Evidence mapPaperPMID 39639351Full record

ArticleOrphanet journal of rare diseases2024

Global longitudinal strain in pre-symptomatic patients with mutation for transthyretin amyloidosis.

Grazia Canciello, Stefano Tozza, Gaetano Todde, Maria Nolano, Felice Borrelli, Giovanni Palumbo, Raffaella Lombardi, Emanuele Cassano, Wanda Acampa, Giovanni Esposito and 2 more

Abstract read
In one paragraph

Article in Orphanet journal of rare diseases, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

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2citing papers in PubMed
field-weighted citation impact
1 · What the graph read from it

What it found

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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

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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

2 citing papers in PubMed.

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4 · The record

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5 · Who and what money

Authors and funding

12 authors.

Grazia Canciello *Department of Advanced Biomedical Sciences, University Federico II of Naples, Naples, Italy.
Stefano Tozza *Department of Neuroscience and Reproductive and Odontostomatological Sciences, University Federico II of Naples, Via S Pansini 5, 80131, Naples, Italy.
Gaetano ToddeDepartment of Advanced Biomedical Sciences, University Federico II of Naples, Naples, Italy.
Maria NolanoDepartment of Neuroscience and Reproductive and Odontostomatological Sciences, University Federico II of Naples, Via S Pansini 5, 80131, Naples, Italy.
Felice BorrelliDepartment of Advanced Biomedical Sciences, University Federico II of Naples, Naples, Italy.
Giovanni PalumboDepartment of Neuroscience and Reproductive and Odontostomatological Sciences, University Federico II of Naples, Via S Pansini 5, 80131, Naples, Italy.
Raffaella LombardiDepartment of Advanced Biomedical Sciences, University Federico II of Naples, Naples, Italy.
Emanuele CassanoDepartment of Neuroscience and Reproductive and Odontostomatological Sciences, University Federico II of Naples, Via S Pansini 5, 80131, Naples, Italy.
Wanda AcampaDepartment of Advanced Biomedical Sciences, University Federico II of Naples, Naples, Italy.
Giovanni EspositoDepartment of Advanced Biomedical Sciences, University Federico II of Naples, Naples, Italy.
Fiore ManganelliDepartment of Neuroscience and Reproductive and Odontostomatological Sciences, University Federico II of Naples, Via S Pansini 5, 80131, Naples, Italy. fioremanganelli@gmail.com.ORCID 0000-0002-1442-9604
Maria Angela LosiDepartment of Advanced Biomedical Sciences, University Federico II of Naples, Naples, Italy.

Funding

Ministero dell'Istruzione, dell'Università e della Ricerca National RecoveryMinistero dell'Istruzione, dell'Università e della Ricerca Resilience Plan
6 · The paper itself

Abstract

backgroundHereditary transthyretin (ATTRv) amyloidosis is rare, autosomal dominant disease with a fatal outcome if left untreated. Early stages detection is crucial for intervention. We aimed identifying early indexes of cardiac involvement and their eventual correlation with neurological indexes, in pre-symptomatic subjects with TTR gene mutation.

methodsSixteen TTR-mutation carriers (mean age 51 ± 9 years, 6 males, 7 with Val30Met and 9 with Phe64Leu mutation) without left ventricular hypertrophy were studied. Predicted Age of Disease Onset (PADO) and time to PADO (Time-to PADO = PADO-age at evaluation) were computed. Subjects underwent: cardiological and echocardiographic assessment including global longitudinal strain (GLS); tactile and thermal quantitative sensory testing (QST); Perugini score by bone scintigraphy.

resultsTime to PADO was 30 ± 15 years. Nine subjects showed abnormal GLS (> -20%), unrelated to age, LVMi, MWT, E/e', NT-proBNP or Time-to PADO. QST findings were abnormal in most subjects. At a worse cold pain threshold corresponded a worse GLS (r = 0.786, p < 0.001). Perugini score was positive in 1 subject.

conclusionsGLS and QST findings support an early involvement of heart and small nerve fibers even many years before PADO. Interestingly, cardiac impairment seems to parallel that of small, nerve fibers, at least in the earliest stage of disease.

Indexed as

Amyloid Neuropathies, FamilialMutationPrealbuminAdultAgedEchocardiographyFemaleGlobal Longitudinal StrainHumansMaleMiddle AgedPrealbuminGlobal longitudinal strainHereditary transthyretin amyloidosisPre-clinical diseaseThermal quantitative sensory testing

Identifiers

PMID39639351
PMCPMC11619093

What Socratic holds

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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.