Department of Internal Medicine I, Johannes Gutenberg University, Langenbeckstrasse 1, 55101 Mainz, Germany.
Liver Transpl. 2010 Mar;16(3):314-23. doi: 10.1002/lt.21996.
Liver transplantation (LT) is the only curative option for patients with familial amyloid polyneuropathy (FAP) at present. Twenty patients with FAP underwent LT between May 1998 and June 2007. Transthyretin mutations included predominantly the Val30Met mutation but also 10 other mutations. Seven patients received a pacemaker prior to LT, and because of impairment of mechanical cardiac function, 4 combined heart-liver transplants were performed, 1 simultaneously and 3 sequentially. The first patient, who underwent simultaneous transplantation, died. Seven patients died after LT, with 5 dying within the first year after transplantation. The causes of death were cardiac complications (4 patients), infections (2 patients), and malnutrition (1 patient). One-year survival was 75.0%, and 5-year survival was 64.2%. Gly47Glu and Leu12Pro mutations showed an aggressive clinical manifestation: 2 patients with the Gly47Glu mutation, the youngest patients of all the non-Val30Met patients, suffered from severe cardiac symptoms leading to death despite LT. Two siblings with the Leu12Pro mutation, who presented only with grand mal seizures, died after LT because of sepsis. In conclusion, the clinical course in patients with FAP is very variable. Cardiac symptoms occurred predominantly in patients with non-Val30Met mutations and prompted combined heart-liver transplantation in 4 patients. Although early LT in Val30Met is indicated in order to halt the typical symptoms of polyneuropathy, additional complications occurring predominantly with other mutations may prevail and lead to life-threatening complications or a fatal outcome. Combined heart-liver transplantation should be considered in patients with restrictive cardiomyopathy.
肝移植(LT)是目前治疗家族性淀粉样多神经病(FAP)患者的唯一根治方法。1998 年 5 月至 2007 年 6 月期间,20 例 FAP 患者接受了 LT。转甲状腺素突变主要包括 Val30Met 突变,但也有 10 个其他突变。7 例患者在 LT 前安装了起搏器,由于机械心脏功能受损,4 例联合进行了心脏-肝移植,1 例同时进行,3 例序贯进行。第一例同时接受移植的患者死亡。LT 后 7 例患者死亡,其中 5 例在移植后 1 年内死亡。死亡原因是心脏并发症(4 例)、感染(2 例)和营养不良(1 例)。1 年生存率为 75.0%,5 年生存率为 64.2%。Gly47Glu 和 Leu12Pro 突变表现出侵袭性的临床表现:2 例 Gly47Glu 突变患者,是所有非 Val30Met 患者中最年轻的,尽管接受了 LT,但仍因严重的心脏症状而死亡。2 例 Leu12Pro 突变的同胞,仅表现为全身强直阵挛发作,LT 后因败血症死亡。总之,FAP 患者的临床表现非常多变。非 Val30Met 突变患者主要出现心脏症状,并促使 4 例患者进行联合心脏-肝移植。虽然早期对 Val30Met 进行 LT 是为了阻止多发性神经病的典型症状,但其他突变主要引起的额外并发症可能占主导地位,并导致危及生命的并发症或致命结局。对于限制性心肌病患者,应考虑联合心脏-肝移植。