Screening for Fabry disease in unknown origin axonal polyneuropathy: to do or not to do, this is the question!


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
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

216

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Auteurs

Eugenia Rota (E)

Neurology Unit, ASL Alessandria (AL), Via E. Raggio 12, 15067, Novi Ligure, AL, Italy. eugenia.rota.md@gmail.com.

Marina Grandis (M)

Department of Neuroscience, Rehabilitation, Ophthalmology, Genetics and Maternal Infantile Sciences, University of Genoa, Genoa, Italy.
IRCCS Policlinico San Martino Hospital, Genoa, Italy.

Alessia Di Sapio (A)

Neurology Unit, Regina Montis Regalis Hospital, ASL CN1, Mondovì, CN, Italy.

Elisabetta Ghiglione (E)

Neurology Unit, ASL Alessandria (AL), Via E. Raggio 12, 15067, Novi Ligure, AL, Italy.

Pietro Fiorentino (P)

Neurology Unit, ASL Alessandria (AL), Via E. Raggio 12, 15067, Novi Ligure, AL, Italy.

Alessandra Repetto (A)

Neurology Unit, ASL Alessandria (AL), Via E. Raggio 12, 15067, Novi Ligure, AL, Italy.

Claudia Giliberto (C)

Neurology Unit, Regina Montis Regalis Hospital, ASL CN1, Mondovì, CN, Italy.

Chiara Gemelli (C)

Department of Neuroscience, Rehabilitation, Ophthalmology, Genetics and Maternal Infantile Sciences, University of Genoa, Genoa, Italy.

Nicola Morelli (N)

Neurology and Radiology Unit, Guglielmo da Saliceto Hospital, AUSLPC, Piacenza, PC, Italy.

Angelo Schenone (A)

Department of Neuroscience, Rehabilitation, Ophthalmology, Genetics and Maternal Infantile Sciences, University of Genoa, Genoa, Italy.
IRCCS Policlinico San Martino Hospital, Genoa, Italy.

Dario Cocito (D)

Istituti Clinici-Scientifici Maugeri, Torino, Italy.

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Classifications MeSH