Reproductive decision-making for Barth syndrome carriers: Unexplored complexities

巴特综合征携带者的生育决策:尚未探索的复杂性

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Abstract

Reproductive decision-making is a complex process, especially for carriers of a pathogenic X-linked genetic variant. Studies of X-linked carriers have identified various personal values and concerns that influence reproductive decisions, but these studies provide limited insight into decisional conflict during the reproductive decision-making process. This study aims to explore how carriers of variants known to cause Barth syndrome, a rare X-linked disorder, navigate and experience decisional conflict during reproductive decision-making. Semi-structured interviews were conducted with 27 Barth syndrome carriers recruited through the Barth Syndrome Foundation. Data was analyzed using reflexive thematic analysis with special attention paid to narratives of high and low decisional conflict and the values around which decisional conflict occurred. Findings showed that decisional conflict was a result of internal conflicts between competing value systems. To address decisional conflict, carriers re-prioritized their values, reconsidered the extent to which they allowed certain values to influence reproductive decisions, and altered the content of their values. Value systems could also be reshaped over time by personal experiences, which then could modify levels of decisional conflict in reproductive decisions. Decisional conflict often induces feelings of stress and guilt and could be exacerbated by the opinions of others. Genetic counselors are well equipped to provide decisional and emotional support to X-linked carriers during reproductive decision-making, and their services may help reduce decisional conflict and the negative emotions that accompany it. Counselors whose practice involves creating long-term relationships with patients should help carriers to identify factors that are contributing to internal value conflict and to decide whether and how they want support in this. This support may be provided through long-term genetic counseling structures or partnerships between genetic counselors, support organizations, and mental health professionals.

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