Abstract：
Spinal muscular atrophy (SMA) has been reported in both Amish and Mennonite (Plain) communities, and a higher incidence has been observed in certain Mennonite communities compared to the general population. There are several therapies for SMA, but all are most effective in pre-symptomatic newborns. To identify couples from the Wisconsin Plain community who are most likely to have a child with SMA, carrier screening is offered via mailed kits with at-home specimen collection. Our survey data about Plain families\' perspectives on genetic testing suggest educational materials are needed for individuals providing informed consent with at-home specimen collection. We therefore developed a Plain population-specific educational trifold brochure about SMA carrier screening by incorporating existing medical education strategies and feedback from Plain community members and their health care providers. Along with the brochure, surveys were included in the kits to assess baseline knowledge about SMA carrier screening (\"pre-education\") as well as improvement in knowledge after reviewing the brochure and cultural appropriateness of the brochure (\"post-education\"). Fifty-five testing kits were distributed, and 26 survey pairs (pre- and post-education) were returned and analyzed (response rate 47%). Respondents had high baseline knowledge with an average of 5 of 7 questions (71%) answered correctly on the pre-education survey. Knowledge improved after reviewing the brochure as the average score increased to 6.5 of 7 questions (93%) answered correctly. Questions about risks of having an affected child after positive or negative carrier screening showed the most improvement from the pre-education to post-education surveys. Most respondents indicated the brochure was helpful, was easy to understand, and contained the right amount of information. Overall, incorporating elements of existing medical education strategies with feedback from the target population and stakeholders about appropriate language seems to be an effective method for creating beneficial, culturally responsive educational materials for the Plain population.
摘要：
在阿米什人和门诺人（平原）社区中都有脊髓性肌萎缩（SMA）的报道，与普通人群相比，某些门诺族社区的发病率更高。SMA有几种疗法，但所有这些都对症状前的新生儿最有效。为了确定威斯康星州平原社区最有可能生SMA孩子的夫妇，承运人筛选是通过邮寄工具包提供的，并在家中收集标本。我们关于平原家庭的调查数据对基因检测的观点表明，提供知情同意并在家中收集标本的个人需要教育材料。因此，我们通过结合现有的医学教育策略以及平原社区成员及其医疗保健提供者的反馈，开发了有关SMA携带者筛查的针对平原人群的三重教育手册。连同小册子,调查包括在工具包中，以评估有关SMA携带者筛查的基线知识（\“教育前\”）以及在审查手册和手册的文化适用性（\“教育后\”）后知识的改善。分发了55个检测包,和26个调查对(教育前和教育后)进行了返回和分析(回应率47%)。受访者具有较高的基线知识，在教育前调查中，平均有7个问题中的5个（71％）回答正确。阅读小册子后，知识得到了改善，因为平均分数增加到7个问题中的6.5个（93％）回答正确。关于在积极或消极的携带者筛查后有受影响儿童的风险的问题显示，从教育前到教育后的调查有最大的改善。大多数受访者表示这本小册子很有帮助，很容易理解,并包含适当数量的信息。总的来说,将现有医学教育策略的要素与目标人群和利益相关者对适当语言的反馈相结合，似乎是创造有益的有效方法，平原人口的文化响应性教育材料。
