World Heart Federation, US.
Geisinger Health, US.
Glob Heart. 2024 May 3;19(1):43. doi: 10.5334/gh.1316. eCollection 2024.
Homozygous familial hypercholesterolemia (HoFH) is an ultra-rare inherited condition that affects approximately one in 300,000 people. The disorder is characterized by extremely high, life-threatening levels of low-density lipoprotein (LDL) cholesterol from birth, leading to significant premature cardiovascular morbidity and mortality, if left untreated. Homozygous familial hypercholesterolemia is severely underdiagnosed and undertreated in the United States (US), despite guidelines recommendations for universal pediatric lipid screening in children aged 9-11. Early diagnosis and adequate treatment are critical in averting premature cardiovascular disease in individuals affected by HoFH. Yet, an unacceptably high number of people living with HoFH remain undiagnosed, misdiagnosed, and/or receive a late diagnosis, often after a major cardiovascular event. The emergence of novel lipid-lowering therapies, along with the realization that diagnosis is too often delayed, have highlighted an urgency to implement policies that ensure timely detection of HoFH in the US. Evidence from around the world suggests that a combination of universal pediatric screening and cascade screening strategies constitutes an effective approach to identifying heterozygous familial hypercholesterolemia (HeFH). Nevertheless, HoFH and its complications manifest much earlier in life compared to HeFH. To date, little focus has been placed on the detection of HoFH in very young children and/or infants. The 2023 Updated European Atherosclerosis Society Consensus Statement on HoFH has recommended, for the first time, broadening pediatric guidelines to include lipid screening of newborn infants. Some unique aspects of HoFH need to be considered before implementing newborn screening. As such, insights from pilot studies conducted in Europe may provide some preliminary guidance. Our paper proposes a set of actionable measures that states can implement to reduce the burden of HoFH. It also outlines key research and policy gaps that need to be addressed in order to pave the way for universal newborn screening of HoFH in the US.
纯合子家族性高胆固醇血症(HoFH)是一种极为罕见的遗传性疾病,影响大约每 30 万人中有 1 人。这种疾病的特征是从出生起就存在极高的、危及生命的低密度脂蛋白(LDL)胆固醇水平,导致如果不进行治疗,会出现显著的早发性心血管发病率和死亡率。尽管指南建议对 9-11 岁的儿童进行普遍儿科血脂筛查,但纯合子家族性高胆固醇血症在美国(US)仍严重漏诊和治疗不足。早期诊断和充分治疗对于避免 HoFH 患者的早发性心血管疾病至关重要。然而,仍有相当数量的 HoFH 患者未被诊断、误诊和/或延迟诊断,通常是在发生重大心血管事件之后。新型降脂治疗方法的出现,以及认识到诊断经常被延迟,突显了在美国实施确保及时发现 HoFH 的政策的紧迫性。世界各地的证据表明,普遍的儿科筛查和级联筛查策略相结合是识别杂合子家族性高胆固醇血症(HeFH)的有效方法。然而,HoFH 及其并发症在生命早期的表现要比 HeFH 早得多。迄今为止,人们很少关注非常年幼的儿童和/或婴儿的 HoFH 检测。2023 年更新的欧洲动脉粥样硬化学会 HoFH 共识声明首次建议扩大儿科指南,包括对新生儿进行血脂筛查。在实施新生儿筛查之前,需要考虑 HoFH 的一些独特方面。因此,欧洲进行的试点研究的结果可能提供一些初步的指导。我们的论文提出了一系列可行的措施,各州可以实施这些措施来减轻 HoFH 的负担。它还概述了需要解决的关键研究和政策差距,为在美国普遍开展 HoFH 新生儿筛查铺平道路。