自身免疫淋巴增生性综合征:消失的HDL综合征的罕见原因。

IF 0.7 Q4 IMMUNOLOGY Case Reports in Immunology Pub Date : 2016-01-01 Epub Date: 2016-08-08 DOI:10.1155/2016/7945953
Swetha Sriram, Avni Y Joshi, Vilmarie Rodriguez, Seema Kumar
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引用次数: 5

摘要

HDL消失综合征是指原本HDL- c和甘油三酯水平正常的非危重病人出现严重的高密度脂蛋白胆固醇(HDL- c)缺乏。自身免疫性淋巴细胞增生性综合征(ALPS)是一种由于无法调节淋巴细胞稳态而导致的免疫系统疾病,可导致淋巴结病和肝脾肿大。我们描述了一个17岁的男孩,他在脂质临床评估了不可检测或低HDL-C和低密度脂蛋白胆固醇(LDL-C)水平的历史。过去的病史对于10岁时诊断为双侧宫颈腺病的ALPS IA具有重要意义。已知他在FAS蛋白胞外结构域的一个等位基因中存在与ALPS 1A型一致的错义突变。HDL-C和LDL-C水平在很多情况下都检测不到,尽管在诊断为ALPS之前没有测量过脂质。他一直在接受西罗莫司治疗免疫抑制。HDL-C和LDL-C水平与疾病活动相关,并在控制ALPS活动期间改善到正常水平。本病例强调了在有淋巴增生证据的儿童中,考虑ALPS作为低HDL-C和LDL-C水平的原因的重要性。
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Autoimmune Lymphoproliferative Syndrome: A Rare Cause of Disappearing HDL Syndrome.

The term disappearing HDL syndrome refers to development of severe high density lipoprotein cholesterol (HDL-C) deficiency in noncritically ill patients with previously normal HDL-C and triglyceride levels. Autoimmune lymphoproliferative syndrome (ALPS) is a disorder of the immune system due to an inability to regulate lymphocyte homeostasis resulting in lymphadenopathy and hepatosplenomegaly. We describe a 17-year-old boy who was evaluated in the lipid clinic for history of undetectable or low HDL-C and low density lipoprotein cholesterol (LDL-C) levels. Past medical history was significant for ALPS IA diagnosed at 10 years of age when he presented with bilateral cervical adenopathy. He was known to have a missense mutation in one allele of the FAS protein extracellular domain consistent with ALPS type 1A. HDL-C and LDL-C levels had been undetectable on multiple occasions, though lipids had not been measured prior to the diagnosis of ALPS. He had been receiving sirolimus for immunosuppression. The HDL-C and LDL-C levels correlated with disease activity and improved to normal levels during times when the activity of ALPS was controlled. This case highlights the importance of considering ALPS as a cause of low HDL-C and LDL-C levels in a child with evidence of lymphoproliferation.

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来源期刊
CiteScore
1.90
自引率
0.00%
发文量
14
审稿时长
15 weeks
期刊介绍: Case Reports in Immunology is a peer-reviewed, Open Access journal that publishes case reports and case series related to allergies, immunodeficiencies, autoimmune diseases, immune disorders, cancer immunology and transplantation immunology.
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