Uzielli M L, Guarducci S, Lapi E, Cecconi A, Ricci U, Ricotti G, Biondi C, Scarselli B, Vieri F, Scarnato P, Gori F, Sereni A
Department of Paediatrics, University of Florence, Italy.
Am J Med Genet. 1999 May 28;84(3):300-3.
Early menopause in the fragile X carriers has been well documented in several reports. All surveys demonstrated that 13-25% of fragile X carriers experienced premature ovarian failure (POF), defined as menopause before the age of 40 years. In 1995 we started screening two groups of subjects as a part of a Fragile X Research Program: 1) women previously diagnosed as fragile X carriers from the register of our center and 2) women with POF and without a family history of fragile X or other forms of mental retardation. In this study we report the preliminary data collected from 75 fragile X families; in 30 of them, POF was present in one or several subjects, all of whom had a fragile X premutation. None of the women with a full mutation experienced POF in our series of patients. We also identified 89 families without a family history of fragile X or mental retardation, and there were 108 subjects who experienced POF, of which 6.5% had a fragile X premutation. This is 70-fold higher than the background prevalence of fragile X premutation in the Italian population and suggests an association with POF. These data confirm the results of other surveys.
脆性X携带者的早期绝经在多篇报道中已有充分记录。所有调查均表明,13%至25%的脆性X携带者经历过早发性卵巢功能衰竭(POF),即40岁之前绝经。1995年,作为脆性X研究项目的一部分,我们开始对两组受试者进行筛查:1)从我们中心登记册中先前被诊断为脆性X携带者的女性;2)患有POF且无脆性X或其他形式智力障碍家族史的女性。在本研究中,我们报告了从75个脆性X家族收集的初步数据;其中30个家族中,一名或多名受试者存在POF,所有这些受试者均有脆性X前突变。在我们的一系列患者中,没有一名携带完全突变的女性经历POF。我们还识别出89个无脆性X或智力障碍家族史的家族,其中有108名受试者经历POF,其中6.5%有脆性X前突变。这比意大利人群中脆性X前突变的背景患病率高70倍,提示与POF有关联。这些数据证实了其他调查的结果。