The motivation of at‐risk individuals and their partners in deciding for or against predictive testing for Huntington's disease

Abstract

Sixty‐six percent of the at‐risk persons and 74% of the partners in a large survey in Belgium have the intention of making use of predictive testing for Huntington's disease. One third of them, however, have expressed the intention of postponing the final decision for various reasons. The intention to be tested is not at all related to sociodemographic characteristics. A thorough exploration of the reasons for being in favour of or against taking the test reveals that the motivation inspiring this very personal decision is very complex. In the group of at‐risk persons, less than half of the variation in the intention to be tested is explained by the role of a series of specific reasons as predictor variables in a regression analysis. The proportion of explained variation is slightly higher in the group of partners. ‘To have certainty about my own future’ and ‘to make arrangements for the future’ play a major part in the decision of the total group. ‘Making decisions concerning children’ and to a larger extent ‘informing children about their risk status’ are important factors in deciding in favour of the test.

Predictive testing for Huntington disease: interpretation and significance of intermediate alleles

“…Furthermore, attention is required to document the specific needs of patients and medical genetics professionals in relation to intermediate alleles. Previous research has indicated one reason that individuals choose to undergo predictive testing for HD is to relieve uncertainty (55)(56)(57). Based on the results of the pilot study, when predictive testing identifies an intermediate allele, there appears to be an increase in uncertainty, not a reduction.…”

A review of quality of life after predictive testing for and earlier identification of neurodegenerative diseases

“…Surveys administered prior to the availability of genetic testing suggested that 56-81% of at-risk individuals would take advantage of a predictive test (Evers-Kiebooms et al, 1989; Kessler et al, 1987; Koller and Davenport, 1984; Markel et al, 1987; Mastromauro et al, 1987; Meissen and Berchek, 1988; Tyler and Harper, 1983). Twenty years after genetic testing was made available, uptake remained at 10-20% (Babul et al, 1993; Craufurd et al, 1989; Quaid and Morris, 1993), though some recent reports suggest that uptake is increasing (Sizer et al, 2012).…”

Psychological Impact of Predictive Genetic Testing in VCP Inclusion Body Myopathy, Paget Disease of Bone and Frontotemporal Dementia

“…Reasons for choosing to pursue genetic testing included the desire to end uncertainty, preparing for the future, possible utility in making reproductive decisions and informing children about risk (Tibben, 2007). Reasons given for declining testing included the desire to maintain hope, the concerns about their ability to cope with adverse results and to limit insurance and employment discrimination (Babul et al, 1993; Dufrasne, Roy, Galvez, & Rosenblatt, 2011; Evers-Kiebooms, Swerts, Cassiman, & Van den Berghe, 1989; Hagberg, Bui, & Winnberg, 2011; Quaid et al, 2008). There was some evidence to suggest that those who proceeded with testing were self-selected and mentally resourceful (Codori, Hanson, & Brandt, 1994; Decruyenaere et al, 1995; Kessler, 1994; Tyler, Ball, & Craufurd, 1992).…”