Diagnostic exome sequencing in children: A survey of parental understanding, experience and psychological impact

被引:42
作者
Wynn, J. [1 ]
Ottman, R. [2 ,3 ,4 ,5 ]
Duong, J. [6 ]
Wilson, A. L. [7 ]
Ahimaz, P. [1 ]
Martinez, J. [1 ]
Rabin, R. [8 ]
Rosen, E. [8 ]
Webster, R. [9 ]
Au, C. [7 ]
Cho, M. T. [7 ,10 ]
Egan, C. [7 ]
Guzman, E. [7 ]
Primiano, M. [7 ]
Shaw, J. E. [7 ]
Sisson, R. [7 ]
Klitzman, R. L. [5 ,11 ]
Appelbaum, P. S. [5 ,11 ]
Lichter-Konecki, U. [1 ]
Anyane-Yeboa, K. [1 ]
Iglesias, A. [1 ]
Chung, W. K. [1 ,12 ]
机构
[1] Columbia Univ, Dept Pediat, Med Ctr, Div Clin Genet, New York, NY 10027 USA
[2] Columbia Univ, GH Sergievsky Ctr, Med Ctr, New York, NY USA
[3] Columbia Univ, Dept Epidemiol, Med Ctr, New York, NY USA
[4] Columbia Univ, Dept Neurol, Med Ctr, New York, NY USA
[5] NY State Psychiat Inst, New York, NY USA
[6] Columbia Univ, Mailman Sch Publ Hlth, Dept Sociomed Sci, New York, NY USA
[7] Columbia Univ, Dept Pediat, New York Presbyterian Hosp, Div Clin Genet, New York, NY 10027 USA
[8] Long Isl Univ Post Campus, Coll Liberal Arts & Sci, Brookville, NY USA
[9] Columbia Univ, Sch Med, New York, NY USA
[10] GeneDx, Gaithersburg, MD USA
[11] Columbia Univ, Dept Psychiat, Med Ctr, New York, NY USA
[12] Columbia Univ, Dept Med, Med Ctr, New York, NY USA
关键词
exome sequencing; genetic counseling; genomic medicine; parental experience; psychological impact; CLINICAL EXOME; INCIDENTAL FINDINGS; COLORECTAL-CANCER; INFORMED-CONSENT; RISK-ASSESSMENT; PERCEPTIONS; DISORDERS; ATTITUDES; BELIEFS; DISEASE;
D O I
10.1111/cge.13200
中图分类号
Q3 [遗传学];
学科分类号
071007 ; 090102 ;
摘要
Clinical exome sequencing (CES) is increasingly being used as an effective diagnostic tool in the field of pediatric genetics. We sought to evaluate the parental experience, understanding and psychological impact of CES by conducting a survey study of English-speaking parents of children who had diagnostic CES. Parents of 192 unique patients participated. The parent's interpretation of the child's result agreed with the clinician's interpretation in 79% of cases, with more frequent discordance when the clinician's interpretation was uncertain. The majority (79%) reported no regret with the decision to have CES. Most (65%) reported complete satisfaction with the genetic counseling experience, and satisfaction was positively associated with years of genetic counselor (GC) experience. The psychological impact of CES was greatest for parents of children with positive results and for parents with anxiety or depression. The results of this study are important for helping clinicians to prepare families for the possible results and variable psychological impact of CES. The frequency of parental misinterpretation of test results indicates the need for additional clarity in the communication of results. Finally, while the majority of patients were satisfied with their genetic counseling, satisfaction was lower for new GCs, suggesting a need for targeted GC training for genomic testing.
引用
收藏
页码:1039 / 1048
页数:10
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