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首例40多岁男性双遗传性凝溶胶蛋白和转甲状腺素蛋白野生型心脏淀粉样变性病例报告

First Reported Case of Dual Hereditary Gelsolin and Transthyretin Wild-Type Cardiac Amyloidosis in a Man in his late 40s.

作者信息

Theis Jeanne L, Dasari Surendra, Theis Jason D, Vrana Julie A, Hasadsri Linda, Fernandez Joel, McPhail Ellen D

机构信息

Division of Laboratory Genetics and Genomics, Mayo Clinic, Rochester, MN.

Division of Computational Biology, Mayo Clinic, Rochester, MN.

出版信息

Mayo Clin Proc Innov Qual Outcomes. 2025 Aug 19;9(5):100648. doi: 10.1016/j.mayocpiqo.2025.100648. eCollection 2025 Oct.

DOI:10.1016/j.mayocpiqo.2025.100648
PMID:40896811
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12396264/
Abstract

Amyloidosis is a group of disorders characterized by abnormal deposition of amyloid proteins in various tissues and organs, leading to progressive organ dysfunction. With over 40 precursor proteins linked to amyloid formation, identification of the amyloid type is critical to guide treatment. A man in his late 40s presenting with heart failure was diagnosed with cardiac amyloidosis based on an endomyocardial biopsy. Amyloid typing performed on the heart biopsy at Mayo Clinic Laboratories using differential laser microdissection and shotgun proteomics with mass spectrometry reported gelsolin amyloid (AGel) deposits exclusively in the vasculature and transthyretin amyloid deposits exclusively within the interstitium. Mutational analysis identified a novel p.Y474N in the gelsolin gene, establishing a diagnosis of hereditary AGel amyloidosis. Transthyretin gene mutations were absent, confirming a concurrent diagnosis of acquired transthyretin wild-type amyloidosis. The patient, who had been treated with guideline-directed medical therapy since his initial presentation, was subsequently started on tafamidis, with subsequent improvement of his ejection fraction after 6-7 months. Although rare, 2 different amyloid types may arise in the same anatomic site. Identification of all amyloid types is crucial for optimal patient management. In this case, the co-existence of 2 rare amyloid types (AGel with a novel mutation coupled with ATTRwt in a patient under 50 years of age) in mutually exclusive anatomic compartments in the same cardiac biopsy raises the possibility that an unknown systemic factor may play a role in amyloidogenesis in dual amyloid cases.

摘要

淀粉样变性是一组以淀粉样蛋白在各种组织和器官中异常沉积为特征的疾病,可导致进行性器官功能障碍。有40多种前体蛋白与淀粉样蛋白形成有关,确定淀粉样蛋白类型对于指导治疗至关重要。一名40多岁出现心力衰竭的男性经心内膜心肌活检被诊断为心脏淀粉样变性。在梅奥诊所实验室对心脏活检组织进行的淀粉样蛋白分型,采用差分激光显微切割和基于质谱的鸟枪法蛋白质组学,报告凝溶胶蛋白淀粉样变性(AGel)仅沉积于脉管系统,而转甲状腺素蛋白淀粉样变性仅沉积于间质。突变分析在凝溶胶蛋白基因中鉴定出一种新的p.Y474N突变,确诊为遗传性AGel淀粉样变性。未检测到转甲状腺素蛋白基因突变,证实同时诊断为获得性转甲状腺素蛋白野生型淀粉样变性。该患者自首次就诊以来一直接受指南指导的药物治疗,随后开始服用塔非酰胺,6至7个月后射血分数有所改善。虽然罕见,但同一解剖部位可能出现两种不同类型的淀粉样蛋白。识别所有淀粉样蛋白类型对于优化患者管理至关重要。在本病例中,同一份心脏活检组织中,两种罕见的淀粉样蛋白类型(一种具有新突变的AGel和50岁以下患者中的ATTRwt)在相互排斥的解剖区域共存,这增加了一种未知的全身因素可能在双重淀粉样变性病例的淀粉样蛋白生成中起作用的可能性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/40c6/12396264/c1b574df644a/gr2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/40c6/12396264/a15aee5beb7f/gr1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/40c6/12396264/c1b574df644a/gr2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/40c6/12396264/a15aee5beb7f/gr1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/40c6/12396264/c1b574df644a/gr2.jpg

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本文引用的文献

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Hereditary gelsolin amyloidosis: a rare cause of cranial, peripheral and autonomic neuropathies linked to D187N and Y447H substitutions.遗传性凝胶蛋白淀粉样变性:一种与 D187N 和 Y447H 替换相关的导致颅神经、周围神经和自主神经病的罕见病因。
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