Abstract
A total of 135 participants in the Canadian predictive testing programme for HD were followed for at least one year in one of four study groups: increased risk (n = 37), decreased risk ( n = 58), uninformative (n = 17), or not tested (n = 23). Clinical criteria for an adverse event were a suicide attempt or formulation of a suicide attempt plan, psychiatric hospitalisation, depression lasting longer than two months, a marked increase in substance abuse, and the breakdown of important relationships. Quantitative criteria, as measured by changes on the General Severity Index of the Symptom Checklist 90-R and the Beck Depression Inventory, were also used to identify people who had adverse events. Twenty of the 135 participants (14.8%) had an adverse event. There were no significant differences between those with or without an adverse event with respect to age, sex, marital status, education, psychiatric history, general psychiatric distress, or social supports at baseline. However, evidence for depression was associated with an increased frequency of adverse events (p < 0.04). The adverse events were similar and seen with equivalent frequency in those receiving an increased risk or decreased risk and persons at risk who did not receive a modification of risk. However, a significant difference was found in the timing of adverse events for the increased and decreased risk groups (p < 0.0002). In the increased risk group all of the adverse events occurred within 10 days after results whereas, in the decreased risk group, all of the adverse events occurred six months or later after reviewing test results. These results suggest that people entering into predictive testing with some evidence of clinical depression warrant special vigilance and also suggest that counselling and support should be available for all participants in predictive testing irrespective of the direction of test results.
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Selected References
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- Bloch M., Adam S., Wiggins S., Huggins M., Hayden M. R. Predictive testing for Huntington disease in Canada: the experience of those receiving an increased risk. Am J Med Genet. 1992 Feb 15;42(4):499–507. doi: 10.1002/ajmg.1320420416. [DOI] [PubMed] [Google Scholar]
- Brandt J., Quaid K. A., Folstein S. E., Garber P., Maestri N. E., Abbott M. H., Slavney P. R., Franz M. L., Kasch L., Kazazian H. H., Jr Presymptomatic diagnosis of delayed-onset disease with linked DNA markers. The experience in Huntington's disease. JAMA. 1989 Jun 2;261(21):3108–3114. [PubMed] [Google Scholar]
- Codori A. M., Brandt J. Psychological costs and benefits of predictive testing for Huntington's disease. Am J Med Genet. 1994 Sep 15;54(3):174–184. doi: 10.1002/ajmg.1320540304. [DOI] [PubMed] [Google Scholar]
- Codori A. M., Hanson R., Brandt J. Self-selection in predictive testing for Huntington's disease. Am J Med Genet. 1994 Sep 15;54(3):167–173. doi: 10.1002/ajmg.1320540303. [DOI] [PubMed] [Google Scholar]
- Copley T. T., Wiggins S., Dufrasne S., Bloch M., Adam S., McKellin W., Hayden M. R. Are we all of one mind? Clinicians' and patients' opinions regarding the development of a service protocol for predictive testing for Huntington disease. Canadian Collaborative Study for Predictive Testing for Huntington Disease. Am J Med Genet. 1995 Jul 31;58(1):59–69. doi: 10.1002/ajmg.1320580113. [DOI] [PubMed] [Google Scholar]
- Evers-Kiebooms G., Swerts A., Cassiman J. J., Van den Berghe H. The motivation of at-risk individuals and their partners in deciding for or against predictive testing for Huntington's disease. Clin Genet. 1989 Jan;35(1):29–40. doi: 10.1111/j.1399-0004.1989.tb02902.x. [DOI] [PubMed] [Google Scholar]
- Farrer L. A. Suicide and attempted suicide in Huntington disease: implications for preclinical testing of persons at risk. Am J Med Genet. 1986 Jun;24(2):305–311. doi: 10.1002/ajmg.1320240211. [DOI] [PubMed] [Google Scholar]
- Fox S., Bloch M., Fahy M., Hayden M. R. Predictive testing for Huntington disease: I. Description of a pilot project in British Columbia. Am J Med Genet. 1989 Feb;32(2):211–216. doi: 10.1002/ajmg.1320320214. [DOI] [PubMed] [Google Scholar]
- Huggins M., Bloch M., Wiggins S., Adam S., Suchowersky O., Trew M., Klimek M., Greenberg C. R., Eleff M., Thompson L. P. Predictive testing for Huntington disease in Canada: adverse effects and unexpected results in those receiving a decreased risk. Am J Med Genet. 1992 Feb 15;42(4):508–515. doi: 10.1002/ajmg.1320420417. [DOI] [PubMed] [Google Scholar]
- Kessler S., Field T., Worth L., Mosbarger H. Attitudes of persons at risk for Huntington disease toward predictive testing. Am J Med Genet. 1987 Feb;26(2):259–270. doi: 10.1002/ajmg.1320260204. [DOI] [PubMed] [Google Scholar]
- Kessler S. Predictive testing for Huntington disease: a psychologist's view. Am J Med Genet. 1994 Sep 15;54(3):161–166. doi: 10.1002/ajmg.1320540302. [DOI] [PubMed] [Google Scholar]
- Kessler S. Psychiatric implications of presymptomatic testing for Huntington's disease. Am J Orthopsychiatry. 1987 Apr;57(2):212–219. doi: 10.1111/j.1939-0025.1987.tb03531.x. [DOI] [PubMed] [Google Scholar]
- Mastromauro C., Myers R. H., Berkman B. Attitudes toward presymptomatic testing in Huntington disease. Am J Med Genet. 1987 Feb;26(2):271–282. doi: 10.1002/ajmg.1320260205. [DOI] [PubMed] [Google Scholar]
- Quaid K. A., Wesson M. K. Exploration of the effects of predictive testing for Huntington disease on intimate relationships. Am J Med Genet. 1995 May 22;57(1):46–51. doi: 10.1002/ajmg.1320570111. [DOI] [PubMed] [Google Scholar]
- Tibben A., Duivenvoorden H. J., Niermeijer M. F., Vegter-van der Vlis M., Roos R. A., Verhage F. Psychological effects of presymptomatic DNA testing for Huntington's disease in the Dutch program. Psychosom Med. 1994 Nov-Dec;56(6):526–532. doi: 10.1097/00006842-199411000-00008. [DOI] [PubMed] [Google Scholar]
- Tibben A., Frets P. G., van de Kamp J. J., Niermeijer M. F., Vegtervan der Vlis M., Roos R. A., Rooymans H. G., van Ommen G. J., Verhage F. On attitudes and appreciation 6 months after predictive DNA testing for Huntington disease in the Dutch program. Am J Med Genet. 1993 Jul 15;48(2):103–111. doi: 10.1002/ajmg.1320480209. [DOI] [PubMed] [Google Scholar]
- Wiggins S., Whyte P., Huggins M., Adam S., Theilmann J., Bloch M., Sheps S. B., Schechter M. T., Hayden M. R. The psychological consequences of predictive testing for Huntington's disease. Canadian Collaborative Study of Predictive Testing. N Engl J Med. 1992 Nov 12;327(20):1401–1405. doi: 10.1056/NEJM199211123272001. [DOI] [PubMed] [Google Scholar]
- Wolff G., Walter W. Attitudes of at-risk persons for Huntington disease toward predictive genetic testing. Birth Defects Orig Artic Ser. 1992;28(1):119–126. [PubMed] [Google Scholar]
