Screening for Fabry disease in unknown origin axonal polyneuropathy: to do or not to do, this is the question!
Axonal
Fabry disease
Lysosomal disorder
Neuropathy
Polyneuropathy
Screening
Journal
Orphanet journal of rare diseases
ISSN: 1750-1172
Titre abrégé: Orphanet J Rare Dis
Pays: England
ID NLM: 101266602
Informations de publication
Date de publication:
20 08 2020
20 08 2020
Historique:
received:
28
04
2020
accepted:
07
08
2020
entrez:
22
8
2020
pubmed:
21
8
2020
medline:
22
6
2021
Statut:
epublish
Résumé
Fabry disease (FD) is a systemic X-linked lysosomal disorder. A 'peripheral nerve variant' of FD has been hypothesized in subjects with neuropathy, without the early manifestations of the classic phenotype. A cohort of undiagnosed neuropathy patients with chronic polyneuropathy of undetermined aetiology and demyelinating neuropathy, unresponsive to immunomodulating treatment, were screened for FD. A total of 103 patients (64% males), were enrolled. No typical pathogenetic mutations for FD were identified. We are aware that the study sample was very small, but only a large, unfeasible theoretical sample size could demonstrate a statistically significant increased prevalence of FD in neuropathy patients, as peripheral neuropathy of undetermined cause is uncommon and there is a low prevalence of FD in the general population. Therefore, we are of the opinion that including tailored FD screening in the neuropathy diagnostic work-up, particularly when there are additional clinical characteristics, should be considered.
Identifiants
pubmed: 32819406
doi: 10.1186/s13023-020-01501-w
pii: 10.1186/s13023-020-01501-w
pmc: PMC7439676
doi:
Types de publication
Letter
Langues
eng
Sous-ensembles de citation
IM
Pagination
216Références
Int J Mol Sci. 2018 Nov 23;19(12):
pubmed: 30477121
Kidney Int. 2003 Sep;64(3):801-7
pubmed: 12911529
PLoS One. 2016 Feb 11;11(2):e0148316
pubmed: 26866599
J Inherit Metab Dis. 2018 Mar;41(2):209-219
pubmed: 29143201
N Engl J Med. 1991 Feb 7;324(6):395-9
pubmed: 1846223
Neurol India. 2014 Mar-Apr;62(2):219-21
pubmed: 24823749
Eur J Neurol. 2011 Apr;18(4):631-6
pubmed: 20860754
J Med Genet. 2018 Apr;55(4):261-268
pubmed: 29330335
J Neurol Neurosurg Psychiatry. 1984 May;47(5):530-5
pubmed: 6330306
Muscle Nerve. 2019 Mar;59(3):354-357
pubmed: 30246259
J Peripher Nerv Syst. 2010 Mar;15(1):1-9
pubmed: 20433600