Bukhanovskiĭ A O, Kit'ian V A
Zh Nevropatol Psikhiatr Im S S Korsakova. 1983;83(5):688-92.
Clinicogenetic assessment of a family whose six members proved to be carriers of an identical pathological trait allowed the delineation of the clinical picture of the hereditary disease described. Its major components include congenital cranial and facial abnormalities, malformed extremities characteristic of acrocephalosyndactyly, and a progressive pattern of psychic disturbances and higher cortical dysfunctions arising in old age. The first to come are increasing acoustic agnosia and disconnected delusions (stage of psychotic disorders) which are followed by progressive overall mental retardation with concomitant deactualization and disappearance of delusional feelings (stage of dementia). It has been suggested that cases of this kind make up a new (viz., the sixth) type of acrocephalosyndactyly with an autosomal dominant pattern of hereditary transmission. The mutant gene appears to be pleiotropic and characterized by complete penetrance and slightly varying expressivity.
对一个有六名成员被证实携带相同病理特征的家庭进行临床遗传学评估,从而勾勒出了所描述的遗传性疾病的临床症状。其主要组成部分包括先天性颅面异常、尖头并指畸形所特有的肢体畸形,以及老年时出现的进行性精神障碍和高级皮层功能障碍模式。首先出现的是听觉失认症加重和分离性妄想(精神障碍阶段),随后是进行性全面智力迟钝,同时妄想情绪减退和消失(痴呆阶段)。有人提出,这类病例构成了一种新的(即第六种)尖头并指畸形,具有常染色体显性遗传模式。突变基因似乎具有多效性,其特征是完全显性和表达略有差异。