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Record W2530762184 · doi:10.1111/ejh.12819

Novel G6B gene variant causes familial autosomal recessive thrombocytopenia and anemia

2016· article· en· W2530762184 on OpenAlex
Motasem Melhem, Mohamed Abu‐Farha, Dinu Antony, Ashraf Al Madhoun, Chiara Bacchelli, Fadi Alkayal, Irina Al‐Khairi, Sumi Elsa John, Mohamad Alomari, Phillip Beales, Osama Alsmadi

Why this work is in the frame

A frame that forgets how it found something cannot be audited. These are the routes that admitted this work.

affAt least one author lists a Canadian institution in the pinned OpenAlex snapshot.

Bibliographic record

VenueEuropean Journal Of Haematology · 2016
Typearticle
Languageen
FieldMedicine
TopicPlatelet Disorders and Treatments
Canadian institutionsWindsor Regional Hospital
FundersBiotechnology and Biological Sciences Research CouncilDasman Diabetes InstituteKuwait Foundation for the Advancement of SciencesNational Institute for Health and Care Research
KeywordsBiologyExome sequencingGeneticsHaploinsufficiencyGeneMutationPhenotype

Abstract

fetched live from OpenAlex

Abstract Objective To characterize the underlying genetic and molecular defects in a consanguineous family with lifelong blood disorder manifested with thrombocytopenia (low platelets count) and anemia. Methods Genetic linkage analysis, exome sequencing, and functional genomics were carried out to identify and characterize the defective gene. Results We identified a novel truncation mutation (p.C108*) in chromosome 6 open reading frame 25 ( C6orf25 ) gene in this family. We also showed the p.C108* mutation was responsible for destabilizing the encoded truncated G6B protein. Unlike the truncated form, wild‐type G6B expression resulted in enhanced K562 differentiation into megakaryocytes and erythrocytes. C6orf25, also known as G6B , is an effector protein for the key hematopoiesis regulators, Src homology region 2 domain‐containing phosphatases SHP ‐1 and SHP ‐2. Conclusion G6B seems to act through an autosomal recessive mode of disease transmission in this family and regarded as the gene responsible for the observed hematological disorder. This inference is well supported further by in vivo evidence where similar outcomes were reported from G6b −/− and SHP 1/2 DKO mouse models.

Fetched live from OpenAlex and de-inverted. Abstracts are not stored in this database: the inverted indexes are 8.6 GB of the frame’s 9.3 GB of text, and the host has 13 GB free.

Full frame distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.000
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Case report · Consensus signal: Case report
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.338
Threshold uncertainty score0.363

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0010.000
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0000.000

Machine scores (provisional)

The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.

Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.

Opus teacher head0.022
GPT teacher head0.263
Teacher spread0.241 · how far apart the two teachers sit on this one work
Validation statusscore_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from it