Howard Sharp Katianne M, Li Chen, Lu Zhaohua, Clark Mary Egan, Jurbergs Niki, Ouma Annastasia, Harrison Lynn, Gerhardt Elsie, Taylor Leslie, Hamilton Kayla V, McGee Rose B, Nuccio Regina, Hines-Dowell Stacy, Gattuso Jami S, Pritchard Michelle, Mandrell Belinda N, Tercyak Kenneth P, Nichols Kim E, Johnson Liza-Marie
Department of Psychology and Biobehavioral Sciences, St Jude Children's Research Hospital, Memphis, TN.
Department of Biostatistics, St Jude Children's Research Hospital, Memphis, TN.
JCO Precis Oncol. 2023 Sep;7:e2300159. doi: 10.1200/PO.23.00159.
To characterize parents' quality of life (QoL) after germline genomic sequencing for their children with cancer.
Participants were n = 104 parents of children with cancer enrolled in a prospective study of clinical tumor and germline genomic sequencing. Parents completed surveys at study consent (T0), before disclosure of their child's germline results (T1), and again ≥5 weeks after results disclosure (T2). Bivariate associations with QoL were examined, followed by a multivariable regression model predicting parents' psychological distress.
At T2, parental distress significantly differed by their children's germline result type (positive, uncertain, negative; = .038), parent relationship status ( = .04), predisclosure genetics knowledge ( = .006), and predisclosure worry about sequencing ( < .001). Specifically, parents of children with positive (ie, pathogenic or likely pathogenic) results experienced greater distress than those of children with negative results ( = .029), as did parents who were single, more knowledgeable about genetics, and with greater worry. In the adjusted regression model, a positive germline result remained significantly associated with parents' lower QoL at T2 follow-up ( [4,92] = 9.95; < .001; = .30; β = .19; = .031).
Germline genomic sequencing for children with cancer is associated with distress among parents when revealing an underlying cancer predisposition among their affected children. Genetic education and counseling before and after germline sequencing may help attenuate this impact on QoL by addressing parents' concerns about test results and their health implications. Assessing parents' worry early in the testing process may also aid in identifying those most likely in need of psychosocial support.
描述患有癌症儿童的父母在进行种系基因组测序后的生活质量（QoL）。
参与者为104名患有癌症儿童的父母，他们参与了一项关于临床肿瘤和种系基因组测序的前瞻性研究。父母在研究同意时（T0）、在披露其孩子的种系结果之前（T1）以及结果披露后≥5周再次（T2）完成调查问卷。研究了与生活质量的双变量关联，随后建立了一个多变量回归模型来预测父母的心理困扰。
在T2时，父母的困扰因孩子的种系结果类型（阳性、不确定、阴性；P = 0.038）、父母关系状况（P = 0.04）、披露前的遗传学知识（P = 0.006）以及披露前对测序的担忧（P < 0.001）而有显著差异。具体而言，孩子结果为阳性（即致病或可能致病）的父母比孩子结果为阴性的父母经历了更大的困扰（P = 0.029），单身、对遗传学了解更多以及担忧程度更高的父母也是如此。在调整后的回归模型中，种系结果为阳性在T2随访时仍与父母较低的生活质量显著相关（F [4,92] = 9.95；P < 0.001；R² = 0.30；β = 0.19；P = 0.031）。
对患有癌症的儿童进行种系基因组测序，当揭示其患病子女存在潜在的癌症易感性时，会使父母感到困扰。种系测序前后的遗传教育和咨询可能有助于通过解决父母对检测结果及其健康影响的担忧来减轻对生活质量的这种影响。在检测过程早期评估父母的担忧也可能有助于识别那些最可能需要心理社会支持的人。
