Brown Kimberly L, Flume Patrick A
Division of Pulmonary, Critical Care, Allergy and Sleep Medicine, Medical University of South Carolina, 96 Jonathan Lucas Street, 816-CSB, Charleston, SC, 29425, USA.
J Genet Couns. 2018 Sep;27(5):1049-1054. doi: 10.1007/s10897-018-0220-1. Epub 2018 Feb 10.
Four to 5 % of cystic fibrosis (CF) patients are diagnosed as adults and often have subtler symptoms. Their siblings are at genetic risk to also have a subtler disease state. Diagnostic testing is recommended for siblings of newly diagnosed infants, but recommendations are less clear for later diagnoses. This study explored sibling testing recommendations in pediatric and adult practice using a survey that was emailed to CF clinicians. There were 58 respondents. Results revealed that 82.5% of pediatric and 36.4% of adult care respondents reported always recommending diagnostic testing for siblings of a newly diagnosed patient. In adult care, another 33.3% reported recommending diagnostic testing if the sibling has symptoms. In pediatric care, whether the sibling had newborn screening was most influential. Most pediatric respondents prefer the sweat chloride test, while 40% in adult practice prefer familial mutation analysis. Perceived barriers included cost, insurance coverage and logistical concerns in both settings, parental emotional state in pediatrics, and concern making recommendations for someone who is not the patient in adult care. Genetic counselors may be able to meet familial needs in CF care, including sibling testing. Many newly diagnosed patients/families do not see a genetic counselor, especially in adult care. These data reveal opportunities for practice guidelines and standardization.
4%至5%的囊性纤维化(CF)患者在成年期被诊断出来,且症状往往较为隐匿。他们的兄弟姐妹也有遗传风险,可能处于症状更隐匿的疾病状态。对于新诊断婴儿的兄弟姐妹,建议进行诊断检测,但对于后期诊断的建议则不太明确。本研究通过向CF临床医生发送电子邮件进行调查,探讨了儿科和成人医疗实践中对兄弟姐妹检测的建议。共有58名受访者。结果显示,82.5%的儿科受访者和36.4%的成人医疗受访者表示总是建议对新诊断患者的兄弟姐妹进行诊断检测。在成人医疗中,另有33.3%的受访者表示如果兄弟姐妹有症状则建议进行诊断检测。在儿科医疗中,兄弟姐妹是否进行过新生儿筛查影响最大。大多数儿科受访者更喜欢汗液氯化物检测,而在成人医疗实践中,40%的人更喜欢家族性突变分析。感知到的障碍包括两种情况下的成本、保险覆盖范围和后勤问题,儿科中的父母情绪状态,以及在成人医疗中为非患者的人提供建议的担忧。遗传咨询师或许能够满足CF护理中的家族需求,包括对兄弟姐妹的检测。许多新诊断的患者/家庭没有咨询过遗传咨询师,尤其是在成人医疗中。这些数据揭示了制定实践指南和实现标准化的机会。