Evidence mapPaperPMID 35256455Full record

ReviewJournal of neurology, neurosurgery, and psychiatry2022

Novel approaches to diagnosis and management of hereditary transthyretin amyloidosis.

Antonia Carroll, P James Dyck, Mamede de Carvalho, Marina Kennerson, Mary M Reilly, Matthew C Kiernan, Steve Vucic

Open access · hybridAbstract readReview
In one paragraph

Review in Journal of neurology, neurosurgery, and psychiatry, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 53 papers, 3 of them syntheses that pooled it.

0numbers the graph read from it
0cells of the map it votes in
53citing papers in PubMed, 3 pooled it
8.5field-weighted citation impact, top 1% of its field
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

53 citing papers in PubMed, 3 syntheses or guidelines pooled it, 107 citations in OpenAlex.

  1. Pooled it
  2. Pooled it
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  4. Article
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  10. Skin Biopsy as a Diagnostic Tool for ATTRv Amyloid Neuropathy in the UK.Journal of the peripheral nervous system : JPNS · 2025
    Article
  11. Article
  12. Review
  13. NfL as a biomarker in ATTRv amyloidosis: potential and limitations.Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology · 2025
    Article
  14. Article
  15. Review
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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

7 authors at 4 institutions in 4 countries.

Antonia CarrollBrain and Mind Centre, Faculty of Medicine and Health, Translational Research Collective, University of Sydney and Department of Neurology, Royal Prince Alfred Hospital, Sydney, New South Wales, Australia.ORCID 0000-0002-1738-9580
P James DyckNeurology, Mayo Clinic, Rochester, Minnesota, USA.ORCID 0000-0003-0212-8254
Mamede de CarvalhoInstituto de Medicina Molecular, Faculdade de Medicina, Universidade de Lisboa, Lisboa, Portugal.
Marina KennersonNorthcott Neuroscience Laboratory, ANZAC Research Institute, Molecular Medicine Laboratory Concord Repatriation General Hospital, and Concord Clinical School, The University of Sydney, Sydney, New South Wales, Australia.
Mary M ReillyMRC Centre for Neuromuscular Diseases, UCL Institute of Neurology and National Hospital for Neurology and Neurosurgery, London, UK.
Matthew C KiernanBushell Chair of Neurology, Brain and Mind Centre, University of Sydney, Sydney, New South Wales, Australia.ORCID 0000-0001-9054-026X
Steve VucicBrain and Nerve Research Center, Concord Clinical School, The University of Sydney, Sydney, New South Wales, Australia steve.vucic@sydney.edu.au.ORCID 0000-0002-8323-873X
The University of Sydney · AUMayo Clinic · USNational Hospital for Neurology and Neurosurgery · GBUniversity of Lisbon · PT

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Hereditary transthyretin amyloidosis (ATTRv) is a severe, adult-onset autosomal dominant inherited systemic disease predominantly affecting the peripheral and autonomic nervous system, heart, kidney and the eyes. ATTRv is caused by mutations of the transthyretin (TTR) gene, leading to extracellular deposition of amyloid fibrils in multiple organs including the peripheral nervous system. Typically, the neuropathy associated with ATTRv is characterised by a rapidly progressive and disabling sensorimotor axonal neuropathy with early small-fibre involvement. Carpal tunnel syndrome and cardiac dysfunction frequently coexist as part of the ATTRv phenotype. Although awareness of ATTRv polyneuropathy among neurologists has increased, the rate of misdiagnosis remains high, resulting in significant diagnostic delays and accrued disability. A timely and definitive diagnosis is important, given the emergence of effective therapies which have revolutionised the management of transthyretin amyloidosis. TTR protein stabilisers diflunisal and tafamidis can delay the progression of the disease, if treated early in the course. Additionally, TTR gene silencing medications, patisiran and inotersen, have resulted in up to 80% reduction in TTR production, leading to stabilisation or slight improvement of peripheral neuropathy and cardiac dysfunction, as well as improvement in quality of life and functional outcomes. The considerable therapeutic advances have raised additional challenges, including optimisation of diagnostic techniques and management approaches in ATTRv neuropathy. This review highlights the key advances in the diagnostic techniques, current and emerging management strategies, and biomarker development for disease progression in ATTRv.

Indexed as

Amyloid Neuropathies, FamilialHeart DiseasesPolyneuropathiesHumansPrealbuminQuality of LifePrealbuminamyloidneuropathy

Identifiers

PMID35256455
PMCPMC9148983
OpenAlexW4221031684

What Socratic holds

Textmetadata
LicenceCC BY-NC
Read underepoch 390

Registered trials

None linked

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.