Graceffa Anita, Russo Massimo, Vita Gian Luca, Toscano Antonio, Dattola Roberto, Messina Corrado, Vita Giuseppe, Mazzeo Anna
Department of Neurosciences, Psychiatry and Anaesthesiology, University of Messina, Messina, Italy.
Neuromuscul Disord. 2009 Jan;19(1):44-8. doi: 10.1016/j.nmd.2008.09.017. Epub 2008 Dec 11.
Presymptomatic genetic testing of an untreatable disease raises clinical, ethical, legal and psychosocial questions. Investigations in specific disorders are needed to help in understanding the motivation for and the impact of genetic testing in the lives of persons at risk for these diseases. Here, we performed a longitudinal study to investigate the psychological consequences of presymptomatic genetic testing on people at risk for transthyretin-related familial amyloidotic polyneuropathy (TTR-FAP). The aim of the present study was to provide possible guidelines for genetic counselling and psychosocial support. Impact of Event Scale Revised (IES-R), Hospital Anxiety and Depression Scale (HADS) and SF-36 questionnaires were administered to 18 asymptomatic subjects before, immediately after communication of the genetic test result and after 3, 6 and 26 months. Our findings showed evidence of anxiety, depression, avoidance of the disease, and psychological distress, especially for women, including those with a negative genetic test result ("survivor guilt"). A psychological support has to be provided before and continued at long term after presymptomatic genetic testing for TTR-FAP in people with positive result as well as in those with negative result.
对无法治疗的疾病进行症状前基因检测会引发临床、伦理、法律和社会心理问题。需要针对特定疾病进行调查,以帮助理解基因检测对这些疾病高危人群生活的动机和影响。在此,我们进行了一项纵向研究,以调查症状前基因检测对转甲状腺素蛋白相关家族性淀粉样多神经病(TTR-FAP)高危人群的心理影响。本研究的目的是为遗传咨询和社会心理支持提供可能的指导方针。在18名无症状受试者中,在基因检测结果告知前、告知后立即以及3个月、6个月和26个月后,使用事件影响量表修订版(IES-R)、医院焦虑抑郁量表(HADS)和SF-36问卷进行评估。我们的研究结果显示出焦虑、抑郁、对疾病的回避以及心理困扰的证据,尤其是女性,包括那些基因检测结果为阴性的女性(“幸存者内疚”)。对于TTR-FAP症状前基因检测结果为阳性和阴性的人群,在检测前必须提供心理支持,并在检测后长期持续提供。