Kasprzak L, Der Kaloustian V M, Elliott A M, Shevell M, Lejtenyi C, Eydoux P
Division of Medical Genetics, Montreal Children's Hospital, Quebec, Canada.
Am J Med Genet. 1998 Jan 23;75(3):288-91.
We have studied two brothers with submicroscopic 22q11 deletion. One brother had findings suggestive of DiGeorge syndrome, while the other had milder anomalies, including polydactyly. Fluorescence in situ hybridization (FISH) showed a minor cell line with deletion 22q11 in the mother. To our knowledge, this is the first report of a deletion of 22q11 in two sibs with different phenotypes and apparent maternal mosaicism detected with FISH. This family illustrates the variability of the syndrome and further demonstrates the possibility of gonadal mosaicism for a microdeletion. Prenatal diagnosis may be offered after the birth of a child with a 22q11 deletion, even in the absence of parental chromosomal anomalies.
我们研究了两名患有亚微观22q11缺失的兄弟。其中一名兄弟有提示迪乔治综合征的表现,而另一名兄弟有较轻微的异常,包括多指畸形。荧光原位杂交(FISH)显示母亲存在一个带有22q11缺失的小细胞系。据我们所知,这是首次报道两个具有不同表型的同胞出现22q11缺失且通过FISH检测到明显的母源嵌合现象。这个家族说明了该综合征的变异性,并进一步证明了微缺失性腺嵌合的可能性。即使父母染色体无异常,在患有22q11缺失的孩子出生后也可进行产前诊断。