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未经治疗的原发性肉碱缺乏症导致猝死风险增加。

Increased risk of sudden death in untreated primary carnitine deficiency.

机构信息

Department of Internal Medicine, National Hospital, Torshavn, Faroe Islands.

Department of Cardiology, Rigshospitalet University Hospital, Copenhagen, Denmark.

出版信息

J Inherit Metab Dis. 2020 Mar;43(2):290-296. doi: 10.1002/jimd.12158. Epub 2019 Dec 15.

Abstract

Primary carnitine deficiency (PCD) affects fatty acid oxidation and is associated with cardiomyopathy and cardiac arrhythmia, but the risk of sudden death in PCD is unknown. The Faroe Islands have a high prevalence of PCD, 1:300. This study systematically investigated a possible association between untreated PCD and sudden death in young Faroese subjects. We investigated all medico-legal cases of sudden death between 1979 and 2012 among subjects below the age of 45. Stored biomaterial was examined with molecular genetic analysis to reveal PCD. We compared the prevalence of PCD among sudden death cases with that of the background population (0.23%) to calculate the odds ratio (OR) for sudden death with PCD. Biomaterial was available and genetically analyzed from 53 of 65 sudden death cases (82%) in the Faroe Islands. Six (one male and five females) of the 53 cases were homozygous for the PCD related c.95A>G mutation-a prevalence of 11.3% (95% CI 5%-23%) and an OR of 54.3 (95% CI 21-138, P < .0001) for the association between sudden death and untreated PCD. Only 11 of the 53 sudden death cases were women-of whom five were homozygous for the c.95A>G mutation (45.5%) yielding an OR of 348.8 (95% CI 94-1287, P < .0001) for the association between sudden death and untreated PCD in females. This study showed a strong association between sudden death and untreated PCD, especially in females.

摘要

原发性肉碱缺乏症(PCD)影响脂肪酸氧化,与心肌病和心律失常有关,但 PCD 患者发生猝死的风险尚不清楚。法罗群岛 PCD 的患病率很高,为 1:300。本研究系统地调查了未经治疗的 PCD 与法罗群岛年轻受试者猝死之间可能存在的关联。我们调查了 1979 年至 2012 年期间年龄在 45 岁以下的所有法罗群岛法医学猝死案例。通过分子遗传学分析检查储存的生物材料以揭示 PCD。我们将猝死病例中 PCD 的患病率与背景人群(0.23%)进行比较,以计算 PCD 与猝死的比值比(OR)。法罗群岛共有 65 例猝死案例(82%)的生物材料可用且经基因分析,其中 6 例(1 例男性和 5 例女性)为 PCD 相关 c.95A>G 突变的纯合子-患病率为 11.3%(95%CI 5%-23%),OR 为 54.3(95%CI 21-138,P<0.0001),提示未治疗的 PCD 与猝死之间存在关联。在 65 例猝死案例中仅有 11 例为女性,其中 5 例为 c.95A>G 突变的纯合子(45.5%),OR 为 348.8(95%CI 94-1287,P<0.0001),提示未治疗的 PCD 与女性猝死之间存在关联。本研究表明,未治疗的 PCD 与猝死之间存在强烈关联,尤其是在女性中。

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