Gonzalez Carlos, Gomes Elisabete, Kazachkova Nadiya, Bettencourt Conceição, Raposo Mafalda, Kay Teresa Taylor, MacLeod Patrick, Vasconcelos João, Lima Manuela
Department of Clinical Psychology, Hospital Divino Espírito Santo, Ponta Delgada, Portugal.
Genet Test Mol Biomarkers. 2012 Dec;16(12):1363-8. doi: 10.1089/gtmb.2011.0370.
The present study on long-term outcome of presymptomatic testing for Machado-Joseph disease (MJD) aimed to evaluate the psychological well-being and the familial satisfaction of subjects that 5 years prior received an unfavorable result in the predictive testing (PT). The study included 47 testees of Azorean origin (23 from the island of Flores and 24 from S. Miguel) that completed the fourth evaluation session of the MJD protocol, and undertook a neurological examination at the moment of participation in the study. Nearly 50% of testees were symptomatic at the time of the study. Psychological well-being of the 47 participants was evaluated using the Psychological General Well-Being Index (PGWB). The family satisfaction scale by adjectives was applied to obtain information on family dynamics. The average PGWB score of the total participants was of 73.3, a value indicative of psychological well-being. Nearly half of the testees presented scores indicating psychological well-being, whereas scores indicating moderate (28.9%) or severe (23.7%) stress were found in the remaining. The average score in the PGWB scale was lower in symptomatic than in asymptomatic subjects; moreover, the distinct distribution of the well-being categories seen in the two groups shows an impact of the appearance of first symptoms on the psychological state. Motives for undertaking the test, provided 5 years prior, failed to show an impact in well-being. The average score for familial satisfaction was of 134, a value compatible with high familial satisfaction, which represented the most frequent category (59.6%). Results demonstrate that well-being and family satisfaction need to be monitored in confirmed carriers of the MJD mutation. The inclusion of acceptance studies, after PT, as well as the development of acceptance training actions, should be of major importance to anticipate the possibility of psychological damage.
本项关于马查多-约瑟夫病(MJD)症状前检测长期结果的研究旨在评估那些在5年前预测性检测(PT)中得到不利结果的受试者的心理健康状况和家庭满意度。该研究纳入了47名亚速尔群岛血统的受测者(23名来自弗洛雷斯岛,24名来自圣米格尔岛),他们完成了MJD方案的第四次评估环节,并在参与研究时接受了神经学检查。在研究时,近50%的受测者出现了症状。使用心理总体幸福感指数(PGWB)对47名参与者的心理健康状况进行了评估。应用形容词家庭满意度量表以获取有关家庭动态的信息。所有参与者的PGWB平均得分为73.3,这一数值表明心理健康状况良好。近一半的受测者得分表明心理健康状况良好,而其余受测者中出现了表明中度(28.9%)或重度(23.7%)压力的得分。有症状的受试者在PGWB量表上的平均得分低于无症状受试者;此外,两组中幸福感类别分布的差异表明首次症状的出现对心理状态有影响。5年前进行检测的动机并未对幸福感产生影响。家庭满意度的平均得分为134,这一数值与高家庭满意度相符,代表了最常见的类别(59.6%)。结果表明,对于确诊的MJD突变携带者,需要监测其幸福感和家庭满意度。在PT之后纳入接受度研究以及开展接受度培训行动,对于预测心理损伤的可能性应具有重要意义。