Teebi A S, Miller S, Ostrer H, Eydoux P, Colomb-Brockmann C, Oudjhane K, Watters G
Division of Medical Genetics A-608, The Montreal Children's Hospital and McGill University, Quebec, Canada.
J Med Genet. 1998 Sep;35(9):759-62. doi: 10.1136/jmg.35.9.759.
Two female sibs of first cousin Iranian parents were found to have the syndrome of spastic paraplegia, optic atrophy with poor vision, microcephaly, and normal cognitive development. Karyotype analysis showed a normal female constitution in one and a male constitution (46,XY) in the other. The XY female showed normal female external genitalia, normal uterus and tubes, and streak gonads. SRY gene sequencing was normal. We conclude that the present family probably represents a new autosomal recessive trait of pleiotropic effects including XY sex reversal and adds further evidence for the heterogeneity of spastic paraplegia syndromes as well as sex reversal syndromes.
两名表亲为伊朗人的女性同胞被发现患有痉挛性截瘫、视力不佳伴视神经萎缩、小头畸形且认知发育正常的综合征。染色体核型分析显示,其中一名为正常女性核型,另一名为男性核型(46,XY)。XY女性表现出正常的女性外生殖器、正常的子宫和输卵管以及条索状性腺。SRY基因测序正常。我们得出结论,目前这个家族可能代表了一种新的具有多效性的常染色体隐性性状,包括XY性反转,这为痉挛性截瘫综合征以及性反转综合征的异质性增加了进一步的证据。