Evidence map›Paper›PMID 19484238›Full record

ArticleAnnals of hematology2010

The same genetic defect in three Tunisian families with Bernard Soulier syndrome: a probable founder Stop mutation in GPIbβ.

Basma HadjKacem, Henda Elleuch, Ramzi Trigui, Jalel Gargouri, Ali Faouzi Gargouri

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In one paragraph

Article in Annals of hematology, 2010. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
0.3field-weighted citation impact, top 41% 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

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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

1 citing paper in PubMed, 9 citations in OpenAlex.

  1. 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

5 authors at 2 institutions in 2 countries.

Basma HadjKacemLaboratoire de Génétique Moléculaire des Eucaryotes, Centre de Biotechnologie de Sfax, Tunisia.
Henda Elleuch
Ramzi Trigui
Jalel Gargouri
Ali Faouzi Gargouri
Centre of Biotechnology of Sfax · TNService Régional Vaudois de Transfusion Sanguine · CH

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

GPIbα, GPIbβ, and GPIX are three candidate genes for a rare genetic bleeding disorder named Bernard Soulier syndrome (BSS). These genes are unique in the genome and encode for glycoprotein subunits of the GPIb-IX complex. Quantitative or qualitative deficiency in this complex is often associated with BSS. Here, we report the novel variant of BSS in which Ser23 of GPIbβ is substituted by a Stop codon causing a premature termination of translation, recently described in one family. This genetic defect is revealed in three unrelated BSS patients. The pedigree was determined for two families (F1 and F2) and revealed the homozygosity of the mutation in the two patients and its heterozygosity in parents. In the third family, the patient DNA was heterozygote with the same Ser23 Stop mutation in addition to two missense heterozygote mutations (Asp 51 Gly) and (Ala 55 to Pro). We studied the effect of the Ser23 Stop mutation on the expression of the complex. Our findings confirm that the identified GPIbβ mutation is responsible for the BSS phenotype and hampers the GPIb-IX complex to form on the platelets' surface. Regarding the scarcity of the BSS syndrome, the occurrence of the same mutation in three unrelated families would suggest a BSS founder mutation in Tunisia.

Indexed as

AdultBernard-Soulier SyndromeChildCodon, NonsenseCodon, TerminatorFemaleGenetic VariationHumansMalePedigreePeptide Chain Termination, TranslationalPlatelet Glycoprotein GPIb-IX ComplexSerineTunisiaCodon, NonsenseCodon, TerminatorPlatelet Glycoprotein GPIb-IX ComplexSerine

Identifiers

PMID19484238
OpenAlexW1995962740

What OpenQuestion 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 OpenQuestion graph.