Journal of Genetic Counseling

, Volume 14, Issue 3, pp 219–234 | Cite as

Negotiating Responsibility: Case Studies of Reproductive Decision-Making and Prenatal Genetic Testing in Families Facing Huntington Disease

Original Research

Abstract

Three case studies are presented to further our understanding of how responsibility is negotiated in families when making decisions about genetic risk. These draw on a model of responsibility generated in a study of reproductive decision-making in families facing Huntington disease (HD) to illustrate the impact of prenatal testing on this process. This involves analyzing: how people present themselves as acting responsibly whether or not they utilize genetic testing; who they feel responsible to in their family and elsewhere; the impact that testing has on these relationships; and, how negotiating responsibility changes over time with repeated use of prenatal testing, changing risk status and maturational changes. Two key findings are: how decision-making is perceived can become as important as what is decided; and, how responsibility is negotiated depends on which of these relationships are prioritized. Implications of the findings for clinical practice are noted and suggestions made for further applications of the model.

Key Words

Huntington disease responsibility reproductive decision-making prenatal genetic testing case studies 

Preview

Unable to display preview. Download preview PDF.

Unable to display preview. Download preview PDF.

References

  1. Cox, S. M. (Ed.). (2002). Personal perspectives on genetic testing for Huntington disease. Cambridge, Ontario: Huntington Society of Canada.Google Scholar
  2. Cox, S. M. (2003). Stories in decisions: How at risk individuals decide to request predictive testing for Huntington disease. Qual Sociol, 26, 257–280.CrossRefGoogle Scholar
  3. de Die, C., & Heurckmans, N. (2002). Case histories about preimplantation genetic diagnosis. In G. Evers-Kiebooms, M. W. Zoeteweij, & P. S. Harper (Eds.), Prenatal testing for late-onset neurogenetic diseases (pp. 119–127). Oxford: Bios Scientific Publishers Limited.Google Scholar
  4. Downing, C. (2002). Reproductive decision-making in families at risk for Huntington’s disease: Perceptions of responsibility. An unpublished dissertation submitted to the University of Cambridge for the Degree of Doctor of Philosophy.Google Scholar
  5. Evers-Kiebooms, G., Zoeteweij, M. W., & Harper, P. S. (Eds.). (2002). Prenatal testing for late-onset neurogenetic diseases. Oxford: Bios Scientific Publishers Limited.Google Scholar
  6. Finch, J., & Mason, J. (1993). Negotiating family responsibilities. London: Tavistock Routledge.Google Scholar
  7. Hallowell, N. (1999). Doing the right thing: Genetic risk and responsibility. Sociol Health Illness, 21, 597–621.CrossRefGoogle Scholar
  8. Harper, P. S. (2002). Conclusion: Prenatal testing for late-onset genetic disorders: Evidence and insights from Huntington’s disease. In G. Evers-Kiebooms, M. W. Zoeteweij, & P. S. Harper (Eds.), Prenatal testing for late-onset neurogenetic diseases (pp. 203–214). Oxford: Bios Scientific Publishers Limited.Google Scholar
  9. Harper, P. S., Quarrell, O. W. J., & Youngman, S. (1988) Huntington’s disease: Prediction and prevention. Philos Trans Roy Soc, London, B319, 285–298.Google Scholar
  10. Harper, P. S., Lim, C., & Craufurd, D. (2000). Ten years of presymptomatic testing for Huntington’s disease: The experience of the UK Huntington’s Disease Prediction Consortium. J Med Genet, 37, 567–571.CrossRefPubMedGoogle Scholar
  11. Hayden, M. R. (2000). Predictive testing for Huntington’s disease: The calm after the storm. Lancet, 356, 1944.CrossRefPubMedGoogle Scholar
  12. Huniche, L. (2003). Learning from the voiceless. New Genet Soc, 22, 257–269.CrossRefPubMedGoogle Scholar
  13. Jacopini, G., Decruyenaere, M., Harper, R., & Simpson, S. A. (2002). Case histories of prenatal testing for Huntington’s disease. In G. Evers-Kiebooms, M. W. Zoeteweij, & P. S. Harper (Eds.), Prenatal testing for late-onset neurogenetic diseases (pp. 11–24). Oxford: Bios Scientific Publishers Limited.Google Scholar
  14. Kay, E., & Kingston, H. (2002). Feelings associated with being a carrier and characteristics of reproductive decision making in women known to be carriers of X-linked conditions. J Health Psychol, 7, 169–181.CrossRefGoogle Scholar
  15. Kenen, R. (1994). The Human Genome Project: Creator of the potentially sick, potentially vulnerable and potentially stigmatized? In L. Robinson (Ed.), Life and death under high technology medicine (pp. 49–64). Manchester: Manchester University Press.Google Scholar
  16. Kenen, R. H. (1996). The at-risk health status and technology: A diagnostic invitation and the ‘gift’ of knowing. Soc Sci Med, 42, 1545–1553.CrossRefPubMedGoogle Scholar
  17. Kessler, S. (1988). Invited essay on the psychological aspects of genetic counseling: V. Preselection: A family coping strategy in Huntington disease. Am J Med Genet, 31, 617–621.PubMedGoogle Scholar
  18. Maat-Kievit, A., Vegter-van der Vlis, M., Zoeteweij, M., Losekoot, M., van Haeringen, A., Kanhai, H., et al. (1999). Experience in prenatal testing for Huntington’s disease in the The Netherlands: Procedures, results and guidelines (1987–1997). Prenat Diagn, 19, 450–457.CrossRefPubMedGoogle Scholar
  19. McConkie-Rosell, A., & Sullivan, J. A. (2003). Editorial notes. J Genet Couns, 12, 1–3.CrossRefGoogle Scholar
  20. Millan, F. A., Curtis, A., Mennie, M., Holloway, S., Boxer, M., Fard, M. J. W., et al. (1989). Prenatal exclusion testing for Huntington’s disease: A problem of too much information. J Med Genet, 26, 83–85.PubMedGoogle Scholar
  21. Nelson, H. L., & Nelson, J. L. (1995). The patient in the family: An ethics of medicine and family. London: Routledge.Google Scholar
  22. Quaid, K., & Wesson, M. K. (1995). Exploration of the effects of predictive testing for Huntington disease on intimate relationships. Am J Med Genet, 45, 46–51.CrossRefGoogle Scholar
  23. Richards, T. J., & Richards, L. (1994). Using computers in qualitative research. In N. K. Denzin & Y. S. Lincoln (Eds.), Handbook of qualitative research (pp. 445–462). Thousand Oaks: Sage Publications.Google Scholar
  24. Roche, M. I. (2003). Genetic Counseling: Myths and misconceptions. NSGC Press Release. http://www.nsgc.org/newsroom/release_myths_misconseptions.asp
  25. Robertson, A. (2000). Embodying risk, embodying political responsibility: Women’s accounts of risks for breast cancer. Health, Risk Soc, 2, 219–235.Google Scholar
  26. Sarangi, S., Bennert, K., Howell, L., Clarke, A., Harper, P., & Gray, J. (2004). Initiation of reflective frames in counseling for Huntington’s disease predictive testing. J Genet Couns, 13, 135–155.CrossRefPubMedGoogle Scholar
  27. Simpson, S. A., & Harding, A. E. (1993). Predictive testing for Huntington’s disease: After the gene. J Med Genet, 30, 1036–1038.PubMedGoogle Scholar
  28. Simpson, S. A., & Harper, P. S. (2001). Prenatal testing for Huntington’s disease: Experience within the UK 1994–1998. Letter to the Editor. J Med Genet, 38, 333–335.CrossRefPubMedGoogle Scholar
  29. Smith, J. A., Stephenson, M., & Quarrel, O. (1999). Factors influencing the decision whether to take the genetic test for Huntington’s disease: An interpretative phenomenological analysis. Unpublished paper presented at the Qualitative Methods and Genetics Group, Centre for Family Research, University of Cambridge.Google Scholar
  30. Sobel, S. K., & Cowan, D. B. (2000). Impact of genetic testing for Huntington’s disease on the family system. Am J Med Genet, 90, 49–59.CrossRefPubMedGoogle Scholar
  31. Tassiker, R., Savalescu, J., Skene, L., Marshall, P., Fitzgerald, L., & Delatycki, M. (2003). Prenatal diagnostic requests for Huntington disease where the at-risk father does not wish to know his genetic status: Clinical, legal and ethical viewpoint. Br Med J, 326, 331–333.Google Scholar
  32. Taylor, S. D. (2004). Predictive genetic test decisions for Huntington’s disease: Context, appraisal and new moral imperatives. Soc Sci Med, 58, 137–149.CrossRefPubMedGoogle Scholar

Copyright information

© Springer Science + Business Media, Inc. 2005

Authors and Affiliations

  1. 1.Centre for Family ResearchUniversity of CambridgeUnited Kingdom
  2. 2.Centre for Family ResearchUniversity of CambridgeCambridgeUnited Kingdom

Personalised recommendations