Lancet Child & Adolescent Health最新文献_第7页

Cardiovascular outcomes in patients with homozygous familial hypercholesterolaemia on lipoprotein apheresis initiated during childhood: long-term follow-up of an international cohort from two registries 童年时期开始接受脂蛋白分离治疗的同型家族性高胆固醇血症患者的心血管预后：两个登记处国际队列的长期随访。

IF 36.4 1区医学 Q1 Psychology

Lancet Child & Adolescent Health

Pub Date : 2024-05-14 DOI: 10.1016/S2352-4642(24)00073-7

M Doortje Reijman MD , Tycho R Tromp PhD , Barbara A Hutten PhD , Prof G Kees Hovingh PhD , Dirk J Blom PhD , Prof Alberico L Catapano PhD , Marina Cuchel PhD , Prof Eldad J Dann MD , Antonio Gallo PhD , Lisa C Hudgins MD , Prof Frederick J Raal PhD , Prof Kausik K Ray FMedSci , Fouzia Sadiq PhD , Handrean Soran MD , Prof Jaap W Groothoff PhD , Albert Wiegman PhD , D Meeike Kusters PhD , Homozygous Familial Hypercholesterolaemia International Clinical Collaborators (HICC) , Children with Homozygous Hypercholesterolemia on Lipoprotein Apheresis: an International Registry (CHAIN) consortia

Background

Homozygous familial hypercholesterolaemia (HoFH) is a rare genetic disease characterised by extremely high plasma LDL cholesterol from birth, causing atherosclerotic cardiovascular disease at a young age. Lipoprotein apheresis in combination with lipid-lowering drugs effectively reduce LDL cholesterol, but long-term health outcomes of such treatment are unknown. We aimed to investigate the long-term cardiovascular outcomes associated with lipoprotein apheresis initiated in childhood or adolescence.

Methods

In this cohort study, data were drawn from the HoFH International Clinical Collaboration (HICC) and the international registry for Children with Homozygous Hypercholesterolemia on Lipoprotein Apheresis (CHAIN). An overall cohort included patients diagnosed with HoFH aged 0–18 years who were alive and in follow-up between Jan 1, 2010, and Nov 8, 2021, and whose high plasma LDL cholesterol concentrations made them eligible for lipoprotein apheresis. To compare cardiovascular outcomes, patients who initiated lipoprotein apheresis in childhood (lipoprotein apheresis group) and patients who only received lipid-lowering drugs (pharmacotherapy-only group) were matched by sex and untreated plasma LDL cholesterol concentrations. The primary outcome was a composite of cardiovascular death, myocardial infarction, ischaemic stroke, percutaneous coronary intervention, coronary artery bypass grafting, aortic valve replacement, peripheral artery disease, carotid endarterectomy, angina pectoris, and supra-aortic or aortic stenosis (collectively referred to as atherosclerotic cardiovascular disease), for which survival analyses were performed in the matched cohort. Cox regression analyses were used to compare disease-free survival between cohorts and to calculate hazard ratio (HR) and 95% CI adjusted for sex, age at diagnosis, untreated plasma LDL cholesterol concentration, and number of lipid-lowering therapies other than lipoprotein apheresis.

Findings

The overall cohort included 404 patients with a median age at diagnosis of 6·0 years (IQR 3·0–9·5) and median untreated plasma LDL cholesterol of 17·8 mmol/L (14·7–20·8). The matched cohorts included 250 patients (125 patients per group), with a median untreated LDL cholesterol of 17·2 mmol/L (14·8–19·7). Mean reduction in plasma LDL cholesterol concentrations between baseline and final follow-up was greater in the lipoprotein apheresis group (–55% [95% CI –60 to –51] vs –31% [–36 to –25]; p<0·0001). Patients in the lipoprotein apheresis group had longer atherosclerotic cardiovascular disease-free survival (adjusted HR 0·52 [95% CI 0·32–0·85]) and longer cardiovascular death-free survival (0·0301 [0·0021–0·4295]). Cardiovascular death was more common in the pharmacotherapy-only group than in the lipoprotein apheresis group (ten [8%] vs one [1%]; p=0·010), whereas median age at coronary artery bypass grafting was lo

背景杂合子家族性高胆固醇血症（HoFH）是一种罕见的遗传性疾病，其特点是出生时血浆中的低密度脂蛋白胆固醇就极高，导致患者在年轻时就患上动脉粥样硬化性心血管疾病。脂蛋白清除术与降脂药物联合使用可有效降低低密度脂蛋白胆固醇，但这种治疗的长期健康效果尚不清楚。方法在这项队列研究中，数据来自HoFH国际临床合作组织（HICC）和 "接受脂蛋白清除术的高胆固醇血症儿童 "国际登记处（CHAIN）。总体队列包括2010年1月1日至2021年11月8日期间存活并接受随访的0-18岁确诊为HoFH的患者，这些患者的血浆低密度脂蛋白胆固醇浓度较高，因此符合脂蛋白分离治疗的条件。为了比较心血管预后，在儿童期开始接受脂蛋白清除术的患者（脂蛋白清除术组）和只接受降脂药物治疗的患者（单纯药物治疗组）按性别和未经治疗的血浆低密度脂蛋白胆固醇浓度进行了配对。主要结果是心血管死亡、心肌梗死、缺血性中风、经皮冠状动脉介入治疗、冠状动脉旁路移植术、主动脉瓣置换术、外周动脉疾病、颈动脉内膜切除术、心绞痛、主动脉上或主动脉狭窄（统称为动脉粥样硬化性心血管疾病）的综合结果，并对匹配队列进行生存分析。结果总体队列包括 404 名患者，诊断时的中位年龄为 6-0 岁（IQR 3-0-9-5），未经治疗的血浆低密度脂蛋白胆固醇中位数为 17-8 mmol/L (14-7-20-8)。配对队列包括 250 名患者（每组 125 名患者），未经治疗的低密度脂蛋白胆固醇中位数为 17-2 mmol/L (14-8-19-7)。脂蛋白分离组患者从基线到最终随访期间血浆低密度脂蛋白胆固醇浓度的平均降幅更大（-55% [95% CI -60至-51] vs -31% [-36至-25]；p<0-0001）。脂蛋白清除组患者无动脉粥样硬化性心血管疾病生存期更长（调整后 HR 0-52 [95% CI 0-32-0-85] ），无心血管疾病死亡生存期更长（0-0301 [0-0021-0-4295]）。单纯药物治疗组心血管死亡的发生率高于脂蛋白分离组（10 [8%] vs 1 [1%]；P=0-010），而脂蛋白分离组冠状动脉搭桥术的中位年龄低于单纯药物治疗组（15-0岁 [IQR 12-0-24-0] vs 30-5岁 [19-0-33-8]；P=0-037）。解读在HoFH患者中，儿童和青少年时期开始进行脂蛋白清除术与降低动脉粥样硬化性心血管疾病和死亡的长期风险有关，并且发现早期开始高频治疗对降低血浆胆固醇有明显益处。现在需要共识建议来指导更广泛、更及时地使用脂蛋白清除术治疗HoFH患儿，还需要开展研究来进一步优化治疗，确保早期积极治疗的益处与对生活质量的影响相平衡。

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引用次数: 0

Who decides what is “good for” the children? 谁来决定什么对孩子 "好"？

IF 36.4 1区医学 Q1 Psychology

Lancet Child & Adolescent Health

Pub Date : 2024-05-14 DOI: 10.1016/S2352-4642(24)00113-5

Kelley Swain

引用次数: 0

Monitoring change from residential housing care to family-based care for children 监测儿童从寄宿照料向家庭照料的转变。

IF 19.9 1区医学 Q1 PEDIATRICS

Lancet Child & Adolescent Health

Pub Date : 2024-05-14 DOI: 10.1016/S2352-4642(24)00102-0

引用次数: 0

Emma Alesna Llanto: taking the long road to lasting change Emma Alesna Llanto：走持久变革的漫长之路。

IF 19.9 1区医学 Q1 PEDIATRICS

Lancet Child & Adolescent Health

Pub Date : 2024-05-11 DOI: 10.1016/S2352-4642(24)00126-3

引用次数: 0

Eliminating hepatitis B: the ball is in our court 消灭乙型肝炎：球在我们手中。

IF 36.4 1区医学 Q1 Psychology

Lancet Child & Adolescent Health

Pub Date : 2024-05-03 DOI: 10.1016/S2352-4642(24)00112-3

The Lancet Child & Adolescent Health

引用次数: 0

The value of registries for rare and severe adverse events in paediatrics 儿科罕见和严重不良事件登记的价值。

IF 36.4 1区医学 Q1 Psychology

Lancet Child & Adolescent Health

Pub Date : 2024-04-30 DOI: 10.1016/S2352-4642(24)00106-8

Robin J Prescott

引用次数: 0

Rare and severe adverse events in children with inflammatory bowel disease: analysis of data from the PIBD-SETQuality Safety Registry 炎症性肠病患儿的罕见和严重不良事件：PIBD-SETQuality 安全登记数据分析。

IF 36.4 1区医学 Q1 Psychology

Lancet Child & Adolescent Health

Pub Date : 2024-04-30 DOI: 10.1016/S2352-4642(24)00078-6

Renz C W Klomberg MD , Astrid E Hellendoorn MD , Polychronis Kemos MSc , Dimitris Rizopoulos PhD , Prof Frank M Ruemmele PhD , Prof Nicholas M Croft PhD , Lissy de Ridder PhD

Background

Rare and severe adverse events can occur in children with inflammatory bowel disease (IBD), and the relationship with disease or drug treatment is often uncertain. We aimed to establish a method of reporting adverse events of interest in children with IBD, allowing for estimates of incidence rates with comparison between different regions, and, if possible, to compare with published data on rates of adverse events in children overall.

Methods

For this analysis, we used data from the Paediatric Inflammatory Bowel Disease Network for Safety, Efficacy and Treatment and Quality improvement of care (PIBD-SETQuality) Safety Registry, which collects data on multiple rare and severe adverse events in children younger than 19 years with IBD. Overall, the registry collected data on ten prespecified rare and severe adverse events in children with IBD, as established by a panel of paediatric IBD experts, via reports from paediatric gastroenterologists at participating hospitals between Nov 1, 2016, and March 31, 2023. Reporting physicians, who could only be paediatric gastroenterologists or IBD nurses reporting on behalf of paediatric gastroenterologists, were recruited through invitations sent to both national and international IBD networks and at conferences. Once per month, participating paediatric gastroenterologists received an email with an anonymous and unique link to an online survey asking them to report whether any of ten rare and severe adverse events had occurred in a patient in their paediatric-IBD population in the previous month. Prevalent or retrospective rare and severe adverse events were excluded, as were events occurring in children with an unconfirmed diagnosis of IBD or for whom inflammatory colitis was part of a monogenic immunodeficiency disorder. Duplicates and events that did not meet the definitions and criteria were excluded. Physicians could also report other, non-categorised adverse events if they considered them rare and severe. In case of no response, up to two reminders were sent for each per-month survey. Annual denominator data surveys were sent to obtain the total number of person-years for the estimation of incidence rates, which were calculated via Poisson regression models.

Findings

Responses were gathered from 220 paediatric gastroenterologists from 167 centres. 121 centres were in Europe, 23 centres were in North America, 17 centres were in Asia, and six centres were in Oceania. Combined, the total population with paediatric IBD consisted of an estimated 30 193 children with 114 528 person-years of follow-up. 451 adverse events were initially reported. After excluding and reorganising adverse events, 402 were eligible; 261 (65%) were categorised and 141 (35%) were non-categorised. The most frequently reported adverse events were venous-thromboembolic events (n=66), renal failure (n=43), opportunistic infections (n=42), and cancer (n=33). Haem

背景：炎症性肠病（IBD）患儿可能会发生罕见的严重不良事件，而这些不良事件与疾病或药物治疗之间的关系往往并不确定。我们旨在建立一种报告 IBD 儿童相关不良事件的方法，以便估算发病率并在不同地区之间进行比较，并在可能的情况下与已公布的儿童总体不良事件发生率数据进行比较：在这项分析中，我们使用了儿科炎症性肠病安全、疗效和治疗及护理质量改进网络（PIBD-SETQuality）安全登记处的数据，该登记处收集了19岁以下IBD患儿的多种罕见和严重不良事件的数据。总体而言，该登记处通过参与医院的儿科消化科医生在2016年11月1日至2023年3月31日期间提交的报告，收集了儿科IBD专家小组确定的十种IBD患儿预设罕见和严重不良事件的数据。报告医生只能是儿科胃肠病专家或代表儿科胃肠病专家报告的 IBD 护士，他们是通过向国内和国际 IBD 网络发出邀请以及在会议上招募的。参与调查的儿科胃肠病医生每月都会收到一封电子邮件，其中包含一个匿名和唯一的在线调查链接，要求他们报告其儿科 IBD 患者在上个月是否发生了十种罕见和严重不良事件中的任何一种。普遍或回顾性罕见和严重不良事件以及发生在未经确诊为 IBD 或炎症性结肠炎属于单基因免疫缺陷疾病的儿童身上的事件均被排除在外。重复和不符合定义和标准的事件也被排除在外。如果医生认为其他未分类的不良事件比较罕见和严重，也可以报告。如果没有回复，每个月的调查最多会发送两封提醒邮件。每年进行一次分母数据调查，以获得用于估算发病率的总人数-年，发病率通过泊松回归模型计算得出：共收集到来自 167 个中心的 220 名儿科胃肠病专家的回复。欧洲有 121 个中心，北美有 23 个中心，亚洲有 17 个中心，大洋洲有 6 个中心。儿童 IBD 患者总人数约为 30 193 人，随访时间为 114 528 人年。最初报告了 451 例不良事件。在排除和重组不良事件后，有402例符合条件；其中261例（65%）被归类，141例（35%）未被归类。最常报告的不良事件是静脉血栓栓塞事件（66 例）、肾功能衰竭（43 例）、机会性感染（42 例）和癌症（33 例）。嗜血细胞淋巴组织细胞增多症（4 例）和肝功能衰竭（3 例）是报告最少的不良事件。静脉血栓栓塞事件每万人年的发病率为 5-50 (95% CI 4-25-6-97) ，肾衰竭为 3-75 (2-74-4-99)，机会性感染为 3-67 (2-67-4-89)，癌症为 2-88 (2-01-3-98)。在 66 例静脉血栓栓塞事件中，31 例（47%）涉及脑静脉窦血栓，发生率为 2-71 (95% CI 1-86-3-77)：PIBD-SETQuality安全性注册使我们能够确定IBD患儿罕见和严重不良事件的发生率。我们的研究结果可以为医生提供指导，并提高他们对IBD患儿中被认为罕见的不良事件发生率的认识：欧盟地平线2020研究与创新计划。

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引用次数: 0

Come and learn: an invitation from Zvandiri Youth Advocates 来学习：兹万迪里青年倡导者协会的邀请函

IF 36.4 1区医学 Q1 Psychology

Lancet Child & Adolescent Health

Pub Date : 2024-04-25 DOI: 10.1016/S2352-4642(24)00111-1

Loyce Maturu, Paul Mavesere Ndhlovu, Melissa Chiduke, Blessed Ackim Mazvikite

引用次数: 0

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