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罗马尼亚 1 型戈谢病患者在酶替代治疗下的脂质谱动态变化:一项前瞻性研究。

Dynamic changes of lipid profile in Romanian patients with Gaucher disease type 1 under enzyme replacement therapy: a prospective study.

机构信息

Department of Endocrinology and Metabolic Diseases, 1st Clinic of Internal Medicine, Johannes Gutenberg University, Langenbeckstr. 1, 55131, Mainz, Germany,

出版信息

J Inherit Metab Dis. 2013 May;36(3):555-63. doi: 10.1007/s10545-012-9529-3. Epub 2012 Sep 14.

Abstract

BACKGROUND

Dyslipidemia in Gaucher disease includes reduced total, low-density lipoprotein (LDL)-, and high-density lipoprotein (HDL)-cholesterol (C). No prospective analysis of lipid profile changes in treatment-naïve patients under enzyme replacement therapy (ERT) is available.

METHODS

We analyzed lipid profile changes during ERT in a prospective controlled manner. Twelve treatment-naïve patients, Gaucher disease type 1 (GD1), 29.5 ± 12.9 years, 4M/8F. Diagnosis was made by enzymatic measurement and mutational analysis. Total-, LDL-, and HDL-C, triglycerides (TG), and LDL subfractions were assessed before the start of ERT with imiglucerase and biannually for 3 years. Patients were matched with healthy controls before and after 3 years of ERT.

RESULTS

At baseline, we found severely reduced HDL-C concentrations (23.6 ± 5.4 mg/dl) and enhanced LDL/HDL ratios (3.1 ± 0.7). HDL-C increased after 6 months (29.2 ± 5.7, p = 0.023), LDL/HDL ratio decreased after 30 months (2.5 ± 0.5, p = 0.039). TG, even not consistently enhanced at baseline (128 ± 31.3 mg/dl), yet higher than in controls (p < 0.001), decreased after 18 months, being comparable with controls after 3 years of ERT. Small, dense LDL (mg/dl) increased continuously without significant difference to controls. After 3 years of ERT, only reduced HDL-C concentrations persisted as a potentially atherogenic alteration; however, mean concentrations markedly improved (42.9 ± 8.3 mg/dl, p < 0.001). Lipid parameters correlated with six markers of disease severity.

CONCLUSIONS

This is the first prospective controlled study regarding lipid profile dynamics during ERT (glucocerebrosidase) in initially treatment-naïve GD1 patients. The most important changes were reduced HDL-C and enhanced LDL/HDL ratio. Their dynamics during ERT and correlations with markers of disease activity suggest that they can be considered markers of disease severity and follow-up in Gaucher patients under treatment.

摘要

背景

戈谢病中的血脂异常包括总胆固醇(C)、低密度脂蛋白(LDL)和高密度脂蛋白(HDL)水平降低。目前尚无关于酶替代疗法(ERT)治疗前未经治疗的患者脂质谱变化的前瞻性分析。

方法

我们以前瞻性对照的方式分析了 ERT 期间的脂质谱变化。12 名未经治疗的戈谢病 1 型(GD1)患者,年龄 29.5 ± 12.9 岁,男女比为 4:8。通过酶测定和突变分析诊断。在开始 ERT 用伊米苷酶治疗之前和之后 3 年每半年评估一次总胆固醇、LDL-、HDL-C、甘油三酯(TG)和 LDL 亚组分。在 ERT 前后与健康对照组进行匹配。

结果

在基线时,我们发现 HDL-C 浓度严重降低(23.6 ± 5.4 mg/dl)和 LDL/HDL 比值升高(3.1 ± 0.7)。6 个月后 HDL-C 增加(29.2 ± 5.7,p = 0.023),30 个月后 LDL/HDL 比值降低(2.5 ± 0.5,p = 0.039)。即使在基线时甘油三酯也没有持续升高(128 ± 31.3 mg/dl),但仍高于对照组(p < 0.001),18 个月后下降,与 ERT 后 3 年的对照组相当。小而密的 LDL(mg/dl)持续增加,但与对照组无显著差异。ERT 后 3 年,只有降低的 HDL-C 浓度持续存在,可能是一种致动脉粥样硬化的改变;然而,平均浓度显著改善(42.9 ± 8.3 mg/dl,p < 0.001)。脂质参数与 6 个疾病严重程度标志物相关。

结论

这是第一项关于初始未经治疗的 GD1 患者 ERT(葡萄糖脑苷脂酶)期间脂质谱动态的前瞻性对照研究。最重要的变化是 HDL-C 降低和 LDL/HDL 比值升高。它们在 ERT 期间的变化及其与疾病活动标志物的相关性表明,它们可以作为戈谢病患者治疗期间疾病严重程度和随访的标志物。

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