短期治疗免疫介导的获得性卵磷脂-胆固醇酰基转移酶缺乏症可恢复高密度脂蛋白功能:1例报告

IF 2.8 2区 医学 Q2 PERIPHERAL VASCULAR DISEASE
Tomohiro Komatsu, Satomi Abe, Masayuki Kuroda, Yasuhiro Endo, Kei Sasaki, Takafumi Nishida, Manami Teramoto, Junko Arakawa, Koji Kuwata, Toshihiko Imakiire, Yoshiro Maezawa, Koutaro Yokote, Yoshinari Uehara, Katsunori Ikewaki
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引用次数: 0

摘要

家族性卵磷脂-胆固醇酰基转移酶(LCAT)缺乏伴原发性LCAT基因突变可导致多种疾病,包括角膜混浊、贫血、肾脏疾病和低高密度脂蛋白(HDL)水平。近年来,继发性LCAT缺乏与LCAT自身抗体引起的免疫介导的获得性LCAT缺乏是一种罕见的病例,其症状几乎相同。在有限的情况下,强的松龙治疗对于严重的疾病是必需的,并且已经显示出有利的调节LCAT自身抗体和恢复LCAT活性,导致hdl -胆固醇(HDL-C)水平的改善,肾功能障碍和其他并发症。然而,关于强的松开始治疗后LCAT活性、脂质变化和肾功能障碍的详细信息很少。在本研究中,除了对LCAT活性、脂质和蛋白尿的影响外,我们还首次监测了该疾病患者治疗第一个月期间HDL胆固醇外排能力(CEC),这是一种重要的抗动脉粥样硬化HDL功能。我们发现,在此期间,LCAT活性、HDL- c浓度和HDL- CEC从无法检测到或较低的值增加到正常范围,蛋白尿也是如此。具体而言,HDL- CEC和LCAT活性恢复速度快于HDL- c水平。基于这些发现,强的松龙治疗在HDL- c水平之前对LCAT和HDL- CEC活性的影响表明,对于需要治疗的免疫介导的获得性LCAT缺乏症患者,正常的HDL- c水平可能不是必需的治疗靶点。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Short-Term Treatment for Immune-Mediated Acquired Lecithin-Cholesterol Acyltransferase Deficiency Restores the High-Density Lipoprotein Function: A Case Report.

Familial lecithin-cholesterol acyltransferase (LCAT) deficiency with a primary LCAT gene mutation results in various conditions, including corneal opacity, anemia, kidney disease, and low high-density lipoprotein (HDL) levels. In recent years, secondary LCAT deficiency with nearly identical symptoms has been identified as a rare case of immune-mediated acquired LCAT deficiency caused by LCAT autoantibodies. In limited cases, prednisolone treatment is required for severe conditions and has been shown to favorably modulate LCAT autoantibodies and restore LCAT activity, resulting in improved HDL-cholesterol (HDL-C) levels, renal dysfunction, and other complications. However, there is little detailed information regarding LCAT activity, lipid changes, and renal dysfunction after the initiation of prednisorone treatment. In the present study, in addition to the effects on LCAT activity, lipids, and proteinuria, we for the first time monitored the HDL cholesterol efflux capacity (CEC), an important anti-atherosclerotic HDL function, during the first month of treatment in a patient with this disease. We found that the LCAT activity, HDL-C concentration, and HDL CEC increased from undetectable or low values to normal ranges during this period, as did proteinuria. Specifically, the HDL CEC and LCAT activity recovered faster than the HDL-C levels. Based on these findings, the effects of prednisolone treatment on LCAT and HDL CEC activities prior to HDL-C levels suggest that normal HDL-C levels may not be essential as a treatment target in immune-mediated acquired LCAT deficiency patients who require treatment.

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来源期刊
CiteScore
6.60
自引率
15.90%
发文量
271
审稿时长
1 months
期刊介绍: JAT publishes articles focused on all aspects of research on atherosclerosis, vascular biology, thrombosis, lipid and metabolism.
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