The use of haematopoietic stem cell transplantation in Fanconi anaemia patients: A survey of decision making among families in the US and Canada

Sadie P. Hutson, Paul K J Han, Jada G. Hamilton, Sean C. Rife, Mohamad M. Al-Rahawan, Richard P. Moser, Seth P. Duty, Sheeba Anand, Blanche P. Alter

Research output: Contribution to journalArticle

Abstract

Background: Fanconi anaemia (FA) is a rare genetic disorder associated with bone marrow failure (BMF), congenital anomalies and cancer susceptibility. Stem cell transplantation (SCT) offers a potential cure for BMF or leukaemia, but incurs substantial risks. Little is known about factors influencing SCT decision making. Objective: The study objective was to explore factors influencing patients' with FA and family members' decision making about SCT. Design: Using a mixed-methods exploratory design, we surveyed US and Canadian patients with FA and family members who were offered SCT. Main variables studied: Closed-ended survey items measured respondents' beliefs about the necessity, risks and concerns regarding SCT; multivariable logistic regression was used to examine the association between these factors and the decision to undergo SCT. Open-ended survey items measured respondents' perceptions of factors important to the SCT decision; qualitative analysis was used to identify emergent themes. Results: The decision to undergo SCT was significantly associated with greater perceived necessity (OR = 2.81, P = 0.004) and lower concern about harms of SCT (OR = 0.31, P = 0.03). Qualitative analysis revealed a perceived lack of choice among respondents regarding the use of SCT, which was related to physician influence and respondent concerns about patients' quality of life. Conclusions: Overall, study results emphasize the importance of the delicate interplay between provider recommendation of a medical procedure and patient/parental perceptions and decision making. Findings can help providers understand the need to acknowledge family members' perceptions of SCT decision making and offer a comprehensive discussion of the necessity, risks, benefits and potential outcomes.

Original languageEnglish (US)
Pages (from-to)929-941
Number of pages13
JournalHealth Expectations
Volume18
Issue number5
DOIs
StatePublished - Oct 1 2015
Externally publishedYes

Keywords

  • Decision making
  • Fanconi anaemia
  • Genetics
  • Psychosocial factors
  • Stem cell transplantation

ASJC Scopus subject areas

  • Public Health, Environmental and Occupational Health

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    Hutson, S. P., Han, P. K. J., Hamilton, J. G., Rife, S. C., Al-Rahawan, M. M., Moser, R. P., Duty, S. P., Anand, S., & Alter, B. P. (2015). The use of haematopoietic stem cell transplantation in Fanconi anaemia patients: A survey of decision making among families in the US and Canada. Health Expectations, 18(5), 929-941. https://doi.org/10.1111/hex.12066