在儿科癌症遗传学诊所采用基于家庭的级联基因检测方法。

IF 1.8 4区 医学 Q3 GENETICS & HEREDITY Familial Cancer Pub Date : 2024-11-20 DOI:10.1007/s10689-024-00434-8
Rida Haider, Lauren Desrosiers-Battu, Sarah Scollon, Pawel Stankiewicz, Philip J Lupo, Sharon E Plon
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引用次数: 0

摘要

遗传性癌症易感性疾病占所有儿科癌症的 10%。为疑似患者家庭提供的遗传咨询包括风险评估以及对父母和兄弟姐妹进行级联基因检测的建议,但目前还没有标准化的级联检测方法,导致诊所在进行级联基因检测时存在差异。我们在一家为不同种族患者提供服务的儿科癌症遗传学诊所,探讨了以家庭为基础的非综合征癌症易感性疾病级联检测方法的接受情况和相关结果。研究人员通过回顾性病历,评估了 106 名儿科疑似患者的父母和兄弟姐妹接受检测的情况。研究对象包括 99 位母亲、97 位父亲、116 位全亲兄弟姐妹和 53 位同父异母兄弟姐妹,他们都因遗传风险而被建议进行检测。在这些亲属中,有 156 人(43%)在疑似患者结果公布后 24 个月内完成了级联检测。家庭成员的类型和亲缘关系的不同,级联检测的完成情况也不同。41%的母亲(41/99)接受了检测,而26%的父亲(26/97)未接受检测;70.6%的兄弟姐妹(82/116)接受了检测,而13.2%的同父异母兄弟姐妹(7/53)未接受检测。使用卡方检验进行的统计分析显示,兄弟姐妹比父母更有可能完成检测(p
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A family-based approach to cascade genetic testing in a pediatric cancer genetics clinic.

Hereditary cancer predisposition disorders account for up to 10% of all pediatric cancers. Genetic counseling for families of the proband includes risk assessment and recommendations for cascade genetic testing for parents and siblings, but there is no standardized method for cascade testing in place resulting in variability in how clinics approach cascade genetic testing. We explored the uptake and outcomes associated with a family-based approach to cascade testing, for non-syndromic cancer predisposition disorders, at a pediatric cancer genetics clinic serving an ethnically diverse patient population. A retrospective chart review was conducted to evaluate test uptake in the parents and siblings of 106 pediatric probands. The study included 99 mothers, 97 fathers, 116 full siblings, and 53 half siblings who were recommended testing due to genetic risk. Of these relatives, 156 (43%) had documentation of completed cascade testing within twenty-four months after the proband's result disclosure. Completion of cascade testing varied by the type of family member and degree of relatedness. 41% of mothers (41/99) were tested in comparison to 26% of fathers (26/97) and 70.6% of full siblings (82/116) were tested compared to 13.2% of half siblings (7/53). Statistical analysis using chi-squared tests revealed that siblings were more likely to have completed testing than parents (p < 0.001). Furthermore, amongst parents, mothers were more likely to complete testing than fathers (p = 0.03) and amongst siblings, full siblings were more likely to complete testing than half siblings (< 0.001). The proband's age (p = 0.008), parents' preferred language (p = 0.002), and interpreter use during visit (p = 0.004) were the factors associated with differences in test uptake amongst siblings, whereas the proband's race/ethnicity (p = 0.019) was the only factor associated with differences in test uptake amongst parents. The most common barriers noted in charts for lack of test completion included country of residence, lack of insurance, and loss to follow-up. In conclusion, we found that test uptake differed significantly among relatives of a proband with siblings being more likely to test than parents. We also found differences in the demographic and clinical factors associated with test uptake in parents and siblings. Future studies need to validate these differences and further explore the underlying cause of variation in test uptake among relatives.

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来源期刊
Familial Cancer
Familial Cancer 医学-遗传学
CiteScore
4.10
自引率
4.50%
发文量
36
审稿时长
6-12 weeks
期刊介绍: In recent years clinical cancer genetics has become increasingly important. Several events, in particular the developments in DNA-based technology, have contributed to this evolution. Clinical cancer genetics has now matured to a medical discipline which is truly multidisciplinary in which clinical and molecular geneticists work together with clinical and medical oncologists as well as with psycho-social workers. Due to the multidisciplinary nature of clinical cancer genetics most papers are currently being published in a wide variety of journals on epidemiology, oncology and genetics. Familial Cancer provides a forum bringing these topics together focusing on the interests and needs of the clinician. The journal mainly concentrates on clinical cancer genetics. Most major areas in the field shall be included, such as epidemiology of familial cancer, molecular analysis and diagnosis, clinical expression, treatment and prevention, counselling and the health economics of familial cancer.
期刊最新文献
A family-based approach to cascade genetic testing in a pediatric cancer genetics clinic. Cascade genetic testing in hereditary cancer: exploring the boundaries of the Italian legal framework. Hereditary breast and ovarian cancer genetic testing in unselected patients: example of private supplementation of public healthcare service. BRCAIndica: a resource for ACMG/AMP classified BRCA1 and BRCA2 variants. Germline pathogenic variants in RNF43 in patients with and without serrated polyposis syndrome.
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