South Carolina Study for Adolescents and Young Adults with Rare Conditions

南卡罗来纳州针对患有罕见疾病的青少年和年轻人的研究

• 批准号: 8507475
• 负责人: SUZANNE W MCDERMOTT
• 金额: $ 40.22万
• 依托单位: UNIVERSITY OF SOUTH CAROLINA AT COLUMBIA
• 依托单位国家: 美国
• 财政年份: 2011
• 资助国家: 美国
• 起止时间: 2011-09-30 至 2014-12-29
• 项目状态: 已结题
• 来源: https://reporter.nih.gov/project-details/8507475
• 关键词: South; Carolina; Study; Adolescents; Young

PROJECT ABSTRACT The proposed South Carolina project will be a collaboration between and the University of South Carolina and the South Carolina State government (Budget and Control Board- Office for Research and Statistics). This project is possible because South Carolina has the potential to answer important questions about the health and welfare of adolescents and young adults with rare diseases using de-identified (anonymous) data from numerous agencies. For the 15-24 year age group, using only two data sources, the Hospital Discharge Data Set (HDDS) and Medicaid for the period 2000-2009, we have already identified 83 adolescents and young adults with fragile X syndrome, 635 with spinal bifida and 224 with muscular dystrophy. The project will have two parts: cross-sectional (a single point in time) and longitudinal (looking at people over an extended time period) and the study group will be 15-24 year olds with spina bifida, muscular dystrophy and fragile X syndrome. We will use linked administrative datasets (such as Medicaid, State Department of Education, and Vocational Rehabilitation) to describe the well-being and service use of the study group. We will estimate the prevalence of these conditions, their health status, educational, and employment participation. We will estimate the age specific prevalence for each condition and describe the occurrence of other acute and chronic medical conditions, including diabetes mellitus, obesity, heart disease, respiratory disease and other conditions. We will also describe the incidence and prevalence of co-morbid and secondary conditions and the co-occurrence of intellectual disability, paralysis, epilepsy, attention deficit hyperactivity disorder, and other conditions. We will also describe the frequency and type of health care received by each group, including rates of hospitalization, emergency department visits, and outpatient services, and the cost-effectiveness of different approaches to care. In the second and third year of the grant we will interview 125 affected individuals and their families to describe the living situation, education, employment, and other markers of social participation by adolescents and young adults with the three conditions, using the questionnaires from the National Longitudinal Transition Study and the RAND 36. We will describe the transition that occurs as adolescents with each condition mature into young adulthood, in terms of health care and social services (including type and specialty of health care provider by specialty, physical and occupational therapy, speech/language therapy, counseling and other services). Finally, we plan to assess the ability of other states to conduct the linked administrative data analyses and make recommendations that will be generalizable.

项目摘要 拟议的南卡罗来纳州项目将是与南卡罗来纳州大学和 南卡罗来纳州政府（预算和控制委员会-研究和统计办公室）。这 该项目是可能的，因为南卡罗来纳州有潜力回答有关健康的重要问题， 使用来自以下数据的去识别（匿名）数据， 许多机构。对于15-24岁年龄组，仅使用两个数据来源，出院数据 2000-2009年期间，我们已经确定了83名青少年和年轻人， 脆性X综合征的成年人，635例脊柱裂和224例肌肉萎缩症。 该项目将有两个部分：横截面（一个时间点）和纵向（看人超过 研究组为15-24奥尔兹的脊柱裂、肌营养不良和 脆性X综合征我们将使用链接的管理数据集（如Medicaid、State Department of 教育和职业康复）来描述研究组的幸福感和服务使用情况。我们 将估计这些疾病的患病率，他们的健康状况，教育和就业参与。 我们将估计每种疾病的年龄特异性患病率，并描述其他急性和 慢性疾病，包括糖尿病、肥胖、心脏病、呼吸系统疾病和其他 条件我们还将描述并发症和继发性疾病的发病率和患病率， 智力残疾、瘫痪、癫痫、注意缺陷多动障碍和其他的共同发生 条件我们还将描述每个群体接受卫生保健的频率和类型，包括费率 住院、急诊和门诊服务的成本效益，以及不同 接近照顾。在赠款的第二年和第三年，我们将采访125名受影响的个人， 他们的家庭描述生活状况，教育，就业和其他社会参与标志 青少年和年轻人的三个条件，使用问卷调查，从国家 纵向过渡研究和兰德公司36.我们将描述发生在青少年时期的转变 随着每种疾病在成年早期成熟，在医疗保健和社会服务方面（包括类型）， 按专业、物理和职业治疗、言语/语言治疗， 咨询和其他服务）。最后，我们计划评估其他国家开展相关活动的能力。 行政数据分析，并提出建议，将是普遍的。

项目成果

Skin Ulcers and Mortality Among Adolescents and Young Adults With Spina Bifida in South Carolina During 2000-2010.

• DOI: 10.1177/0883073815596611
• 发表时间: 2016-03
• 期刊: Journal of child neurology
• 影响因子: 1.9
• 作者: Cai B;McDermott S;Wang Y;Royer JA;Mann JR;Hardin JW;Ozturk O;Ouyang L
• 通讯作者: Ouyang L

Use of state administrative data sources to study adolescents and young adults with rare conditions.

利用国家行政数据源研究患有罕见疾病的青少年和年轻人。

• DOI: 10.1007/s11606-014-2925-7
• 发表时间: 2014
• 期刊: Journal of general internal medicine
• 影响因子: 5.7
• 作者: Royer,JA;Hardin,JW;McDermott,S;Ouyang,L;Mann,JR;Ozturk,OD;Bolen,J
• 通讯作者: Bolen,J

Development of a tool to describe overall health, social independence and activity limitation of adolescents and young adults with disability.

开发一个工具来描述残疾青少年和年轻人的整体健康、社会独立性和活动限制。

• DOI: 10.1016/j.ridd.2014.12.009
• 发表时间: 2015
• 期刊: Research in developmental disabilities
• 影响因子: 3.1
• 作者: Deroche,ChelseaB;Holland,MargaretM;McDermott,Suzanne;Royer,JulieA;Hardin,JamesW;Mann,JoshuaR;Salzberg,Deborah;Ozturk,Orgul;Ouyang,Lijing
• 通讯作者: Ouyang,Lijing

Emergency department and inpatient hospitalizations for young people with fragile X syndrome.

患有脆性 X 综合征的年轻人的急诊室和住院治疗。

• DOI: 10.1352/1944-7558-120.3.230
• 发表时间: 2015
• 期刊: American journal on intellectual and developmental disabilities
• 作者: McDermott,Suzanne;Hardin,JamesW;Royer,JulieA;Mann,JoshuaR;Tong,Xin;Ozturk,OrgulD;Ouyang,Lijing
• 通讯作者: Ouyang,Lijing

Hospitalizations and emergency room visits for adolescents and young adults with muscular dystrophy living in South Carolina.

居住在南卡罗来纳州的患有肌营养不良症的青少年和年轻人住院和急诊室就诊。

• DOI: 10.1002/mus.24599
• 发表时间: 2015
• 期刊: Muscle & nerve
• 影响因子: 3.4
• 作者: Mann,JoshuaR;Royer,JulieA;Mcdermott,Suzanne;Hardin,JamesW;Ozturk,Orgul;Street,Natalie
• 通讯作者: Street,Natalie