Nielsen K B, Langkjaer F
J Med Genet. 1982 Jun;19(3):222-4. doi: 10.1136/jmg.19.3.222.
A mentally retarded male patient with a structurally abnormal X chromosome is reported (karyotype 46, dir dup (X)(p11.2 leads to p21.2)Y). In the normal mother a similar X chromosome duplication was found, which was preferentially inactivated. Xg blood groups were studied in the family. The findings indicated that recombination took place at maternal meiosis, as both karyotypically normal sons and the proband were Xg(a-), the mother being Xg(a+). Functional X chromosome disomy may explain clinical abnormalities in reported patients with X duplication and a normal Y chromosome.
本文报道了一名患有结构异常X染色体的智力发育迟缓男性患者(核型为46,dir dup(X)(p11.2→p21.2)Y)。在正常母亲中发现了类似的X染色体重复,该重复优先失活。对该家族进行了Xg血型研究。结果表明,重组发生在母亲减数分裂时,因为核型正常的儿子和先证者均为Xg(a-),而母亲为Xg(a+)。功能性X染色体二体性可能解释了报道的具有X重复和正常Y染色体患者的临床异常情况。