Understanding Adult Participant and Parent Empowerment Prior to Evaluation in the Undiagnosed Diseases Network.
Understanding Adult Participant and Parent Empowerment Prior to Evaluation in the Undiagnosed Diseases Network.
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DOI：
10.1007/s10897-018-0228-6
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发表时间：
2018-09
影响因子：
1.9
通讯作者：
Undiagnosed Diseases Network
中科院分区：
文献类型：
--
作者：
Palmer CGS;McConkie-Rosell A;Holm IA;LeBlanc K;Sinsheimer JS;Briere LC;Dorrani N;Herzog MR;Lincoln S;Schoch K;Spillmann RC;Brokamp E;Undiagnosed Diseases Network
患有未确诊疾病的负担很高，包括身体和情感上的痛苦，挫折和不确定性。对于经历这些压力源的患者和家庭来说，更高水平的授权可能与更好的结果相关。因此，重要的是要了解未确诊的疾病患者和他们的家庭的经验，受未确诊的疾病，以确定战略，促进赋权。在这项研究中，我们使用遗传咨询结果量表（GCOS-24）来评估未诊断疾病网络（UDN）中35名成人参与者和67名儿童参与者的授权和支持团体参与水平，然后进行UDN亲自评估。我们的研究结果显示，与儿童参与者的父母相比，成年参与者在GCOS-24上的授权得分显著较低[t（100）= − 3.01，p = 0.003，平均差异= − 11.12，95% CI（− 3.78，− 18.46）]，并且支持小组参与与授权得分之间没有显著关联。大多数参与者（84.3%，86/102）目前没有参加任何支持小组，成年参与者和儿童参与者的父母的参与率没有显著差异（分别为11.4%和19.7%，FE p = 0.40）。不限名额的答复提供了更多的见解支持小组的参与，与未确诊的条件下生活的挑战，和积极的应对策略。未来的研究将评估在多大程度上授权分数变化的参与UDN展开。
The burden of living with an undiagnosed condition is high and includes physical and emotional suffering, frustrations, and uncertainty. For patients and families experiencing these stressors, higher levels of empowerment may be associated with better outcomes. Thus, it is important to understand the experiences of patients with undiagnosed conditions and their families affected by undiagnosed conditions in order to identify strategies for fostering empowerment. In this study, we used the Genetic Counseling Outcome Scale (GCOS-24) to assess levels of empowerment and support group participation in 35 adult participants and 67 parents of child participants in the Undiagnosed Diseases Network (UDN) prior to their UDN in-person evaluation. Our results revealed significantly lower empowerment scores on the GCOS-24 in adult participants compared to parents of child participants [t(100) = − 3.01, p = 0.003, average difference = − 11.12, 95% CI (− 3.78, − 18.46)] and no significant association between support group participation and empowerment scores. The majority of participants (84.3%, 86/102) are not currently participating in any support groups, and participation rates were not significantly different for adult participants and parents of child participants (11.4 vs. 19.7%, respectively, FE p = 0.40). Open-ended responses provided additional insight into support group participation, the challenges of living with undiagnosed conditions, and positive coping strategies. Future research will evaluate the extent to which empowerment scores change as participation in the UDN unfolds.
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影响因子：
3.2
作者：
Hsieh, HF;Shannon, SE
通讯作者：
Shannon, SE
影响因子：
3.7
作者：
Spillmann RC;McConkie-Rosell A;Pena L;Jiang YH;Undiagnosed Diseases Network;Schoch K;Walley N;Sanders C;Sullivan J;Hooper SR;Shashi V
通讯作者：
Shashi V
影响因子：
1.9
作者：
Diness, Birgitte Rode;Overbeck, Gritt;McAllister, Marion
通讯作者：
McAllister, Marion
影响因子：
1.4
作者：
Lewis, Celine;Skirton, Heather;Jones, Ray
通讯作者：
Jones, Ray
影响因子：
3.5
作者：
Bartlett, Yvonne Kiera;Coulson, Neil S.
通讯作者：
Coulson, Neil S.