. 1980;7(4):443-60. doi: 10.1002/ajmg.1320070406.
Clinical manifestations and other aspects of nonspecific X-linked mental retardation are reviewed using data from the literature and information on affected males in 24 new families ascertained in British Columbia. A great degree of variability was apparent in the mental abilities of affected males. Speech defects, other CNS disorders and minor physical changes such as "big" ears or a highly arched palate were not present in many cases. Evidence for the existence of a clinical entity of mental retardation associated with the fragile site at Xq27 or 28 and macro-orchidism is discussed. Genetic phenomena of reduced penetrance in males and of partial expression in females with respect of X-linked recessive genes are examined. Consideration is given to the question of whether this type of mental retardation is due to X-linked recessive or autosomal dominant sex-limited genes. Most ascertained cases of X-linked mental retardation are from families of northern European extraction. Recommendations are made regarding the diagnosis and counseling of X-linked mental retardation cases.
利用文献数据以及在不列颠哥伦比亚省确诊的24个新家庭中受影响男性的信息,对非特异性X连锁智力迟钝的临床表现及其他方面进行了综述。受影响男性的智力能力存在很大程度的变异性。在许多病例中不存在言语缺陷、其他中枢神经系统疾病以及诸如“大”耳朵或高拱腭等轻微身体变化。讨论了与Xq27或28处的脆性位点以及巨睾症相关的智力迟钝临床实体存在的证据。研究了X连锁隐性基因在男性中穿透率降低以及在女性中部分表达的遗传现象。考虑了这种类型的智力迟钝是由X连锁隐性基因还是常染色体显性限性基因引起的问题。大多数确诊的X连锁智力迟钝病例来自北欧血统的家庭。针对X连锁智力迟钝病例的诊断和咨询提出了建议。