Pediatric Clinic 1, Department of Pathophysiology and Transplantation, Università degli Studi di Milano, Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Milan, Italy.
Am J Med Genet A. 2013 Jul;161A(7):1666-74. doi: 10.1002/ajmg.a.35982. Epub 2013 May 21.
There are between 5,000 and 8,000 distinct rare diseases (RDs) affecting 6-8% of the population, most of which are caused by genetic defects. Many are highly complex, childhood-onset, multi-system disorders that are often associated with developmental disability, and require lifelong, highly specialized care and support. As larger numbers of children with previously fatal RDs survive into adulthood, they encounter significant challenges in transitioning from family-centered, developmentally focused, multidisciplinary pediatric care to a less supportive adult healthcare system that is often unfamiliar with these conditions. This paper discusses the challenges of the transition from pediatric to adult health care in two groups of patients with multisystem genetic RDs (neurofibromatosis 1 [NF1] and Williams-Beuren syndrome [WBS]), and analyzes strategies for making the process easier for patients with and without developmental disabilities. Our findings show that there are still no guidelines in national healthcare programs on how to transition RD adolescents with and without developmental disabilities, and only a few pediatric centers have implemented the elements of transition in their general practice. Evidence regarding programs to facilitate transition is inconclusive and the transition from pediatric medicine to adult medicine for RDs remains a major challenge. However, transition requires both time and personnel, which are difficult to find in periods of fiscal austerity. Nevertheless, we should strongly advocate for governments investing more into transition infrastructure or they will face increased long-term social and economic costs due to poor treatment compliance, disengagement from services, increased genetic risks, and higher rates of disease-related complications.
有 5000 到 8000 种不同的罕见病(RDs)影响着 6-8%的人口,其中大多数是由遗传缺陷引起的。许多 RD 是高度复杂的、儿童期发病的、多系统疾病,通常伴有发育障碍,需要终身、高度专业化的护理和支持。随着越来越多的患有以前致命 RD 的儿童存活到成年期,他们在从以家庭为中心、以发育为重点、多学科的儿科护理过渡到支持力度较小的成人医疗保健系统时,面临着重大挑战,而后者往往不熟悉这些疾病。本文讨论了两组多系统遗传 RD 患者(神经纤维瘤病 1 [NF1]和威廉姆斯-贝伦综合征 [WBS])从儿科到成人保健的过渡挑战,并分析了使无发育障碍和有发育障碍的患者更容易过渡的策略。我们的研究结果表明,国家医疗保健计划中仍然没有关于如何为有和没有发育障碍的 RD 青少年过渡的指南,只有少数儿科中心在其常规实践中实施了过渡的要素。关于促进过渡的项目的证据尚无定论,RD 从儿科医学到成人医学的过渡仍然是一个主要挑战。然而,过渡既需要时间,也需要人力,而在财政紧缩时期,这两者都很难找到。尽管如此,我们应该强烈主张政府投入更多资金用于过渡基础设施,否则,由于治疗依从性差、脱离服务、遗传风险增加以及与疾病相关的并发症发生率上升,政府将面临更高的长期社会和经济成本。