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多米诺肝移植术后获得性转甲状腺素蛋白淀粉样变性:表型相关性及肝再次移植的意义

Acquired transthyretin amyloidosis after domino liver transplant: Phenotypic correlation, implication of liver retransplantation.

作者信息

Muchtar Eli, Grogan Martha, Dasari Surendra, Kurtin Paul J, Gertz Morie A

机构信息

Division of Hematology, Mayo Clinic, Rochester, MN, USA.

Department of Cardiovascular Diseases, Mayo Clinic, Rochester, MN, USA.

出版信息

J Neurol Sci. 2017 Aug 15;379:192-197. doi: 10.1016/j.jns.2017.06.013. Epub 2017 Jun 13.

Abstract

Reports of acquired ATTRm in domino liver transplant (DLT) recipients are infrequent and the approach to management is unknown. We describe two patients, an inherited ATTRm patient who underwent liver transplant and his liver recipient who developed acquired ATTRm 8years following transplantation. The clinical manifestations of amyloidosis were similar between patients and consisted of peripheral neuropathy, diarrhea and weight loss. Following liver transplantation, the DLT donor went into remission for 4years, but eventually progressed and died. The DLT recipient was re-transplanted with a healthy liver, but amyloidosis progressed after 5years, with symptoms resembling the progression of the ATTRm liver donor. Proteomic analysis was utilized to type the amyloid deposits present in the DLT recipient fat aspirate specimen obtained prior to the second transplantation and stomach biopsy obtained at disease progression. The fat amyloid deposits contained almost equal amounts of mutant TTR and wild-type TTR peptides, but the stomach biopsy showed only wild-type TTR. There is a high concordance of disease phenotype between inherited and acquired ATTRm, which may help in early recognition of acquired ATTRm in DLT recipients. Liver re-transplantation for acquired ATTRm may not interrupt the cycle of amyloid deposition since normal hepatically produced TTR may co-deposit with trace amount of pre-existing TTRm amyloid.

摘要

关于多米诺肝移植(DLT)受者获得性甲状腺转运蛋白淀粉样变(ATTRm)的报道并不常见,且管理方法尚不清楚。我们描述了两名患者,一名患有遗传性ATTRm的患者接受了肝移植,其肝移植受者在移植后8年发生了获得性ATTRm。两名患者淀粉样变性的临床表现相似,包括周围神经病变、腹泻和体重减轻。肝移植后,DLT供者病情缓解了4年,但最终病情进展并死亡。DLT受者接受了健康肝脏的再次移植,但5年后淀粉样变性仍进展,症状类似于ATTRm肝供者的病情进展。在第二次移植前获取的DLT受者脂肪抽吸标本以及疾病进展时获取的胃活检标本中,利用蛋白质组学分析对存在的淀粉样沉积物进行分型。脂肪淀粉样沉积物中突变型甲状腺转运蛋白(TTR)和野生型TTR肽的含量几乎相等,但胃活检仅显示野生型TTR。遗传性和获得性ATTRm之间疾病表型高度一致,这可能有助于早期识别DLT受者中的获得性ATTRm。由于正常肝脏产生的TTR可能与微量预先存在的TTRm淀粉样蛋白共同沉积,因此针对获得性ATTRm进行肝再次移植可能无法中断淀粉样蛋白沉积的循环。

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