Evidence map›Paper›PMID 33596643›Full record

SynthesisHaematologica2022

Identification of a novel genetic locus associated with immune-mediated thrombotic thrombocytopenic purpura.

Matthew J Stubbs, Paul Coppo, Chris Cheshire, Agnès Veyradier, Stephanie Dufek, Adam P Levine, Mari Thomas, Vaksha Patel, John O Connolly, Michael Hubank and 7 more

Open access · goldAbstract readMeta-Analysis
In one paragraph

Synthesis in Haematologica, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
0.2field-weighted citation impact, top 56% 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

2 citing papers in PubMed, 4 citations in OpenAlex.

  1. Article
  2. Review
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

17 authors at 6 institutions in 2 countries.

Matthew J StubbsHaemostasis Research Unit, UCL (London, UK); Department of Renal Medicine. m.stubbs@doctors.org.uk.
Paul CoppoCentre de Référence des Microangiopathies Thrombotiques, Hôpital Saint-Antoine (Paris, France).
Chris CheshireDepartment of Renal Medicine.
Agnès VeyradierDepartment d'Hematologie, Centre de Référence des Microangiopathies Thrombotiques, Hôpital Lariboisière (Paris, France).
Stephanie DufekDepartment of Renal Medicine.
Adam P LevineDepartment of Renal Medicine.
Mari ThomasHaemostasis Research Unit, UCL (London, UK); National Institute for Health Research Cardiometabolic Programme, UCLH/UCL Cardiovascular BRC (London, UK).
Vaksha PatelDepartment of Renal Medicine.
John O ConnollyDepartment of Renal Medicine.
Michael HubankClinical Genomics, Royal Marsden Hospital (London, UK).
Ygal BenhamouCentre de Référence des Microangiopathies Thrombotiques, Hôpital Saint-Antoine (Paris, France).
Lionel GalicierCentre de Référence des Microangiopathies Thrombotiques, Hôpital Saint-Antoine (Paris, France).
Pascale PoullinCentre de Référence des Microangiopathies Thrombotiques, Hôpital Saint-Antoine (Paris, France).
Robert KletaDepartment of Renal Medicine.
Daniel P GaleDepartment of Renal Medicine.
Horia StanescuDepartment of Renal Medicine.
Marie A ScullyHaemostasis Research Unit, UCL (London, UK); National Institute for Health Research Cardiometabolic Programme, UCLH/UCL Cardiovascular BRC (London, UK).
Sorbonne Université · FRUniversity College London · GBHôpital Lariboisière · FRHôpital Saint-Antoine · FRNational Institute for Health Research · GBRoyal Marsden Hospital · GB

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Immune thrombotic thrombocytopenic purpura (iTTP) is an ultra-rare, life-threatening disorder, mediated through severe ADAMTS13 deficiency causing multi-system micro-thrombi formation, and has specific human leukocyte antigen associations. We undertook a large genome-wide association study to investigate additional genetically distinct associations in iTTP. We compared two iTTP patient cohorts with controls, following standardized genome-wide quality control procedures for single-nucleotide polymorphisms and imputed HLA types. Associations were functionally investigated using expression quantitative trait loci (eQTL), and motif binding prediction software. Independent associations consistent with previous findings in iTTP were detected at the HLA locus and in addition a novel association was detected on chromosome 3 (rs9884090, P=5.22x10-10, odds ratio 0.40) in the UK discovery cohort. Meta-analysis, including the French replication cohort, strengthened the associations. The haploblock containing rs9884090 is associated with reduced protein O-glycosyltransferase 1 (POGLUT1) expression (eQTL P<0.05), and functional annotation suggested a potential causative variant (rs71767581). This work implicates POGLUT1 in iTTP pathophysiology and suggests altered post-translational modification of its targets may influence disease susceptibility.

Indexed as

Purpura, Thrombocytopenic, IdiopathicPurpura, Thrombotic ThrombocytopenicADAMTS13 ProteinGenetic LociGenome-Wide Association StudyGlucosyltransferasesHumansADAMTS13 ProteinGlucosyltransferasesPOGLUT1 protein, human

Identifiers

PMID33596643
PMCPMC8883548
OpenAlexW3132963212

What OpenQuestion 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 OpenQuestion graph.