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DOI 10.1186/s13023-020-01501-w

Медицинская библиотека · Orphanet journal of rare diseases · 2020

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

Rota Eugenia, Grandis Marina, Di Sapio Alessia, Ghiglione Elisabetta, Fiorentino Pietro, Repetto Alessandra, Giliberto Claudia, Gemelli Chiara, Morelli Nicola, Schenone Angelo, Cocito Dario

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Аннотация

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.

Для цитирования: Rota Eugenia, Grandis Marina, Di Sapio Alessia, Ghiglione Elisabetta, Fiorentino Pietro, Repetto Alessandra, Giliberto Claudia, Gemelli Chiara, Morelli Nicola, Schenone Angelo, Cocito Dario. Screening for Fabry disease in unknown origin axonal polyneuropathy: to do or not to do, this is the question!. Orphanet journal of rare diseases. 2020. DOI: 10.1186/s13023-020-01501-w.

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