McPherson E W, Laneri G, Clemens M M, Kochmar S J, Surti U
Department of Genetics, Magee-Womens Hospital, Pittsburgh, PA 15213, USA.
Am J Med Genet. 1997 Sep 5;71(4):430-3. doi: 10.1002/(sici)1096-8628(19970905)71:4<430::aid-ajmg11>3.0.co;2-h.
Coffin-Siris syndrome is a multiple anomaly/mental retardation syndrome characterized by "coarse" facial appearance, hypoplastic or absent nails on the fifth digits, generalized hirsutism with sparse scalp hair, hypotonia, and developmental delay. Due to several reports of affected sibs with or without a mildly affected parent, both autosomal recessive and autosomal dominant inheritance have been suggested. All previous patients with well-documented Coffin-Siris syndrome are chromosomally normal, and the gene has not been mapped. We report on an infant with typical findings of Coffin-Siris syndrome who also has a de novo apparently balanced translocation of chromosomes 1 and 7, karyotype 46,XY,t(1;7)(q21.3;q34). The parental chromosomes are normal and none of the relatives have signs of Coffin-Siris syndrome. The breakpoints 1q21.3 and 7q34 are suggested as possible locations for a Coffin-Siris gene.
科芬-西里斯综合征是一种多发畸形/智力发育迟缓综合征,其特征为面部外观“粗糙”、第五指指甲发育不全或缺失、全身性多毛且头皮毛发稀疏、肌张力减退以及发育迟缓。由于有几例关于患病同胞的报道,有或没有轻度患病的父母,因此有人提出了常染色体隐性遗传和常染色体显性遗传两种方式。之前所有有充分记录的科芬-西里斯综合征患者染色体均正常,且该基因尚未定位。我们报告了一名患有典型科芬-西里斯综合征表现的婴儿,其还存在1号和7号染色体的新发明显平衡易位,核型为46,XY,t(1;7)(q21.3;q34)。父母的染色体正常,且没有亲属有科芬-西里斯综合征的体征。1q21.3和7q34断点被认为可能是科芬-西里斯基因的位置。