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

儿童严重视力障碍和失明（SVIBL）是罕见的，但对发展、教育和日常生活活动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“视力丧失”负担不成比例，因为它发生在生命历程的早期。关于儿童SVIBL的国家代表性流行病学数据很少。英国儿童视觉损伤与失明研究（BCVIS1）是英国2000年开展的一项SVIBL国家监测研究，是目前英国唯一一项公布了全国儿童SVIBL发病率数据的研究。BCVIS2 -调查全谱视觉障碍（即包括视觉损伤（VI）类别以及BCVIS1中研究的SVI/BL）已于近期完成，论文正在撰写中。在进入DTP之前，我是BCVIS2研究的研究助理。目的探讨儿童期诊断为全谱视力障碍和失明的儿童成年早期的健康、教育和社会结果，并首次量化英国儿童期SVIBL的经济负担。该项目很重要，因为它提供了VI和SVIBL儿童的基线生存和成本数据，否则这些数据将无法获得，并且有助于建立未来的模型，用于公共资金所需的新治疗或技术的成本效益分析。方法将获得NHS保密咨询小组和卫生研究管理局研究委员会的伦理批准，以：a)将在BCVIS1研究中确定的SVI/BL儿童（使用部分标识符：出生日期、邮政编码和没有记录NHS号码的儿童的种族）与国家管理数据集进行未经同意的确定性关联，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。除了对健康结果的纵向分析外，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BCVIS2队列成员（现为学龄前）的家庭直接联系，邀请家长通过标准化问卷和深度访谈参与评估健康和社会结果，并通过其眼科医生获得进一步的临床随访数据（在管理眼科医生已收集的一年随访数据的基础上增加）。这是一个学科衔接博士项目，反映在我的督导小组中。我将招募儿童健康和行政数据领域的专家加入我的小组，但目前还没有列出名单，因为他们将在夏季年假结束后开会。

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.