Benson Merrill D, Breall Jeffrey, Cummings Oscar W, Liepnieks Juris J
Department of Pathology and Laboratory Medicine, Indiana University School of Medicine, Indianapolis, IN 46202-5126, USA.
Amyloid. 2009 Mar;16(1):9-14. doi: 10.1080/13506120802676914.
Cardiomyopathy is a major cause of death in patients with systemic amyloidosis. There are several forms of systemic amyloidosis which cause cardiomyopathy and determination of the exact type of amyloid in each affected patient is essential for treatment and determination of prognosis. In this study, we tested the feasibility of determining the type of amyloidosis by biochemical analysis of endomyocardial biopsies. Right ventricular endomyocardial biopsies were obtained from 10 patients with restrictive cardiomyopathy. Three patients had monoclonal protein demonstrated in serum or urine and all three had bone marrow findings consistent with monoclonal gammopathy. Seven patients had isolated cardiomyopathy without evidence of monoclonal gammopathy. A portion of each myocardial biopsy was submitted for histologic evaluation and all demonstrated amyloid by Congo red staining. Each biopsy was analysed biochemically by isolation of amyloid fibrils and the protein characterised by amino acid sequence analysis. Four amyloid isolates were characterised as immunoglobulin light chain proteins. Two specimens obtained from patients with transthyretin (TTR) DNA mutations contained TTR peptides proving the hereditary nature of the disease. Biopsies from four patients without a TTR mutation contained TTR and were consistent with the diagnosis of senile cardiac amyloidosis (SCA). All endomyocardial biopsy specimens that were analysed had sufficient amyloid fibril subunit protein to allow characterisation by amino acid sequence analysis. This methodology is particularly useful in differentiating SCA with TTR amyloid fibrils from immunoglobulin light chain amyloidosis which also occurs in the elderly age group.
心肌病是系统性淀粉样变性患者死亡的主要原因。有几种形式的系统性淀粉样变性可导致心肌病,确定每位受影响患者的确切淀粉样蛋白类型对于治疗和预后判断至关重要。在本研究中,我们测试了通过心内膜心肌活检的生化分析来确定淀粉样变性类型的可行性。从10例限制性心肌病患者获取右心室心内膜心肌活检标本。3例患者血清或尿液中检测到单克隆蛋白,且这3例患者的骨髓检查结果均与单克隆丙种球蛋白病相符。7例患者为孤立性心肌病,无单克隆丙种球蛋白病证据。每份心肌活检标本的一部分送去做组织学评估,所有标本经刚果红染色均显示有淀粉样蛋白。通过分离淀粉样纤维对每份活检标本进行生化分析,并通过氨基酸序列分析对蛋白质进行鉴定。4种淀粉样蛋白分离物被鉴定为免疫球蛋白轻链蛋白。从患有转甲状腺素蛋白(TTR)DNA突变的患者获取的两份标本含有TTR肽段,证实了该疾病的遗传性。4例无TTR突变患者的活检标本含有TTR,与老年心脏淀粉样变性(SCA)的诊断相符。所有经分析的心内膜心肌活检标本都有足够的淀粉样纤维亚基蛋白,可通过氨基酸序列分析进行鉴定。这种方法在区分伴有TTR淀粉样纤维的SCA与免疫球蛋白轻链淀粉样变性方面特别有用,后者也发生在老年人群中。