Analysis of Risks and Outcomes for Rare Cancers

罕见癌症的风险和结果分析

• 批准号: 8453390
• 负责人: DIANNE M FINKELSTEIN
• 金额: $ 8.18万
• 依托单位: MASSACHUSETTS GENERAL HOSPITAL
• 依托单位国家: 美国
• 财政年份: 2012
• 资助国家: 美国
• 起止时间: 2012-04-04 至 2016-03-31
• 项目状态: 已结题
• 来源: https://reporter.nih.gov/project-details/8453390
• 关键词: Accounting; Affect; Age; Cancer Genetics Network; Cancer Patient; Cancer Survivor; Characteristics; Choices and Control; Clinical; Comorbidity; Control Groups; Data; Data Analyses; Data Set; Diagnosis; Disease; Disease Outcome; Environmental Exposure; Etiology; Family; Family Cancer History; Family history of; Family member; Fatigue; Funding; Genetic Predisposition to Disease; Genetic screening method; Goals; Grant; Heart Diseases; Incidence; Institution; Life Style; Malignant Neoplasms; Memory Loss; Mental Depression; Methodology; Orphan; Outcome; Participant; Patients; Rare Diseases; Recording of previous events; Recruitment Activity; Registries; Research; Research Infrastructure; Risk; Second Primary Cancers; Site; Smoking; Symptoms; Test Result; United States National Institutes of Health; anticancer research; cancer diagnosis; cancer genetics; cancer site; cancer type; demographics; follow-up; genetic registry; insight; interest; mortality; programs; psychologic; tumor

DESCRIPTION (provided by applicant): Taken as an aggregate, rare cancers (those affecting fewer than 200,000 people in the US according to the Orphan Disease Act definition) account for 27% of the US cancer diagnoses and 25% of cancer mortality. However, while new therapies have changed the way some common cancers are treated, there has been little advance in the treatment of most rare cancers and research directed at these diseases is sparse. The main goal of this application is to launch a research program aimed at deepening the understanding of the etiology and long-term outcomes of patients diagnosed with rare cancers. For our analyses, we will utilize data collected by the Cancer Genetics Network (CGN) and the Rare Cancer Genetics Registry (RCGR). The CGN, developed in 1998, has over 15,000 participants with cancer (of whom 873 had a rare cancer) in addition to over 5,000 of their unaffected family members. The RCGR, funded by an NIH Challenge grant in 2009, has over 400 participants with rare cancers recruited by a subset of the CGN sites. Dr. Finkelstein is the PI of the both the CGN and the RCGR. Neither the CGN nor the RCGR provided funding to support analysis of the data collected for the registries. This application plans to undertake analyses of the characteristics associated with elevated risk of rare cancers, as well as long-term outcomes of these diseases. Specific Aims include: 1. Create a single research data set from data collected in the CGN and RCGR consisting of subjects with rare cancers, those with more common cancers, and unaffected family members of participants. Available data include demographics, lifestyle and environmental exposures, medical and family history, genetic test results, cancer diagnoses, co-morbidities, treatment and survival status. On CGN participants, we have over 10 years follow-up of clinical, psychological and physical outcomes of disease and treatment. 2. Using the combined CGN and RCGR data, determine the clinical, demographic, environmental, life-style, and family history characteristics associated with an elevated risk of each rare cancer type. Questions of interest include whether rare cancer patients have a higher rate of exposures such as smoking or a different profile of family cancer history than people who do not get these cancers. 3. Using long-term (10 year) follow-up data from CGN registrants, determine the long-term outcomes in rare cancer survivors. Questions of interest include what are the risks of co-morbidities (such as heart disease) and second primary cancers, and how do these risks compare between patients with the rare cancer versus unaffected controls? What are the risks of psychological symptoms (memory loss, fatigue, depression) and are these more common than unaffected (controls)? Than in patients with common cancers? What are the demographic, clinical, lifestyle and treatment predictors of physical and psychological long-term complications in patients with rare cancers? Analyses will be done separately within each rare cancer site when possible. and an aggregated analysis using all data, accounting for age, diagnosis, registry and site will be done.

描述（由申请人提供）：总体而言，罕见癌症（根据孤儿疾病法案定义，在美国影响不到20万人的癌症）占美国癌症诊断的27%和癌症死亡率的25%。然而，尽管新疗法改变了一些常见癌症的治疗方式，但大多数罕见癌症的治疗进展甚微，针对这些疾病的研究也很少。该申请的主要目标是启动一项研究计划，旨在加深对诊断为罕见癌症的患者的病因和长期结局的理解。在我们的分析中，我们将利用癌症遗传学网络（CGN）和罕见癌症遗传学登记处（RCGR）收集的数据。CGN于1998年开发，有超过15，000名癌症患者（其中873人患有罕见癌症）以及超过5，000名未受影响的家庭成员。2009年，由NIH挑战基金资助的RCGR有400多名患有罕见癌症的参与者，他们被CGN的一个子集招募。芬克尔斯坦博士是这两个人的私家侦探 CGN和RCGR。CGN和RCGR均未提供资金支持对登记研究收集的数据进行分析。该应用计划对与罕见癌症风险升高相关的特征以及这些疾病的长期结果进行分析。具体目标包括：1。从CGN和RCGR中收集的数据创建一个单一的研究数据集，包括罕见癌症受试者、常见癌症受试者和受试者未受影响的家庭成员。现有数据包括人口统计学、生活方式和环境暴露、病史和家族史、基因检测结果、癌症诊断、合并症、治疗和生存状况。对于CGN参与者，我们对疾病和治疗的临床，心理和身体结局进行了10年以上的随访。2.使用CGN和RCGR数据，确定与每种罕见癌症风险升高相关的临床、人口统计学、环境、生活方式和家族史特征 类型.感兴趣的问题包括罕见癌症患者是否有更高的暴露率，如吸烟或不同的家族癌症史，而不是那些没有患上这些癌症的人。3.使用CGN注册者的长期（10年）随访数据，确定罕见癌症幸存者的长期结局。感兴趣的问题包括合并症（如心脏病）和第二原发性癌症的风险是什么，以及这些风险在罕见癌症患者与未受影响的对照组之间如何比较？心理症状（记忆丧失、疲劳、抑郁）的风险是什么？这些症状比未受影响的（对照组）更常见吗？比普通癌症患者更好？罕见癌症患者生理和心理长期并发症的人口统计学、临床、生活方式和治疗预测因素是什么？如果可能，将在每个罕见癌症部位单独进行分析。将使用所有数据进行汇总分析，包括年龄、诊断、登记和研究中心。

项目成果

Physical symptoms in long-term survivors of rare cancer.

罕见癌症长期幸存者的身体症状。

• DOI: 10.1007/s11764-018-0721-9
• 发表时间: 2018
• 期刊: Journal of cancer survivorship : research and practice
• 作者: Horick,NoraK;Muzikansky,Ariela;Gutierrez,HildaL;Boyd,KristinaL;Finkelstein,DianneM
• 通讯作者: Finkelstein,DianneM