Real life experience of tafamidis for the treatment of Spanish patients with Val30Met transthyretin amyloidosis with polyneuropathy
Med Clin (Barc). 2024 May 17;162(9):e27-e32.
doi: 10.1016/j.medcli.2024.01.008.
Epub 2024 Mar 30.
[Article in
English,
Spanish]
Authors
Maria Antonia Ribot Sanso
1
, Adrián Rodriguez Rodriguez
1
, Laura Martínez Vicente
2
, Teresa Sevilla
3
, Cristina Borrachero Garro
4
, Julian Fernández Martín
5
, Adrián Antón Vicente
6
, Moises Morales de la Prida
7
, Lucía Galán Dávila
2
, Laura González Vázquez
8
, Ferran Martínez Valle
6
, Carlos Casasnovas Pons
9
, Arturo Fraga Bau
5
, Eugenia Cisneros Barroso
1
, Inés Losada López
1
, Juan González-Moreno
10
Affiliations
- 1 Servicio de Medicina Interna, Unidad Amiloidosis por Trastirretina, Hospital Universitario Son Llàtzer, Instituto de Investigación Sanitaria Illes Balears (idISBA), Palma de Mallorca, Spain.
- 2 Servicio de Neurología, Unidad de Neuromuscular, IdISSC, Hospital Clínico San Carlos, Madrid, Spain.
- 3 Servicio de Neurología, Hospital Universitari i Politècnic La Fe/IISLAFE, Universitat de Valencia, CIBERER (ERN EURO-NMD), Valencia, Spain.
- 4 Servicio de Medicina Interna, UMAH, Hospital Universitario Juan Ramón Jiménez, Huelva, Spain.
- 5 Servicio de Medicina Interna, Hospital Álvaro Cunqueiro, Vigo, Spain.
- 6 Servicio de Medicina Interna, Hospital Vall d'Hebron, Barcelona, Spain.
- 7 Neuromuscular Unit, Neurology Department, Bellvitge University Hospital-IDIBELL, Spain.
- 8 Servicio de Medicina Interna, Hospital Ribera-Povisa, Vigo, Spain.
- 9 Neuromuscular Unit, Neurology Department, Bellvitge University Hospital-IDIBELL, Spain; Multidisciplinary Unit of Familial Amyloidosis, Bellvitge University Hospital-IDIBELL, Neurometabolic Diseases Group, Bellvitge Biomedical Research Institute (IDIBELL), Barcelona, Spain; Biomedical Research Network Center in Rare Diseases (CIBERER), Valencia, Spain.
- 10 Servicio de Medicina Interna, Unidad Amiloidosis por Trastirretina, Hospital Universitario Son Llàtzer, Instituto de Investigación Sanitaria Illes Balears (idISBA), Palma de Mallorca, Spain. Electronic address: [email protected].
Abstract
Introduction:
Tafamidis is the only approved transthyretin stabiliser approved for the treatment of variant transthyretin amyloidosis (A-ATTRv) related polyneuropathy (PNP). The aim of this study is to analyse the effectiveness of tafamidis in a real-world setting in Spain.
Methods:
This is a national multicenter study in which patients with V30M A-ATTR related PN treated with tafamidis for at least 1 year were included. Clinical, demographic, analytical and neurophysiological variables were analysed.
Results:
100 patients were recruited. Overall, 47 patients (47%) were classified as complete responders, 32 (32%) as partial responders and 21 (21%) as non-responders. The median duration of treatment with tafamidis was 35 months. Better treatment response was shown in patients with in polyneuropathy disability score (PND) I, lower neuropathy impairment score (NIS), compound muscle action potential (CMAP) and Norfolk QoL questionnaire. Higher albumin levels and lower NTproBNP levels were also associated with better treatment response. A basal NIS≥15 predicts that the patient could be a non-responder with a 60% probability.
Conclusions:
Our results reinforce the tafamidis efficacy to treat A-ATTRv-PNP if started early in the disease course. Patients with the V30M variant, NIS<15 and PND I are the most appropriate subjects for this treatment.
Keywords:
ATTR; Amiloidosis por transtirretina; Polineuropatía; Polyneuropathy; Real-life; Tafamidis; Transthyretin amyloidosis; Vida real.
Copyright © 2024 Elsevier España, S.L.U. All rights reserved.
MeSH terms
-
Aged
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Aged, 80 and over
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Amyloid Neuropathies, Familial* / complications
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Amyloid Neuropathies, Familial* / drug therapy
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Benzoxazoles* / therapeutic use
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Female
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Humans
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Male
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Middle Aged
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Peptide Fragments / blood
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Polyneuropathies* / drug therapy
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Polyneuropathies* / etiology
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Prealbumin / genetics
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Spain
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Treatment Outcome
Substances
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tafamidis
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Benzoxazoles
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Prealbumin
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Peptide Fragments
Supplementary concepts
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Amyloidosis, Hereditary, Transthyretin-Related