GP1BB

Chr 22AR

glycoprotein Ib platelet subunit beta

Also known as: BDPLT1, BS, CD42C, GP-Ib beta, GPIBB, GPIbbeta

Platelet glycoprotein Ib (GPIb) is a heterodimeric transmembrane protein consisting of a disulfide-linked 140 kD alpha chain and 22 kD beta chain. It is part of the GPIb-V-IX system that constitutes the receptor for von Willebrand factor (VWF), and mediates platelet adhesion in the arterial circulation. GPIb alpha chain provides the VWF binding site, and GPIb beta contributes to surface expression of the receptor and participates in transmembrane signaling through phosphorylation of its intracellular domain. Mutations in the GPIb beta subunit have been associated with Bernard-Soulier syndrome, velocardiofacial syndrome and giant platelet disorder. The 206 amino acid precursor of GPIb beta is synthesized from a 1.0 kb mRNA expressed in plateletes and megakaryocytes. A 411 amino acid protein arising from a longer, unspliced transcript in endothelial cells has been described; however, the authenticity of this product has been questioned. Yet another less abundant GPIb beta mRNA species of 3.5 kb, expressed in nonhematopoietic tissues such as endothelium, brain and heart, was shown to result from inefficient usage of a non-consensus polyA signal in the neighboring upstream gene (SEPT5, septin 5). In the absence of polyadenylation from its own imperfect site, the SEPT5 gene produces read-through transcripts that use the consensus polyA signal of this gene. [provided by RefSeq, Dec 2010]

OMIMResearchGenerating clinical summary…
MultiplemechanismARLOEUF 1.332 OMIM phenotypes
Clinical SummaryGP1BB
🧬
Gene-Disease Validity (ClinGen)
Bernard-Soulier syndrome · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Moderately constrained gene (pLI 0.51) — some intolerance to loss-of-function variants.
📋
ClinVar Variants
47 unique Pathogenic / Likely Pathogenic· 84 VUS of 162 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint?
1.33LOEUF
pLI 0.512
Z-score 1.33
OE 0.00 (0.001.33)
Moderately constrained

Highly tolerant — LoF variants common in population

Missense Constraint?
0.46Z-score
OE missense 0.84 (0.671.05)
53 obs / 63.3 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios?
LoF OE?0.00 (0.001.33)
00.351.4
Missense OE?0.84 (0.671.05)
00.61.4
Synonymous OE?1.14
01.21.6
LoF obs/exp: 0 / 2.0Missense obs/exp: 53 / 63.3Syn Z: -0.64

This gene — mechanism propensity

DN
0.6552th %ile
GOF
0.80top 10%
LOF
0.3648th %ile

This gene has evidence for multiple mechanisms of pathogenicity (gain-of-function and dominant-negative). Both the Badonyi & Marsh prediction and the broader genomic evidence point to gain-of-function as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

GOFprediction above median
DNprediction above median

Note: In-silico variant effect predictors (SIFT, PolyPhen, REVEL, CADD) may underestimate pathogenicity of missense variants in genes with GOF or DN mechanisms. Consider functional evidence and clinical context.

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312.

ClinVar Variant Classifications

162 submitted variants in ClinVar

Classification Summary

Pathogenic11
Likely Pathogenic36
VUS84
Likely Benign23
Benign4
Conflicting4
11
Pathogenic
36
Likely Pathogenic
84
VUS
23
Likely Benign
4
Benign
4
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
7
2
2
0
11
Likely Pathogenic
14
22
0
0
36
VUS
0
77
7
0
84
Likely Benign
0
0
2
21
23
Benign
0
3
1
0
4
Conflicting
4
Total211041221162

LoF = frameshift, stop gained/lost, canonical splice · Counts from ClinVar esearch · Updated hourly

View in ClinVar →

396 pathogenic / likely-pathogenic (of 414) ClinVar copy-number / structural variants overlap GP1BB — these span large chromosomal regions, not the gene specifically, and are excluded from the counts above. Explore in CNV tools →

Protein Context — Lollipop Plot

GP1BB · protein map & ClinVar variants

Showing all ClinVar variants across the protein. Search a specific variant to highlight its position.

Clinical Trials

Active and recruiting trials from ClinicalTrials.gov

No active trials found for this gene.

Search ClinicalTrials.gov →