Maraschio P, Tupler R, Barbierato L, Dainotti E, Larizza D, Bernardi F, Hoeller H, Garau A, Tiepolo L
Biologia Generale e Genetica Medica, Università di Pavia, Italy.
Hum Genet. 1996 Mar;97(3):375-81. doi: 10.1007/BF02185777.
We characterized by fluorescence in situ hybridization and Southern blotting 14 partial Xq monosomies, 11 due to terminal deletions and 3 secondary to X/autosome translocations. Three cases were mosaics with a XO cell line. In view of the possible role played by telomeres in chromosome segregation, we hypothesize a relationship between the loss of telomeric sequences in terminal deletions and the presence of 45,X cells. A correlation between phenotype and extent of deletion reveal that there is no correspondence between the size of the deletion and impairment of gonadal function. Turner stigmata are absent in patients without an XO cell line, when the breakpoint is distal to Xq24. A low birthweight is present whenever the breakpoint is at q22 or more proximal.
我们通过荧光原位杂交和Southern印迹法对14例部分Xq单体进行了特征分析,其中11例由于末端缺失,3例继发于X/常染色体易位。3例为具有XO细胞系的嵌合体。鉴于端粒在染色体分离中可能发挥的作用,我们推测末端缺失中端粒序列的丢失与45,X细胞的存在之间存在关联。表型与缺失范围之间的相关性表明,缺失大小与性腺功能损害之间不存在对应关系。当断点位于Xq24远端时,没有XO细胞系的患者不存在特纳体征。只要断点位于q22或更靠近近端,就会出现低出生体重。