Evidence mapPaperPMID 40670759Full record

ArticleJournal of neurology2025

Prevalence of hereditary transthyretin amyloidosis in CIDP patients with red flags: a multicenter genetic screening and misdiagnosis analysis.

Pietro Emiliano Doneddu, Giulia Moretti, Vincenzo Di Stefano, Yuri Falzone, Luca Leonardi, Marco Luigetti, Giorgia Mataluni, Luca Gentile, Marinella Carpo, Alessandro Barilaro and 19 more

Abstract readMulticenter Study
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In one paragraph

Article in Journal of neurology, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.

0numbers the graph read from it
0cells of the map it votes in
3citing papers in PubMed
field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

3 citing papers in PubMed.

  1. The role of "red flags" in the diagnostic work-up of hereditary transthyretin amyloidosis: a study using a machine-learning approach.Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology · 2026
    Article
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4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

29 authors.

Pietro Emiliano Doneddu *Neuromuscular and Neuroimmunology Unit, Neuromuscular and Neuroimmunology Service, IRCCS Humanitas Research Hospital, Via Manzoni 56, 20089, Milan, Rozzano, Italy.
Giulia Moretti *Neuromuscular and Neuroimmunology Unit, Neuromuscular and Neuroimmunology Service, IRCCS Humanitas Research Hospital, Via Manzoni 56, 20089, Milan, Rozzano, Italy.
Vincenzo Di StefanoRegional Center for Diagnosis and Treatment of Neuromuscular Disease, Department of Biomedicine, Neuroscience and Advanced Diagnostic (BIND), University of Palermo, Palermo, Italy.
Yuri FalzoneNeurology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy.
Luca LeonardiNeuromuscular and Rare Disease Centre, Neurology Unit, Sant'Andrea Hospital, 00189, Rome, Italy.
Marco LuigettiDipartimento Di Neuroscienze, Università Cattolica del Sacro Cuore, 00168, Rome, Italy.
Giorgia MataluniArea Neuropatie Infiammatorie Immuno-mediate del Centro di riferimento per le Malattie Neurologiche, Neuromuscolari E Distrofie Miotoniche Rare, Policlinico Tor Vergata, Rome, Italy.
Luca GentileDepartment of Clinical and Experimental Medicine, Unit of Neurology, University of Messina, Messina, Italy.
Marinella CarpoASST Bergamo Ovest-Ospedale Treviglio, Treviglio, Italy.
Alessandro BarilaroAOU Careggi and Department of Neurosciences, Drug and Child Health, University of Florence, Florence, Italy.
Massimiliano FilostoNeMO-Brescia Clinical Center for Neuromuscular Diseases, Brescia, Italy.
Elisa VegezziNeurooncology and Neuroinflammation Unit, IRCCS Mondino Foundation, Pavia, Italy.
Maurizio InghilleriDepartment of Human Neurosciences, Sapienza University of Rome, Rome, Italy.
Fabrizio CanaleDivision of Neurology and Neurophysiopathology, Department of Medical and Surgical Sciences, University of Campania "Luigi Vanvitelli", Naples, Italy.
Filippo BrighinaRegional Center for Diagnosis and Treatment of Neuromuscular Disease, Department of Biomedicine, Neuroscience and Advanced Diagnostic (BIND), University of Palermo, Palermo, Italy.
Sabrina MatàDipartimento Neuromuscoloscheletrico E Degli Organi Di Senso, Neurology Unit, Careggi University Hospital, Florence, Italy.
Adele RattiNeurology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy.
Francesca ForcinaNeuromuscular and Rare Disease Centre, Neurology Unit, Sant'Andrea Hospital, 00189, Rome, Italy.
Giovanni SiconolfiDipartimento Di Neuroscienze, Università Cattolica del Sacro Cuore, 00168, Rome, Italy.
Claudia LoziArea Neuropatie Infiammatorie Immuno-mediate del Centro di riferimento per le Malattie Neurologiche, Neuromuscolari E Distrofie Miotoniche Rare, Policlinico Tor Vergata, Rome, Italy.
Anna MazzeoDepartment of Clinical and Experimental Medicine, Unit of Neurology, University of Messina, Messina, Italy.
Ugo MolloAOU Careggi and Department of Neurosciences, Drug and Child Health, University of Florence, Florence, Italy.
Barbara RisiNeMO-Brescia Clinical Center for Neuromuscular Diseases, Brescia, Italy.
Giuseppe CosentinoDepartment of Brain and Behavioral Sciences, University of Pavia, Via Bassi 21, 27100, Pavia, Italy.
Federica MoretDepartment of Human Neurosciences, Sapienza University of Rome, Rome, Italy.
Carla FasanoAOU Careggi and Department of Neurosciences, Drug and Child Health, University of Florence, Florence, Italy.
Vincenzo TodiscoDivision of Neurology and Neurophysiopathology, Department of Medical and Surgical Sciences, University of Campania "Luigi Vanvitelli", Naples, Italy.
Massimo RussoDepartment of Clinical and Experimental Medicine, University of Messina, Policlinico "G. Martino", Via Consolare Valeria N°1, Messina, Italy.
Eduardo Nobile-OrazioNeuromuscular and Neuroimmunology Unit, Neuromuscular and Neuroimmunology Service, IRCCS Humanitas Research Hospital, Via Manzoni 56, 20089, Milan, Rozzano, Italy. eduardo.nobile@unimi.it.ORCID http://orcid.org/0000-0003-2624-8138

Funding

Ministero della Salute Ricerca Finalizzata (Progetto RF-2016-02361887)
6 · The paper itself

Abstract

backgroundHereditary transthyretin amyloidosis (ATTRv) is a rare, multisystemic disorder often presenting with peripheral neuropathy and can be misdiagnosed with chronic inflammatory demyelinating polyradiculoneuropathy (CIDP), especially in non-endemic areas. While clinical red flags have been proposed to aid diagnosis, their predictive value remains uncertain. This study prospectively assessed the prevalence of TTR variants in CIDP patients with red flags for ATTRv and retrospectively analyzed features of genetically confirmed ATTRv cases initially misdiagnosed as CIDP.

methodsThirteen Italian tertiary neuromuscular centers consecutively screened CIDP patients with at least one red flag for TTR gene variants. A retrospective analysis was also conducted on ATTRv patients initially misdiagnosed as CIDP, comparing clinical, electrophysiological, and treatment response features to confirmed CIDP cases.

resultsNo TTR variants were identified among 124 screened CIDP patients despite 65% presenting with ≥ 2 red flags and 14% not responding to standard therapies. Among 17 retrospectively identified ATTRv patients, 5 (29%) met electrodiagnostic criteria for CIDP. In nearly half, CIDP was diagnosed without fulfilling electrodiagnostic criteria or obtaining appropriate supportive investigations. Compared to confirmed CIDP patients, ATTRv cases exhibited significantly more red flags, later onset, more insidious and distal presentations, a progressive course, lower rates of demyelination criteria fulfillment, and no response to immunomodulatory therapy.

conclusionsRed flags alone have limited predictive value in specialized settings. However, ATTRv should be considered in distal, progressive, treatment-resistant neuropathies, especially with multisystem features. Greater diagnostic rigor and increased awareness in non-specialist settings is essential to reduce misdiagnosis and improve access to therapy.

Indexed as

Amyloid Neuropathies, FamilialDiagnostic ErrorsPolyradiculoneuropathy, Chronic Inflammatory DemyelinatingAdultAgedFemaleGenetic TestingHumansItalyMaleMiddle AgedPrealbuminPrevalenceRetrospective StudiesPrealbuminTTR protein, humanAmyloidosisATTRChronic inflammatory demyelinating polyradiculoneuropathyCIDPDiagnosisMisdiagnosis

Identifiers

What Socratic holds

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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the Socratic graph.