Freund L S, Reiss A L, Hagerman R, Vinogradov S
Department of Psychiatry, Johns Hopkins University School of Medicine, Baltimore, Md.
Arch Gen Psychiatry. 1992 Jan;49(1):54-60. doi: 10.1001/archpsyc.1992.01820010054007.
The relationship between fragility (the percentage of cells exhibiting the fragile X chromosome abnormality) and psychopathological conditions was investigated in a sample of 40 obligate female carriers of the fragile X chromosome. Subjects were categorized by those with positive fragility greater than 0% (n = 19) and those with 0% fragility (n = 21). Compared with women with 0% fragility, it was expected that women with positive fragility would have a higher likelihood of manifesting a spectrum of social and psychological disability previously shown to be associated with fragile X syndrome in women. It was also expected that within the group with positive fragility, degree of fragility would be related to severity of symptoms. Results partially supported the hypotheses: women with fragility over 0% were more likely to be assigned a diagnosis of schizotypal features, were rated higher on symptoms associated with the schizophrenia spectrum, and scored lower on IQ, level of healthiest functioning, education, and socioeconomic status than women with 0% fragility. Subsequent comparisons with a control group indicated that the group with 0% fragility and normal controls did not differ on these variables. Within the group with positive fragility, increasing fragility was related to greater severity of symptoms and lower IQ, education, socioeconomic status, and levels of adaptive functioning, as predicted. Contrary to expectations, positive fragility was not associated with proportion of affective disorder diagnoses or ratings on affective disorder symptoms. The results of the study provide evidence that degree of fragility is a potentially important predictor of psychopathology among women with normal IQ who are carriers of the fragile X chromosome abnormality.
在一个由40名脆性X染色体的 obligate 女性携带者组成的样本中,研究了脆性(表现出脆性X染色体异常的细胞百分比)与精神病理状况之间的关系。受试者被分为脆性大于0%的阳性组(n = 19)和脆性为0%的组(n = 21)。与脆性为0%的女性相比,预计脆性为阳性的女性更有可能表现出一系列先前已证明与女性脆性X综合征相关的社会和心理残疾。还预计在脆性为阳性的组中,脆性程度将与症状严重程度相关。结果部分支持了这些假设:脆性超过0%的女性更有可能被诊断为分裂样特征,在与精神分裂症谱系相关的症状上评分更高,在智商、最健康功能水平、教育程度和社会经济地位方面的得分低于脆性为0%的女性。随后与对照组的比较表明,脆性为0%的组与正常对照组在这些变量上没有差异。正如预测的那样,在脆性为阳性的组中,脆性增加与症状严重程度增加以及智商、教育程度、社会经济地位和适应性功能水平降低相关。与预期相反,阳性脆性与情感障碍诊断比例或情感障碍症状评分无关。该研究结果提供了证据,表明脆性程度是智商正常的脆性X染色体异常携带者女性精神病理学的一个潜在重要预测指标。