Health and social outcomes of children with visual impairment and blindness in the U.K

儿童期严重视力障碍和失明（SVIBL）是一种罕见的疾病，但对儿童的发育、教育和日常生活的许多方面都有重大影响。与成人发病的双侧视力丧失相比，儿童的“视力年损失”负担不成比例，因为它发生在生命过程的早期。英国儿童视力障碍和失明研究（BCVIS 1）是2000年进行的SVIBL国家监测研究，是目前唯一一项公布英国儿童SVIBL国家发病率数据的研究。BCVIS 2-调查全谱视觉障碍（即包括BCVIS 1中研究的视觉障碍类别和SVI/BL）最近已完成，论文正在进行中。在进入DTP之前，我是BCVIS 2研究的研究助理。目的探讨健康，教育和社会的结果，在成年早期的儿童诊断为全谱视力障碍和失明的儿童在童年和量化的第一次在英国的儿童SVIBL的经济负担。这个项目是重要的，因为它提供了基线生存和成本数据，否则将无法为儿童VI和SVIBL，是有用的，以填充未来的模型，成本效益分析的新的治疗方法或技术，需要公共fund.MethodsEthics批准将寻求从NHS机密咨询小组和健康研究管理局研究委员会：a）对BCVIS 1研究中确定的SVI/BL儿童进行未经同意的确定性联系（使用部分标识符：出生日期、邮政编码和没有记录的NHS号码的儿童的种族）到国家行政数据集，包括国家学生数据库（NPD），医院事件统计（HES）和ONS死亡率数据集。除了对健康结果进行纵向分析外，来自HES的医疗保健利用数据将用于计算医疗保健成本，并从医疗保健角度确定儿童SVIBL的疾病成本。B）与BCVIS 2队列成员的家庭直接联系（现为学龄前儿童）邀请父母参与评估健康和社会成果，使用标准化问卷，并在深入访谈，并通过其眼科医生获得进一步的临床随访数据（添加到已通过管理眼科医生收集的一年随访数据中）。这是一个学科桥接博士项目，这是反映在我的监督小组。我将招募儿童健康和行政数据领域的专家参加我的小组，但尚未列出姓名，因为会议将在他们休完夏季年假后举行。

IntroductionSevere visual impairment and blindness (SVIBL) in childhood is rare, but confers significant impact on many aspects of development, education and activities of everyday life. The burden in terms of 'sight years lost' is disproportionate in children compared to adult-onset bilateral vision loss as it occurs much earlier in the life-course.National, representative epidemiological data on childhood SVIBL are scarce. The British Childhood Visual Impairment & Blindness Study (BCVIS1), a national surveillance study of SVIBL conducted in 2000, is currently the only study that has published national incidence data on paediatric SVIBL in the U.K. The BCVIS2 - investigating full spectrum visual impairment (i.e. including the category of visual impairment (VI) as well as SVI/BL as studied in BCVIS1) has recently been completed and papers are in progress. I was the Research Assistant on the BCVIS2 study prior to entering the DTP. AimTo explore the health, educational and social outcomes in early adulthood of children diagnosed with full spectrum visual impairment and blindness during childhood and to quantify for the first time the economic burden of childhood SVIBL in the UK. This project is important as it provides baseline survival and cost data that would otherwise be unavailable for children with VI and SVIBL, and is useful to populate the future models for cost-effectiveness analyses of novel treatments or technologies that are required for public funding.MethodsEthics approval will be sought from the NHS Confidential Advisory Group and Health Research Authority research committee to:a) carry out unconsented deterministic linkage of children with SVI/BL identified in the BCVIS1 study (using the partial identifiers: date of birth, postcode and ethnicity for children without a recorded NHS number) to national administrative datasets including the national pupil database (NPD), hospital episodes statistics (HES) and ONS mortality dataset. In addition to the longitudinal analysis of health outcomes, healthcare utilisation data from HES will be used to calculate healthcare costs and determine the cost-of-illness of childhood SVIBL from the healthcare perspective.b) make direct contact with families of members of BCVIS2 cohort (now preschool age) to invite parents to participate in assessing health and social outcomes using standardised questionnaires and in-depth interviews and to obtain further clinical follow up data via their ophthalmologists (to add to one year follow up data already collected via managing ophthalmologists). This is a discipline bridging PhD project which is reflected in my supervisory panel. I will be enlisting experts in the field of child health and administrative data to my panel, but have not listed names as yet as meetings are taking place after they return from Summer annual leave.