Thomas C, Mirallié S, Pierres C, Dert C, Clément M-C, Mahlaoui N, Durand-Zaleski I, Fischer A, Audrain M
Service d'hématologie et d'immunologie pédiatrique, hôpital Mère-Enfants, 7, quai Moncousu, 44000 Nantes, France.
Laboratoire ANDEMEGEN, 38, boulevard J.-Monnet, 44093 Nantes cedex1, France.
Arch Pediatr. 2015 Jun;22(6):646-52. doi: 10.1016/j.arcped.2015.03.001. Epub 2015 Apr 18.
Severe combined immunodeficiencies (SCID) are a group of inherited diseases of the immune system characterized by profound abnormalities of T-cell development. Infants with SCID require prompt clinical intervention to prevent life-threatening infection and studies show significantly improved survival in babies diagnosed at birth based on previous family history. SCID follows the criteria for population-based newborn screening because it is asymptomatic at birth and fatal within the 1st year of life if there is no intervention, the confirmation of the disease is easy, there is a curative treatment, and it is known that early hematopoietic stem cell transplantation significantly improves survival, the quality of immune reconstitution, and quality of life. Quantification of T-cell receptor excision circles (TRECs) in DNA extracted from Guthrie samples is a sensitive and specific screening test for SCID. We conducted a nationwide prospective study of neonatal screening of SCID in a population of 200,000 French newborns over a period of 2 years. The objective was to study the clinical utility and the cost-effectiveness ratio, and to demonstrate that universal SCID screening could result in a substantial benefit to detect individuals, making screening relatively cost-effective in spite of the low incidence of the disease.
重症联合免疫缺陷病(SCID)是一组免疫系统遗传性疾病,其特征为T细胞发育存在严重异常。患有SCID的婴儿需要及时进行临床干预以预防危及生命的感染,研究表明,根据既往家族史在出生时就被诊断出的婴儿存活率显著提高。SCID符合基于人群的新生儿筛查标准,因为它在出生时无症状,如果不进行干预,在生命的第1年内会致命,疾病确诊容易,有治愈性治疗方法,而且已知早期造血干细胞移植可显著提高存活率、免疫重建质量和生活质量。对从干血斑样本中提取的DNA中的T细胞受体切除环(TREC)进行定量分析,是一种针对SCID的敏感且特异的筛查试验。我们在两年时间里对20万名法国新生儿进行了一项全国性的SCID新生儿筛查前瞻性研究。目的是研究其临床实用性和成本效益比,并证明普遍的SCID筛查能使检测出的个体获得实质性益处,尽管该疾病发病率低,但筛查仍具有相对成本效益。