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遗传性α-胰蛋白酶血症的遗传基础和临床影响。

The Genetic Basis and Clinical Impact of Hereditary Alpha-Tryptasemia.

机构信息

Division of Allergy, Asthma and Immunology, Scripps Clinic, La Jolla, Calif.

Division of Allergy, Asthma and Immunology, Scripps Clinic, La Jolla, Calif.

出版信息

J Allergy Clin Immunol Pract. 2021 Jun;9(6):2235-2242. doi: 10.1016/j.jaip.2021.03.005. Epub 2021 Mar 17.

Abstract

Hereditary alpha-tryptasemia (HαT) is an autosomal dominant genetic trait found in 4% to 6% of the general population and defined by excess copies of alpha-tryptase at TPSAB1. Elevated basal serum tryptase (sBT >8 ng/mL) is a defining feature of HαT and appears to result from increased pro-alpha-tryptase synthesis and secretion rather than mast cell activation. It is estimated that approximately one-third of individuals with HαT have associated symptoms, including cutaneous, gastrointestinal, atopic, musculoskeletal, autonomic, and neuropsychiatric manifestations. HαT is found at a disproportionately high rate in systemic mastocytosis and idiopathic anaphylaxis, and is a modifying factor that independently increases the incidence and severity of anaphylaxis. The varied phenotypes associated with HαT may, in part, result from coinheritance of other genetic variants, increased expression of α-/ß-tryptase heterotetramers, and/or overexpression of pro-alpha-tryptase, although further studies are needed. There is an accurate diagnostic test available to confirm HαT in patients that can be used in combination with sBT to help risk-stratify individuals in whom bone marrow biopsy is being considered. There is no specific treatment for symptoms associated with HαT, and management is focused on controlling clinical manifestations with mast cell mediator antagonists, aspirin, inhalers, epinephrine, omalizumab, and involvement of other specialists.

摘要

遗传性α-胰蛋白酶血症(HαT)是一种常染色体显性遗传特征,在普通人群中发病率为 4%至 6%,其特征是 TPSAB1 处存在α-胰蛋白酶的额外拷贝。基础血清胰蛋白酶升高(sBT>8ng/mL)是 HαT 的一个明确特征,似乎是由于前α-胰蛋白酶的合成和分泌增加而不是肥大细胞激活所致。据估计,大约三分之一的 HαT 患者存在相关症状,包括皮肤、胃肠道、特应性、肌肉骨骼、自主和神经精神表现。HαT 在系统性肥大细胞增多症和特发性过敏反应中发病率极高,是独立增加过敏反应发生率和严重程度的修饰因子。与 HαT 相关的各种表型部分可能是由于其他遗传变异的共同遗传、α-/β-胰蛋白酶异四聚体表达增加和/或前α-胰蛋白酶过表达所致,尽管还需要进一步研究。目前有一项可用于确认 HαT 的准确诊断测试,可与 sBT 联合使用,有助于对考虑进行骨髓活检的个体进行风险分层。目前尚无针对 HαT 相关症状的特定治疗方法,治疗重点是使用肥大细胞介质拮抗剂、阿司匹林、吸入器、肾上腺素、奥马珠单抗以及其他专科医生的参与来控制临床表现。

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