Factors affecting adherence to a high-risk surveillance protocol among patients with Li-Fraumeni syndrome.

IF 2 4区医学 Q3 ONCOLOGY

Hereditary Cancer in Clinical Practice Pub Date : 2023-08-11 DOI:10.1186/s13053-023-00259-z

Kaylee A Underkofler, Martha H Thomas, Christina J Taylor, Christa L Mazur, Sarah H Erickson, Kari L Ring

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Abstract

Background: High-risk surveillance for patients with Li-Fraumeni syndrome (LFS) has shown a stage shift and improved overall survival, but is demanding. Our objective was to evaluate surveillance adherence in a population of patients with LFS presenting for high-risk care.

Methods: A retrospective analysis of surveillance adherence of adult patients with LFS at a single institution was performed. Adherence was defined by the duration from initial University of Virginia (UVA) LFS clinic visit to the time of first missed surveillance test. Two-sample t-tests and ANOVA tests were used to identify factors associated with duration of adherence.

Results: A total of 42 patients were evaluated in the UVA LFS clinic between 2017 and 2021. Of these, 21 patients met inclusion criteria. At the time of review, 6 patients (29%) were up to date with high-risk surveillance recommendations. The mean duration of adherence was 17 months. Female sex was found to be associated with longer duration of adherence (mean 21 mo vs. 3.5 mo for males, p = 0.02). A personal history or active diagnosis of cancer was also associated with increased adherence (p = 0.02). However, neither age (p = 0.89), geography (p = 0.84), or known family history of LFS (p = 0.08) were associated with duration of adherence.

Conclusion: Female sex as well as a personal history of cancer were associated with longer duration of adherence to recommended high-risk surveillance among patients with LFS. Identification of barriers to surveillance will be essential moving forward to increase adherence and promote early detection of cancer, thereby reducing the morbidity and mortality of LFS.

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影响Li-Fraumeni综合征患者遵守高风险监测方案的因素

背景:对Li-Fraumeni综合征(LFS)患者的高风险监测已经显示出一个阶段的转变和总体生存率的提高，但仍有要求。我们的目的是评估接受高风险治疗的LFS患者的监测依从性。方法:回顾性分析成人LFS患者在单一机构的监测依从性。依从性定义为从弗吉尼亚大学(UVA) LFS门诊就诊到第一次错过监测测试的时间。使用双样本t检验和方差分析检验来确定与依从时间相关的因素。结果:2017年至2021年间，共有42名患者在UVA LFS诊所接受了评估。其中，21例患者符合纳入标准。在回顾时，6例患者(29%)符合最新的高危监测建议。平均服药时间为17个月。研究发现，女性患者坚持服药的时间更长(平均21个月，男性3.5个月，p = 0.02)。个人病史或积极的癌症诊断也与依从性增加有关(p = 0.02)。然而，年龄(p = 0.89)、地理(p = 0.84)或已知的LFS家族史(p = 0.08)与坚持时间无关。结论:女性以及个人癌症病史与LFS患者更长的高危监测依从时间相关。确定监测障碍对于提高依从性和促进癌症的早期发现至关重要，从而降低LFS的发病率和死亡率。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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来源期刊

Hereditary Cancer in Clinical Practice 医学-肿瘤学

CiteScore

3.10

自引率

5.90%

发文量

审稿时长

>12 weeks

期刊介绍： Hereditary Cancer in Clinical Practice is an open access journal that publishes articles of interest for the cancer genetics community and serves as a discussion forum for the development appropriate healthcare strategies. Cancer genetics encompasses a wide variety of disciplines and knowledge in the field is rapidly growing, especially as the amount of information linking genetic differences to inherited cancer predispositions continues expanding. With the increased knowledge of genetic variability and how this relates to cancer risk there is a growing demand not only to disseminate this information into clinical practice but also to enable competent debate concerning how such information is managed and what it implies for patient care. Topics covered by the journal include but are not limited to: Original research articles on any aspect of inherited predispositions to cancer. Reviews of inherited cancer predispositions. Application of molecular and cytogenetic analysis to clinical decision making. Clinical aspects of the management of hereditary cancers. Genetic counselling issues associated with cancer genetics. The role of registries in improving health care of patients with an inherited predisposition to cancer.