Vidavalur Ramesh
Department of Neonatology, Cayuga Medical Center, Ithaca, NY, United States.
Front Pediatr. 2022 May 3;10:788589. doi: 10.3389/fped.2022.788589. eCollection 2022.
Congenital hypothyroidism (CH) is one of the most common preventable causes of mental retardation. Implementing newborn screening (NBS) in >52 countries enabled early detection and to initiate treatment of neonates with CH. India is yet to implement a national NBS program even though an estimated 5-15% of sick newborns suffer from genetic and metabolic disorders. Recent pilot studies confirm that the CH incidence rates range from 1 in 500 to 1 in 3,400 live births. Our objective was to estimate overall incidence rates of congenital hypothyroidism and to evaluate the costs and benefits of implementing universal NBS for CH in India.
We used the best available epidemiological and cost data to synthesize incidence rates and screening costs for CH in India. We conducted a meta-analysis of country-specific published literature and included 14 studies to calculate baseline CH incidence rates. We used two models to estimate intellectual disability in unscreened cohorts. Disability-adjusted life years (DALY) were calculated to quantify burden of disease utilizing disability weights. Direct costs including screening, confirmatory tests, and treatment costs were obtained from public and private market sources. Economic benefits were calculated from lost DALY using human capital approach and value of statistical life methods, utilizing gross national income (GNI) per capita data and value of statistical life year (VSLY), respectively. Cost discounting was used to estimate the present value of future benefits over lifetime of affected newborns.
The incidence rate of CH in India is 72 (95% CI: 58, 85) cases per 100,000 live births. Based on this data, 1 in 1,388 (95% CI: 1166, 1714) infants were diagnosed with CH in India for the year 2018. The estimated annual incidence ranged from 14,000 to 20,730 cases, and those at risk for intellectual disability ranged from 5,397 to 13,929 cases. Estimated discounted and undiscounted lost DALYs were 57,640 and 410,000, respectively. Direct annual costs for universal screening for CH in India is around USD187 million. Based on current incidence and expected severity of sequelae, economic losses ranged from USD 159 million to 1.1 billion. Benefit-cost ratios ranged from 1.8 to 6.
Universal NBS for CH is one of the healthcare interventions that is beneficial to prevent morbidity and cost saving. The cumulative economic benefits, derived from prevention of intellectual disability, assuming cost effectiveness threshold of three times of gross domestic product per capita, far outweigh the direct and indirect costs of screening, treatment, and surveillance throughout the life of the affected individuals. Our analysis strongly supports the argument for investing in NBS that provides good value for money and would yield substantial financial gains for the country.
先天性甲状腺功能减退症(CH)是导致智力发育迟缓的最常见可预防病因之一。超过52个国家实施了新生儿筛查(NBS),从而能够早期发现并对患有CH的新生儿进行治疗。尽管估计有5%-15%的患病新生儿患有遗传和代谢紊乱,但印度尚未实施全国性的NBS计划。最近的试点研究证实,CH的发病率在每500至3400例活产中有1例。我们的目标是估计先天性甲状腺功能减退症的总体发病率,并评估在印度实施CH通用NBS的成本和效益。
我们使用了现有的最佳流行病学和成本数据来综合印度CH的发病率和筛查成本。我们对各国发表的文献进行了荟萃分析,并纳入了14项研究来计算CH的基线发病率。我们使用两种模型来估计未筛查队列中的智力残疾情况。利用残疾权重计算残疾调整生命年(DALY)以量化疾病负担。包括筛查、确诊试验和治疗成本在内的直接成本来自公共和私人市场来源。经济效益分别使用人力资本方法和统计生命价值方法,根据人均国民总收入(GNI)数据和统计生命年价值(VSLY),从损失的DALY中计算得出。成本贴现用于估计受影响新生儿一生未来效益的现值。
印度CH的发病率为每10万例活产中有72例(95%置信区间:58, 85)。基于这些数据,2018年印度每1388例(95%置信区间:1166, 1714)婴儿中有1例被诊断为CH。估计的年发病率在14000至20730例之间,有智力残疾风险的病例在5397至13929例之间。估计的贴现和未贴现损失的DALY分别为57640和410000。印度CH通用筛查的直接年度成本约为1.87亿美元。根据目前的发病率和后遗症的预期严重程度,经济损失在1.59亿美元至11亿美元之间。效益成本比在1.8至6之间。
CH通用NBS是有利于预防发病和节省成本的医疗保健干预措施之一。假设成本效益阈值为人均国内生产总值的三倍,因预防智力残疾而产生的累积经济效益远远超过受影响个体一生的筛查、治疗和监测的直接和间接成本。我们的分析有力地支持了投资NBS的观点,NBS具有良好的性价比,将为该国带来可观的经济收益。