Osipova G R, Karmanov M E, Kozlova S I, Evgrafov O V
Department of Medical Genetics, Russian Medical Academy of Postgraduate Education, Moscow.
Am J Med Genet. 1998 Apr 1;76(4):283-7.
Cytogenetic analysis of patients with Ullrich-Turner syndrome (UTS) may fail to detect low levels of Y chromosome mosaicism or Y-derived marker chromosomes. More sensitive polymerase chain reaction (PCR)-based tests have been developed; however, applicability of these data to prognosis of virilization and gonadoblastoma development has not been investigated adequately. We used a multiplex PCR-based method to detect two Y-specific sequences, SRY and AMGLY. Thirteen patients with UTS without cytogenetically detected Y chromosomes were studied. Y-specific sequences were detected in 5 patients by multiplex PCR. A cryptic translocation involving the Y chromosome was found in one patient with severe virilization of external genitalia and a male phenotype. Y chromosomal mosaicism was detected in peripheral blood and in both gonads of one patient, and only in the left gonad of another patient. Existence of a Y-derived marker was demonstrated in 2 patients, one of whom had no testicular tissue or virilization. Consistent with previous reports, we conclude that PCR is more sensitive than classical cytogenetic analysis and detects patients with Y-specific sequences in blood cells. However, the absence of Y-specific material in blood is not a sufficient reason to reject surgical treatment in case of virilization.
对乌尔里希-特纳综合征(UTS)患者进行细胞遗传学分析可能无法检测到低水平的Y染色体嵌合体或Y衍生的标记染色体。已经开发出更灵敏的基于聚合酶链反应(PCR)的检测方法;然而,这些数据对男性化和性腺母细胞瘤发生预后的适用性尚未得到充分研究。我们使用基于多重PCR的方法来检测两个Y特异性序列,即SRY和AMGLY。对13例细胞遗传学检测未发现Y染色体的UTS患者进行了研究。通过多重PCR在5例患者中检测到Y特异性序列。在1例具有严重外生殖器男性化和男性表型的患者中发现了涉及Y染色体的隐匿性易位。在1例患者的外周血和双侧性腺中检测到Y染色体嵌合体,而在另1例患者中仅在左侧性腺中检测到。在2例患者中证实存在Y衍生的标记,其中1例没有睾丸组织或男性化表现。与先前的报告一致,我们得出结论,PCR比经典细胞遗传学分析更灵敏,能够检测出血细胞中具有Y特异性序列的患者。然而,对于男性化的情况,血液中不存在Y特异性物质并不是拒绝手术治疗的充分理由。