Hereditary transthyretin amyloidosis: a model of medical progress for a fatal disease

被引:0
作者
David Adams
Haruki Koike
Michel Slama
Teresa Coelho
机构
[1] University of Paris-Sud,APHP, National Reference Center for FAP, INSERM U1195
[2] Nagoya University Graduate School of Medicine,Department of Neurology
[3] University of Paris-Sud,APHP, National Reference Center for FAP, Cardiology Department, Hôpital Bichat
[4] Centro Hospitalar do Porto,Andrade’s Center and Neurophysiology Department
来源
Nature Reviews Neurology | 2019年 / 15卷
关键词
D O I
暂无
中图分类号
学科分类号
摘要
Hereditary amyloidogenic transthyretin (ATTRv) amyloidosis with polyneuropathy (also known as familial amyloid polyneuropathy) is a condition with adult onset caused by mutation of transthyretin (TTR) and characterized by extracellular deposition of amyloid and destruction of the somatic and autonomic PNS, leading to loss of autonomy and death. This disease represents a model of the scientific and medical progress of the past 30 years. ATTRv amyloidosis is a worldwide disease with broad genetic and phenotypic heterogeneity that presents a diagnostic challenge for neurologists. The pathophysiology of the neuropathy is increasingly understood and includes instability and proteolysis of mutant TTR leading to deposition of amyloid with variable lengths of fibrils, microangiopathy and involvement of Schwann cells. Wild-type TTR is amyloidogenic in older individuals. The main symptoms are neuropathic, but the disease is systemic; neurologists should be aware of cardiac, eye and kidney involvement that justify a multidisciplinary approach to management. Infiltrative cardiomyopathy is usually latent but present in half of patients. Disease-modifying therapeutics that have been developed include liver transplantation and TTR stabilizers, both of which can slow progression of the disease and increase survival in the early stages. Most recently, gene-silencing drugs have been used to control disease in the more advanced stages and produce some degree of improvement.
引用
收藏
页码:387 / 404
页数:17
相关论文
共 324 条
  • [1] Benson MD(2018)Amyloid nomenclature 2018: recommendations by the International Society of Amyloidosis (ISA) nomenclature committee Amyloid 25 215-219
  • [2] Pihlamaa T(2012)Familial amyloidotic polyneuropathy type IV—gelsolin amyloidosis Amyloid 19 30-33
  • [3] Suominen S(1988)Variant apolipoprotein AI as a major constituent of a human hereditary amyloid Biochem. Biophys. Res. Commun. 156 762-768
  • [4] Kiuru-Enari S(2012)Hereditary systemic amyloidosis due to Asp76Asn variant beta2-microglobulin N. Engl. J. Med. 366 2276-2283
  • [5] Nichols WC(2013)A novel prion disease associated with diarrhea and autonomic neuropathy N. Engl. J. Med. 369 1904-1914
  • [6] Dwulet FE(1952)A peculiar form of peripheral neuropathy; familiar atypical generalized amyloidosis with special involvement of the peripheral nerves Brain 75 408-427
  • [7] Liepnieks J(1984)Type I familial amyloidotic polyneuropathy (Japanese type) Brain Dev. 6 128-133
  • [8] Benson MD(1993)Familial amyloidotic polyneuropathy in Sweden: geographical distribution, age of onset, and prevalence Hum. Hered. 43 288-294
  • [9] Valleix S(1995)Transthyretin gene analysis in European patients with suspected familial amyloid polyneuropathy Brain 118 849-856
  • [10] Mead S(1993)Clinical improvement and amyloid regression after liver transplantation in hereditary transthyretin amyloidosis Lancet 341 1113-1116