Evidence map›Paper›PMID 33317601›Full record

ArticleOrphanet journal of rare diseases2020

Recommendations for pre-symptomatic genetic testing for hereditary transthyretin amyloidosis in the era of effective therapy: a multicenter Italian consensus.

M Grandis, L Obici, M Luigetti, C Briani, F Benedicenti, G Bisogni, M Canepa, F Cappelli, C Danesino, G M Fabrizi and 14 more

Abstract readMulticenter StudyConsensus Statement
In one paragraph

Article in Orphanet journal of rare diseases, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 28 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
28citing papers in PubMed, 1 pooled it
–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

28 citing papers in PubMed, 1 synthesis or guideline pooled it.

  1. Guideline
  2. Pre-symptomatic scintigraphic and genetic cascade screening in cardiac transthyretin amyloidosis.European journal of nuclear medicine and molecular imaging · 2025
    Trial
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  12. Review
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  19. Review
  20. Hereditary transthyretin amyloid cardiomyopathy.Singapore medical journal · 2023
    Article
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

24 authors.

M GrandisDepartment of Neuroscience, Rehabilitation, Ophthalmology, Genetics and Maternal and Child Health (DINOGMI), Section of Medical Genetics, University of Genoa, c/o DIMI Viale Benedetto XV, 6, 16132, Genova, Italy.
L ObiciAmyloidosis Research and Treatment Center, IRCCS Fondazione Policlinico San Matteo, Pavia, Italy.
M LuigettiUOC Neurologia, Fondazione Policlinico Universitario A. Gemelli IRCCS, Rome, Italy.
C BrianiDepartment of Neuroscience, University of Padova, Padova, Italy.
F BenedicentiMedical Genetics, Azienda Sanitaria Dell'Alto Adige, Bolzano, Italy.
G BisogniCentro Clinico Nemo Adulti-Fondazione Policlinico Universitario Agostino Gemelli IRCCS, Rome, Italy.
M CanepaCardiovascular Disease Unit, IRCCS Policlinico San Martino, Genova, and IRCCS Italian Cardiovascular Network, Department of Internal Medicine, University of Genova, Genova, Italy.
F CappelliTuscan Regional Amyloidosis Center, Careggi University Hospital, Firenze, Italy.
C DanesinoMolecular Medicine Department, University of Pavia, Pavia, Italy.
G M FabriziDepartment of Neurosciences, Biomedicine and Movement Sciences, Section of Neurology, University of Verona and University Hospital GB Rossi, Verona, Italy.
S FenuUnit of Rare Neurodegenerative and Neurometabolic Diseases, Department of Clinical Neurosciences, Fondazione IRCCS Istituto Neurologico Carlo Besta, Milan, Italy.
G FerrandesIRCCS Policlinico San Martino, Genova, Italy.
C GemelliDepartment of Neuroscience, Rehabilitation, Ophthalmology, Genetics and Maternal and Child Health (DINOGMI), Section of Medical Genetics, University of Genoa, c/o DIMI Viale Benedetto XV, 6, 16132, Genova, Italy.
F ManganelliDepartment of Neuroscience, Reproductive and Odontostomatological Sciences, University of Naples "Federico II", Napoli, Italy.
A MazzeoUnit of Neurology and Neuromuscular Diseases, Department of Clinical and Experimental Medicine, University of Messina, Messina, Italy.
L MelchiorriMedical Genetics Unit, Azienda Ospedaliero Universitaria Di Ferrara, Ferrara, Italy.
F PerfettoTuscan Regional Amyloidosis Center, Careggi University Hospital, Firenze, Italy.
L G PradottoDepartment of Neurosciences, University of Turin, Torino, Italy.
P RimessiMedical Genetics Unit, Azienda Ospedaliero Universitaria Di Ferrara, Ferrara, Italy.
G TiniCardiovascular Disease Unit, IRCCS Policlinico San Martino, Genova, and IRCCS Italian Cardiovascular Network, Department of Internal Medicine, University of Genova, Genova, Italy.
S TozzaDepartment of Neuroscience, Reproductive and Odontostomatological Sciences, University of Naples "Federico II", Napoli, Italy.
L TrevisanDepartment of Neuroscience, Rehabilitation, Ophthalmology, Genetics and Maternal and Child Health (DINOGMI), Section of Medical Genetics, University of Genoa, c/o DIMI Viale Benedetto XV, 6, 16132, Genova, Italy.
D PareysonUnit of Rare Neurodegenerative and Neurometabolic Diseases, Department of Clinical Neurosciences, Fondazione IRCCS Istituto Neurologico Carlo Besta, Milan, Italy.
P MandichDepartment of Neuroscience, Rehabilitation, Ophthalmology, Genetics and Maternal and Child Health (DINOGMI), Section of Medical Genetics, University of Genoa, c/o DIMI Viale Benedetto XV, 6, 16132, Genova, Italy. pmandich@unige.it.ORCID 0000-0003-3123-3512

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Hereditary transthyretin amyloidosis (ATTRv, v for variant) is a late-onset, autosomal dominant disease caused by progressive extracellular deposition of transthyretin amyloid fibrils, leading to organ damage and death. For other late-onset fatal diseases, as Huntington's disease, protocols for pre-symptomatic genetic testing (PST) are available since decades. For ATTRv, limited experience has been reported to date, mostly gathered before the availability of approved therapies. We aimed at developing recommendations for a safe and feasible PST protocol in ATTRv in the era of emerging treatments, taking also into account Italian patients' characteristics and healthcare system rules. After an initial survey on ongoing approaches to PST for ATTRv in Italy, two roundtable meetings were attended by 24 experts from 16 Italian centers involved in the diagnosis and care of this disease. Minimal requirements for PST offer and potential critical issues were highlighted. By November 2019, 457 families affected by ATTRv with 209 molecularly confirmed pre-symptomatic carriers were counted. The median age at PST was 41.3 years of age, regardless of the specific mutation. Half of the Italian centers had a multidisciplinary team, including a neurologist, an internist, a cardiologist, a medical geneticist and a psychologist, although in most cases not all the specialists were available in the same center. A variable number of visits was performed at each site. Experts agreed that PST should be offered only in the context of genetic counselling to at risk individuals aged 18 or older. Advertised commercial options for DNA testing should be avoided. The protocol should consist of several steps, including a preliminary clinical examination, a pre-test information session, an interval time, the genetic test and a post-test session with the disclosure of the test results, in the context of an experienced multidisciplinary team. Recommendations for best timing were also defined. Protocols for PST in the context of ATTRv can be refined to offer at risk individuals the best chance for early diagnosis and timely treatment start, while respecting autonomous decisions and promoting safe psychological adjustment to the genetic result.

Indexed as

Amyloid Neuropathies, FamilialGenetic TestingHumansItalyAttrvHereditary transthyretin amyloidosisPre-symptomatic genetic testing; PST

Identifiers

PMID33317601
PMCPMC7734774

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