| Literature DB >> 30328212 |
Dorota Rowczenio1, Candida C Quarta1, Marianna Fontana1, Carol J Whelan1, Ana Martinez-Naharro1, Hadija Trojer1, Anna Baginska1, Stuart M Ferguson1, Janet Gilbertson1, Tamer Rezk1, Sajitha Sachchithanantham1, Shameem Mahmood1, Richa Manwani1, Faye Sharpley1, Ashutosh D Wechalekar1, Philip N Hawkins1, Julian D Gillmore1, Helen J Lachmann1.
Abstract
Transthyretin amyloidosis (ATTR) is caused by deposition of either wild-type (ATTRwt) or variant (ATTRm) transthyretin. ATTRwt presents with restrictive cardiomyopathy, while ATTRm displays a range of organ involvement. This retrospective analysis includes all patients referred to a single UK center in the last 25 years for clinical and laboratory assessment of known or suspected amyloidosis who underwent TTR gene sequencing. A total of 4459 patients were included in this study; 37% had final diagnosis of ATTR amyloidosis; 27% light chain amyloidosis; 0.7% other types of amyloidosis; 21.3% had no amyloid and 14% had no data. TTR variants were found in 770 (17%) cases; the most prevalent were p.V142I, p.T80A, and p.V50M identified in 42, 25, and 16%, respectively. The median age at referral in each group was: 76 (range 47-93), 66 (40-81), and 45 years (21-86), respectively. Overall 42 rare or novel variants were identified. Forty-two percent patients with ATTRm died at a median age of 73 years (33-89) with a median survival from diagnosis of 50 months. ATTRwt was the final diagnosis in 20% of patients undergoing genetic testing. Our findings of TTR variants in 17% of screened patients highlight the need for routine genetic testing in the evaluation of suspected ATTR amyloidosis.Entities:
Keywords: TTR gene variants; cardiomyopathy; neuropathy; transthyretin amyloidosis (ATTR); variant (ATTRm) transthyretin amyloidosis; wild-type (ATTRwt) transthyretin amyloidosis
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Year: 2018 PMID: 30328212 DOI: 10.1002/humu.23669
Source DB: PubMed Journal: Hum Mutat ISSN: 1059-7794 Impact factor: 4.878