A novel variant Glanzmann thrombasthenia due to co-inheritance of a loss- and a gain-of-function mutation of ITGB3: evidence of a dominant effect of gain-of-function mutations
PMID: 29439184
Gene: ITGB3
HGNC: 6156
A novel variant Glanzmann thrombasthenia due to co-inheritance of a loss- and a gain-of-function mutation of ITGB3: evidence of a dominant effect of gain-of-function mutations
PMID: 29439184
Gene: ITGB3
HGNC: 6156
DDAVP infusion can modify VASP phosphorylation in humanplatelets
PMID: 15102025
Gene: ITGA2B and/or ITGB3
HGNC: 6138 and 6156
InsufficientEvidence: This article mentions "a platelet defect as a heterozygous Glanzmann thrombasthenia" however unable to access the article they cited after that sentence to get further information on the heterozygous variant
Supplementary Table S1 & S2 Proband and Variant Information:
Case44: GT43b, Male, 11 years old, Morrocan
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Affected sister is 43a (See Supplementary Table S1)
ParentalGenotype: N/A
CasePresentingHPOs: HP:0000421, HP:0000979, HP:0031364, HP:0000967, HP:0030137
(Epistaxis, purpura, ecchymosis, petechiae, prolonged bleeding at circumcision)
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry showed <5% expression of αIIbβ3 (See Supplementary Table 1)
PlateletAggregation: Absent platelet aggregation (See Supplementary Table 1)
CasePreviousTesting: Sanger sequencing of ITGA2B and ITGB3
Gene: ITGB3
Variant: NM_000212.2:c.614+1G>T and NM_000212.2:c.683G>A (p.Arg228His)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA400023344 and CA8623025
gnomAD: MAF in the African population is 0.00008012 http://gnomad.broadinstitute.org/variant/17-45363694-G-A
Supplementary Table S1 & S2 Proband and Variant Information:
Case56: GT57, Female, 10, Argentinian/European
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: HP:0000421, HP:0000967
(Epistaxis, petechia)
CaseHPOFreeText: Hematomas
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry showed <5% expression of αIIbβ3 (See Supplementary Table 1)
PlateletAggregation: Absent platelet aggregation (See Supplementary Table 1)
CasePreviousTesting: Sanger sequencing of ITGA2B and ITGB3
Gene: ITGA2B
Variant: NM_000419.3:c.571T>G (p.Phe191Val) and NM_000419.3:c.961G>T (p.Phe191Alafs*32)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA399805699 and CA399804768
gnomAD: N/A
Supplementary Table S1 & S2 Proband and Variant Information:
Case44: GT43b, Male, 11 years old, Morrocan
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Affected sister is 43a (See Supplementary Table S1)
ParentalGenotype: N/A
CasePresentingHPOs: HP:0000421, HP:0000979, HP:0031364, HP:0000967
(Epistaxis, purpura, ecchymosis, petechiae)
CaseHPOFreeText: Lots of bleeding at circumcision
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry showed <5% expression of αIIbβ3 (See Supplementary Table 1)
PlateletAggregation: Absent platelet aggregation (See Supplementary Table 1)
CasePreviousTesting: Sanger sequencing of ITGA2B and ITGB3
Gene: ITGB3
Variant: NM_000212.2:c.614+1G>T and NM_000212.2:c.683G>A (p.Arg228His)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA400023344 and CA8623025
gnomAD: MAF in the African population is 0.00008012 http://gnomad.broadinstitute.org/variant/17-45363694-G-A
Supplementary Table S1 & S2 Proband and Variant Information:
Case62: GT65, Female, 31 years old, French
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: HP:0000421, HP:0000225, HP:0000132, HP:0000790, HP:0002239, HP:0006298
(Epistaxis, gingival bleeding, menorrhagia, hematuria, GI bleeding, bleeding after dental procedure)
CaseHPOFreeText: Tonsil bleeding, muscle hematomas
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry showed <5% expression of αIIbβ3 (See Supplementary Table 1)
PlateletAggregation: Absent platelet aggregation (See Supplementary Table 1)
CasePreviousTesting: Sanger sequencing of ITGA2B and ITGB3
Gene: ITGA2B
Variant: NM_000419.3:c.574+1G>A and NM_000419.3:c.1946+3G>T
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA399805691 and CA626121071
gnomAD: MAF in the European (non-Finnish) population is 0.00001235 https://gnomad.broadinstitute.org/variant/17-42462918-C-T and MAF in the African population is 0.000 https://gnomad.broadinstitute.org/variant/17-42456008-C-A
Supplementary Table S1 & S2 Proband and Variant Information:
Case13: GT14, Female, 10, French
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: HP:0000979 and HP:0006298
(Purpura, prolonged bleeding after dental extraction)
CaseHPOFreeText: Moderate cutaneous bleeding from birth
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry showed <5% variant expression of αIIbβ3 (See Supplementary Table 1)
PlateletAggregation: Absent platelet aggregation (See Supplementary Table 1)
CasePreviousTesting: Sanger sequencing of ITGA2B and ITGB3
Gene: ITGA2B
Variant: NM_000419.3:c.423G>T (p.Trp141Cys) and NM_000419.3:c.2902del (p.Tyr968Metfs*163)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA399806022 and CA915940332
gnomAD: N/A
Supplementary Table S1 & S2 Proband and Variant Information:
Case51: GT51, Male, 29 years old, French/African
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: HP:0000421 (Epistaxis)
CaseHPOFreeText: Mucocutaneous bleeding
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: N/A
PlateletAggregation: Absent platelet aggregation (See Supplementary Table 1)
CasePreviousTesting: Sanger sequencing of ITGA2B and ITGB3
Gene: ITGB3
Variant: NM_000212.2:c.861del (p.Arg287Serfs * 34) and NM_000212.2:c.1456del (p.Cys486Alafs * 183)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA915940375 and CA915940376
gnomAD: N/A
Supplementary Table S1 & S2 Proband and Variant Information:
Case58: GT60, Male, 54 years old, French/Italian
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: HP:0000421, HP:0000978, HP:0002239, HP:0000225
(Epistaxis, easy bruising, GI bleeding, gingival bleeding)
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry showed <5% expression of αIIbβ3 (See Supplementary Table 1)
PlateletAggregation: Absent platelet aggregation (See Supplementary Table 1)
CasePreviousTesting: Sanger sequencing of ITGA2B and ITGB3
Gene: ITGB3
Variant: NM_000212.2:c.1801T>C (p.Cys601Arg)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA8623361
gnomAD: MAF in the European (non-Finnish) population is 0.00001758 https://gnomad.broadinstitute.org/variant/17-45376784-T-C
Supplementary Table S1 & S2 Proband and Variant Information:
Case65: GT68, Male, 62 years old, French
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Occasional spontaneous bleeding and frequency decreased with age
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry showed <5% expression of αIIbβ3 (See Supplementary Table 1)
PlateletAggregation: Absent platelet aggregation (See Supplementary Table 1)
CasePreviousTesting: Sanger sequencing of ITGA2B and ITGB3
Gene: ITGA2B
Variant: NM_000419.3:c.1413C>G (p.Tyr471Ter) and "LR deletion" which is the only specification we are given
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA290950376
gnomAD: MAF in the European (non-Finnish) population is 0.000008791 https://gnomad.broadinstitute.org/variant/17-42457994-G-C
Supplementary Table S1 & S2 Proband and Variant Information:
Case22: GT25, Male, 81, French
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: HP:0002239
Gastrointestinal bleeding
CaseHPOFreeText: Life-long bleeding disorder
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry showed <5% expression of αIIbβ3 (See Supplementary Table 1)
PlateletAggregation: Absent platelet aggregation (See Supplementary Table 1)
CasePreviousTesting: Sanger sequencing of ITGA2B and ITGB3
Gene: ITGA2B
Variant: NM_000419.3:c.1413C>G (p.Tyr471Ter) and NM_000419.3:c.1882C>T (p.Arg628Ter)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA290950376 and CA399801310
gnomAD: MAF in the European (non-Finnish) population 0.000008791 http://gnomad.broadinstitute.org/variant/17-42457994-G-C and MAF in Latino population is 0.00003983 http://gnomad.broadinstitute.org/variant/17-42456075-G-A
M10
Case18: (M10) Female, 37, French Gypsy
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Mucocutaneous bleeding
CaseNotHPOs: N/A
ProteinExpression: Failed to express alphaIIbbeta3 on the surface of their platelets
Platelet Aggregation: Absent platelet aggregation to all agonists (no mention for which agonists or response to ristocetin)
CasePreviousTesting: All the information we are given is "mutation screening of the ITGA2B and ITGB3 genes" the method for sequencing etc. was not stated
Gene: ITGA2B
Variant: NM_000419.3:c.1544+1G>A
LegacyVariant: N/A
ClinVar: 2895
CAiD: CA10575473
gnomAD N/A
Natural history of platelet antibody formation against αIIbβ3 in a French cohort of Glanzmann thrombasthenia patients M. FIORE Centre de Référence des Pathologies Plaquettaires (CRPP), Plateforme Technologique d’Innovation Biomédicale, Hôpital Xavier Arnozan, PessacSearch for more papers by this author N. FIRAH Hôpital des Enfants ‐ Hôpital Pellegrin – CHU Bordeaux, Unité d’Hématologie Pédiatrique, BordeauxSearch for more papers by this author X. PILLOIS Centre de Référence des Pathologies Plaquettaires (CRPP), Plateforme Technologique d’Innovation Biomédicale, Hôpital Xavier Arnozan, PessacSearch for more papers by this author P. NURDEN Centre de Référence des Pathologies Plaquettaires (CRPP), Plateforme Technologique d’Innovation Biomédicale, Hôpital Xavier Arnozan, PessacSearch for more papers by this author R. HEILIG Genoscope, Centre National de Séquençage, 91000, Evry, FranceSearch for more papers by this author A. T. NURDEN Centre de Référence des Pathologies Plaquettaires (CRPP), Plateforme Technologique d’Innovation Biomédicale, Hôpital Xavier Arnozan, PessacSearch for more papers by this author
PMID: 22250950
Gene: ITGA2B and ITGB3
HGNC: 6138 and 6156
M9
Case17: (M9) Female, 69 years old, French Gypsy
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Mucocutaneous bleeding
CaseNotHPOs: N/A
ProteinExpression: Failed to express alphaIIbbeta3 on the surface of their platelets
Platelet Aggregation: Absent platelet aggregation to all agonists (no mention for which agonists or response to ristocetin)
CasePreviousTesting: All the information we are given is "mutation screening of the ITGA2B and ITGB3 genes" the method for sequencing etc. was not stated
Gene: ITGA2B
Variant: NM_000419.3:c.1544+1G>A
LegacyVariant: N/A
ClinVar: 2895
CAiD: CA10575473
gnomAD N/A
GT2
Case2: (GT2) Male, 76 years old, French
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Mucocutaneous bleeding
CaseNotHPOs: N/A
ProteinExpression: <5% surface expression levels of αIIbβ3
Platelet Aggregation: Absent platelet aggregation to all agonists (no mention for which agonists or response to ristocetin)
CasePreviousTesting: All the information we are given is "mutation screening of the ITGA2B and ITGB3 genes" the method for sequencing etc. was not stated
Gene: ITGB3
Variant: NM_000212.2:c.1260G>A
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA8623200
gnomAD MAF in the African population is 0.00004005 https://gnomad.broadinstitute.org/variant/17-45368454-G-A
Mutation analysis for a patient with Glanzmann thrombasthenia who produced a landmark isoantibody to the αIIbβ3 integrin
PMID: 20492470
Gene: ITGA2B
HGNC: 6138
13
Case13: Female, 9 years old, Indian
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: HP:0002248, HP:0000421, HP:0031364 (Haematemesis, epistaxis, ecchymosis)
CaseHPOFreeText:
CaseNotHPOs: N/A
CaseNotHPOFreeText: N/A
ProteinExpression: Diagnosis of GT was made on the basis of quantitive determination of receptor count by flow cytometric analysis of GPIIb-IIIa
PlateletAggregation: Diagnosis of GT was made on the basis of impaired platelet aggregation (platelet aggregation was tested against ristocetin, ADP, collagen, epinephrine and arachidonic acid)
CasePreviousTesting: Multiplex PCR by using GPIIb primers and CSGE analysis were performed
Gene: ITGA2B
Variant: NM_000419.3:c.2674del
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA915940582
gnomAD: N/A
12
Case12: Male, 3 years old, Indian
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Sibling is proband number 11 in this article
ParentalGenotype: N/A
CasePresentingHPOs: HP:0002249, HP:0000421 (Melena, epistaxis)
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOFreeText: N/A
ProteinExpression: Diagnosis of GT was made on the basis of quantitive determination of receptor count by flow cytometric analysis of GPIIb-IIIa
PlateletAggregation: Diagnosis of GT was made on the basis of impaired platelet aggregation (platelet aggregation was tested against ristocetin, ADP, collagen, epinephrine and arachidonic acid)
CasePreviousTesting: Multiplex PCR by using GPIIb primers and CSGE analysis were performed
Gene: ITGA2B
Variant: NM_000419.3:c.1600+1G>A
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA399802454
gnomAD: N/A
9
Case9: Female, 5 years old, Indian
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: HP:0000421 (Epistaxis)
CaseHPOFreeText: Hematomas at the site of injury
CaseNotHPOs: N/A
CaseNotHPOFreeText: N/A
ProteinExpression: HP:0000421
PlateletAggregation: Diagnosis of GT was made on the basis of impaired platelet aggregation (platelet aggregation was tested against ristocetin, ADP, collagen, epinephrine and arachidonic acid)
CasePreviousTesting: Multiplex PCR by using GPIIb primers and CSGE analysis were performed
Gene: ITGA2B
Variant: NM_000419.3:c.1230del
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA915940214
gnomAD: N/A
8
Case8: Female, 3 years old, Indian
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: HP:0000421, HP:0000225, HP:0031364 (Epistaxis, gingival bleeding, ecchymosis)
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOFreeText: N/A
ProteinExpression: Diagnosis of GT was made on the basis of quantitive determination of receptor count by flow cytometric analysis of GPIIb-IIIa
PlateletAggregation: Diagnosis of GT was made on the basis of impaired platelet aggregation (platelet aggregation was tested against ristocetin, ADP, collagen, epinephrine and arachidonic acid)
CasePreviousTesting: Multiplex PCR by using GPIIb primers and CSGE analysis were performed
Gene: ITGA2B
Variant: NM_000419.3:c.1073del
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA915940580
gnomAD: N/A
7
Case7: Male, 1.5 years old, Indian
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: HP:0031364, HP:0000421 (Ecchymosis, epistaxis)
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOFreeText: N/A
ProteinExpression: Diagnosis of GT was made on the basis of quantitive determination of receptor count by flow cytometric analysis of GPIIb-IIIa
PlateletAggregation: Diagnosis of GT was made on the basis of impaired platelet aggregation (platelet aggregation was tested against ristocetin, ADP, collagen, epinephrine and arachidonic acid)
CasePreviousTesting: Multiplex PCR by using GPIIb primers and CSGE analysis were performed
Gene: ITGA2B
Variant: NM_000419.3:c.2315C>G (p.Pro772Arg)
LegacyVariant: (p.Pro741Arg)
ClinVar: N/A
CAiD: CA8602740
gnomAD: MAF in the South Asian population is 0.0002613 https://gnomad.broadinstitute.org/variant/17-42453709-G-C
6
Case6: Female, 7 years old, Indian
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: HP:0000225 (Gingival bleeding)
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOFreeText: N/A
ProteinExpression: Diagnosis of GT was made on the basis of quantitive determination of receptor count by flow cytometric analysis of GPIIb-IIIa
PlateletAggregation: Diagnosis of GT was made on the basis of impaired platelet aggregation (platelet aggregation was tested against ristocetin, ADP, collagen, epinephrine and arachidonic acid)
CasePreviousTesting: Multiplex PCR by using GPIIb primers and CSGE analysis were performed
Gene: ITGA2B
Variant: NM_000419.3:c.1374C>G (p.Ile458Met)
LegacyVariant: (p.Ile427Met)
ClinVar: N/A
CAiD: CA8603157
gnomAD: MAF in the South Asian population is 0.003658 https://gnomad.broadinstitute.org/variant/17-42458266-G-C
5
Case5: Female, 3 years old, Indian
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: HP:0000421, HP:0000225
(Epistaxis, gingival bleeding)
CaseHPOFreeText: Severe epistaxis and mild gum bleeding
CaseNotHPOs: N/A
CaseNotHPOFreeText: N/A
ProteinExpression: Diagnosis of GT was made on the basis of quantitive determination of receptor count by flow cytometric analysis of GPIIb-IIIa
PlateletAggregation: Diagnosis of GT was made on the basis of impaired platelet aggregation (platelet aggregation was tested against ristocetin, ADP, collagen, epinephrine and arachidonic acid)
CasePreviousTesting: Multiplex PCR by using GPIIb primers and CSGE analysis were performed
Gene: ITGA2B
Variant: NM_000419.3:c.859G>C (p.Gly287Arg)
LegacyVariant: (p.Gly256Arg)
ClinVar: N/A
CAiD: CA399805008
gnomAD: N/A
4
Case4: Female, 18 years old, Indian
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: HP:0000225, HP:0000132
(Gingival bleeding, menorrhagia)
CaseHPOFreeText: Menorrhagia only during puberty
CaseNotHPOs: N/A
CaseNotHPOFreeText:N/A
ProteinExpression: Diagnosis of GT was made on the basis of quantitive determination of receptor count by flow cytometric analysis of GPIIb-IIIa
PlateletAggregation: Diagnosis of GT was made on the basis of impaired platelet aggregation (platelet aggregation was tested against ristocetin, ADP, collagen, epinephrine and arachidonic acid)
CasePreviousTesting: Multiplex PCR by using GPIIb primers and CSGE analysis were performed
Gene: ITGA2B
Variant: NM_000419.3:c.2264G>C (p.Arg755Pro)
LegacyVariant: (p.Arg724Pro)
ClinVar: N/A
CAiD: CA8602772
gnomAD: MAF in the European (non-Finnish) population is 0.00001060 https://gnomad.broadinstitute.org/variant/17-42454380-C-G
Supplementary Table S1:
Case29: GT34, USA, gender and age not specified
DiseaseAssertion: Glanzmann thrombasthenia type I
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: All patients had a life-long history of mucocutaneous bleeding
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: All patients in this study met the clinical and platelet function requirements for GT-- this includes an absent or strongly reduced expression of alphaIIbBeta3 in platelets
PlateletAggregation: All patients in this study met the clinical and platelet function requirements for GT-- this includes absent or severely reduced platelet aggregation with at least three physiological agonists
CasePreviousTesting: Sanger sequencing was performed. Sanger sequencing ensured coverage of all exons and splice sties of the ITGA2B and ITGB3 genes and extended 300-500 bp into the introns as well as including the UTR. They sequenced 416 bp upstream of the ITGA2B promoter and 463 bp upstream of the ITGB3 promoter. In total they sequenced 51.2% of the ITGA2B gene and 19.4% of the larger ITGB3 gene
Gene: ITGA2B
Variant: NM_000419.3:c.1553T>A (p.Ile518Asn) and NM_000419.3:c.1946+3G>T
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA8603038 and CA626121071
gnomAD: MAF in the European (non-Finnish) population is 0.000008790 https://gnomad.broadinstitute.org/variant/17-42457661-A-T and MAF in the African population for NM_000419.3:c.1946+3G>T is 0.000 https://gnomad.broadinstitute.org/variant/17-42456008-C-A
Supplementary Table S1:
Case43: GT50, French, age and gender not specified
DiseaseAssertion: Glanzmann thrombasthenia type I
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: All patients had a life-long history of mucocutaneous bleeding
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: All patients in this study met the clinical and platelet function requirements for GT-- this includes an absent or strongly reduced expression of alphaIIbBeta3 in platelets
PlateletAggregation: All patients in this study met the clinical and platelet function requirements for GT-- this includes absent or severely reduced platelet aggregation with at least three physiological agonists
CasePreviousTesting: Sanger sequencing was performed. Sanger sequencing ensured coverage of all exons and splice sties of the ITGA2B and ITGB3 genes and extended 300-500 bp into the introns as well as including the UTR. They sequenced 416 bp upstream of the ITGA2B promoter and 463 bp upstream of the ITGB3 promoter. In total they sequenced 51.2% of the ITGA2B gene and 19.4% of the larger ITGB3 gene
Gene: ITGB3
Variant: NM_000212.2:c.1260G>A (p.Lys376_Thr420del)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA8623200
gnomAD: MAF in the African population is 0.00004005 https://gnomad.broadinstitute.org/variant/17-45368454-G-A
Supplementary Table S1:
Case21: GT24, French Gypsy, gender and age not specified
DiseaseAssertion: Glanzmann thrombasthenia type I
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: All patients had a life-long history of mucocutaneous bleeding
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: All patients in this study met the clinical and platelet function requirements for GT-- this includes an absent or strongly reduced expression of alphaIIbBeta3 in platelets
PlateletAggregation: All patients in this study met the clinical and platelet function requirements for GT-- this includes absent or severely reduced platelet aggregation with at least three physiological agonists
CasePreviousTesting: Sanger sequencing was performed. Sanger sequencing ensured coverage of all exons and splice sties of the ITGA2B and ITGB3 genes and extended 300-500 bp into the introns as well as including the UTR. They sequenced 416 bp upstream of the ITGA2B promoter and 463 bp upstream of the ITGB3 promoter. In total they sequenced 51.2% of the ITGA2B gene and 19.4% of the larger ITGB3 gene
Gene: ITGA2B
Variant: NM_000419.3:c.1544+1G>A (p.Val513Leufs*71)
LegacyVariant: N/A
ClinVar: 2895
CAiD: CA10575473
gnomAD: N/A
Supplementary Table S1:
Case51: GT60, French/Italian, gender and age are not specified
DiseaseAssertion: Glanzmann thrombasthenia type I
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: All patients had a life-long history of mucocutaneous bleeding
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: All patients in this study met the clinical and platelet function requirements for GT-- this includes an absent or strongly reduced expression of alphaIIbBeta3 in platelets
PlateletAggregation: All patients in this study met the clinical and platelet function requirements for GT-- this includes absent or severely reduced platelet aggregation with at least three physiological agonists
CasePreviousTesting: Sanger sequencing was performed. Sanger sequencing ensured coverage of all exons and splice sties of the ITGA2B and ITGB3 genes and extended 300-500 bp into the introns as well as including the UTR. They sequenced 416 bp upstream of the ITGA2B promoter and 463 bp upstream of the ITGB3 promoter. In total they sequenced 51.2% of the ITGA2B gene and 19.4% of the larger ITGB3 gene
Gene: ITGB3
Variant: NM_000212.2:c.1801T>C (p.Cys601Arg)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA8623361
gnomAD: MAF in the European (non-Finnish) population is 0.00001758 https://gnomad.broadinstitute.org/variant/17-45376784-T-C
Supplementary Table S1:
Case53: GT62, French, gender and age are not specified
DiseaseAssertion: Glanzmann thrombasthenia type I
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: All patients had a life-long history of mucocutaneous bleeding
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: All patients in this study met the clinical and platelet function requirements for GT-- this includes an absent or strongly reduced expression of alphaIIbBeta3 in platelets
PlateletAggregation: All patients in this study met the clinical and platelet function requirements for GT-- this includes absent or severely reduced platelet aggregation with at least three physiological agonists
CasePreviousTesting: Sanger sequencing was performed. Sanger sequencing ensured coverage of all exons and splice sties of the ITGA2B and ITGB3 genes and extended 300-500 bp into the introns as well as including the UTR. They sequenced 416 bp upstream of the ITGA2B promoter and 463 bp upstream of the ITGB3 promoter. In total they sequenced 51.2% of the ITGA2B gene and 19.4% of the larger ITGB3 gene
Gene: ITGB3
Variant: NM_000212.2:c.31T>C (p.Trp11Arg)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA291240306
gnomAD: N/A
Supplementary Table S1:
Case26: GT30, "USA"-what the supp table states, gender and age not specified
DiseaseAssertion: Glanzmann thrombasthenia type I
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: All patients had a life-long history of mucocutaneous bleeding
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: All patients in this study met the clinical and platelet function requirements for GT-- this includes an absent or strongly reduced expression of alphaIIbBeta3 in platelets
PlateletAggregation: All patients in this study met the clinical and platelet function requirements for GT-- this includes absent or severely reduced platelet aggregation with at least three physiological agonists
CasePreviousTesting: Sanger sequencing was performed. Sanger sequencing ensured coverage of all exons and splice sties of the ITGA2B and ITGB3 genes and extended 300-500 bp into the introns as well as including the UTR. They sequenced 416 bp upstream of the ITGA2B promoter and 463 bp upstream of the ITGB3 promoter. In total they sequenced 51.2% of the ITGA2B gene and 19.4% of the larger ITGB3 gene
Gene: ITGB3
Variant: NM_000212.2:c.777+1G>A
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA8623036
gnomAD: MAF in the Latino population is 0.0006774 https://gnomad.broadinstitute.org/variant/17-45363789-G-A
Supplementary Table S1:
Case59: GT69, French, age and gender are not specified here
DiseaseAssertion: Glanzmann thrombasthenia type I
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: All patients had a life-long history of mucocutaneous bleeding
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: All patients in this study met the clinical and platelet function requirements for GT-- this includes an absent or strongly reduced expression of alphaIIbBeta3 in platelets
PlateletAggregation: All patients in this study met the clinical and platelet function requirements for GT-- this includes absent or severely reduced platelet aggregation with at least three physiological agonists
CasePreviousTesting: Sanger sequencing was performed. Sanger sequencing ensured coverage of all exons and splice sties of the ITGA2B and ITGB3 genes and extended 300-500 bp into the introns as well as including the UTR. They sequenced 416 bp upstream of the ITGA2B promoter and 463 bp upstream of the ITGB3 promoter. In total they sequenced 51.2% of the ITGA2B gene and 19.4% of the larger ITGB3 gene
Gene: ITGA2B
Variant: NM_000419.3:c.1878G>C (p.Gln626His/ p.Asp585_Gln626del)
LegacyVariant: N/A
ClinVar: 2903
CAiD: CA115852
gnomAD: N/A
Supplementary Table S1:
Case14: GT15, French, gender and age are not specified
DiseaseAssertion: Glanzmann thrombasthenia type I
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: All patients had a life-long history of mucocutaneous bleeding
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: All patients in this study met the clinical and platelet function requirements for GT-- this includes an absent or strongly reduced expression of alphaIIbBeta3 in platelets
PlateletAggregation: All patients in this study met the clinical and platelet function requirements for GT-- this includes absent or severely reduced platelet aggregation with at least three physiological agonists
CasePreviousTesting: Sanger sequencing was performed. Sanger sequencing ensured coverage of all exons and splice sties of the ITGA2B and ITGB3 genes and extended 300-500 bp into the introns as well as including the UTR. They sequenced 416 bp upstream of the ITGA2B promoter and 463 bp upstream of the ITGB3 promoter. In total they sequenced 51.2% of the ITGA2B gene and 19.4% of the larger ITGB3 gene
Gene: ITGA2B
Variant: NM_000419.3:c.2095-19T>A and NM_000419.3:c.2770C>T
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA915940312 and CA399792054
gnomAD: N/A
Supplementary Table S1:
Case37: GT43a, Morrocco, gender and age not specified
DiseaseAssertion: Glanzmann thrombasthenia type I
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: All patients had a life-long history of mucocutaneous bleeding
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: All patients in this study met the clinical and platelet function requirements for GT-- this includes an absent or strongly reduced expression of alphaIIbBeta3 in platelets
PlateletAggregation: All patients in this study met the clinical and platelet function requirements for GT-- this includes absent or severely reduced platelet aggregation with at least three physiological agonists
CasePreviousTesting: Sanger sequencing was performed. Sanger sequencing ensured coverage of all exons and splice sties of the ITGA2B and ITGB3 genes and extended 300-500 bp into the introns as well as including the UTR. They sequenced 416 bp upstream of the ITGA2B promoter and 463 bp upstream of the ITGB3 promoter. In total they sequenced 51.2% of the ITGA2B gene and 19.4% of the larger ITGB3 gene
Gene: ITGB3
Variant: NM_000212.2:c.614+1G>T and NM_000212.2:c.683G>A (p.Arg228His)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA400023344 and CA8623025
gnomAD: MAF in the African population is 0.00008012 https://gnomad.broadinstitute.org/variant/17-45363694-G-A
Supplementary Table S1:
Case35: GT41a, Canadian/Chinese, gender and age not specified here
DiseaseAssertion: Glanzmann thrombasthenia type I
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: All patients had a life-long history of mucocutaneous bleeding
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: All patients in this study met the clinical and platelet function requirements for GT-- this includes an absent or strongly reduced expression of alphaIIbBeta3 in platelets
PlateletAggregation: All patients in this study met the clinical and platelet function requirements for GT-- this includes absent or severely reduced platelet aggregation with at least three physiological agonists
CasePreviousTesting: Sanger sequencing was performed. Sanger sequencing ensured coverage of all exons and splice sties of the ITGA2B and ITGB3 genes and extended 300-500 bp into the introns as well as including the UTR. They sequenced 416 bp upstream of the ITGA2B promoter and 463 bp upstream of the ITGB3 promoter. In total they sequenced 51.2% of the ITGA2B gene and 19.4% of the larger ITGB3 gene
Gene: ITGA2B
Variant: NM_000419.3:c.91del (p.Ala31ProfsTer2) and NM_000419.3:c.2915dup (p.Leu973AlafsTer63)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA913203486 and CA8602531
gnomAD: MAF in the East Asian population is 0.0001088 https://gnomad.broadinstitute.org/variant/17-42452054-C-CG for the variant (p.Leu973Alafs*63)
Supplementary Table S1:
Case1: GT1, French/African, Gender or age not specified
DiseaseAssertion: Glanzmann thrombasthenia type I
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: All patients had a life-long history of mucocutaneous bleeding
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: All patients in this study met the clinical and platelet function requirements for GT-- this includes an absent or strongly reduced expression of alphaIIbBeta3 in platelets
PlateletAggregation: All patients in this study met the clinical and platelet function requirements for GT-- this includes absent or severely reduced platelet aggregation with at least three physiological agonists
CasePreviousTesting: Sanger sequencing was performed. Sanger sequencing ensured coverage of all exons and splice sties of the ITGA2B and ITGB3 genes and extended 300-500 bp into the introns as well as including the UTR. They sequenced 416 bp upstream of the ITGA2B promoter and 463 bp upstream of the ITGB3 promoter. In total they sequenced 51.2% of the ITGA2B gene and 19.4% of the larger ITGB3 gene
Gene: ITGB3
Variant: NM_000212.2:c.1913+1G>A
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA400032825
gnomAD: N/A
Supplementary Table S1:
Case11: GT12, French, age and gender not specified
DiseaseAssertion: Glanzmann thrombasthenia type I
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: All patients had a life-long history of mucocutaneous bleeding
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: All patients in this study met the clinical and platelet function requirements for GT-- this includes an absent or strongly reduced expression of alphaIIbBeta3 in platelets
PlateletAggregation: All patients in this study met the clinical and platelet function requirements for GT-- this includes absent or severely reduced platelet aggregation with at least three physiological agonists
CasePreviousTesting: Sanger sequencing was performed. Sanger sequencing ensured coverage of all exons and splice sties of the ITGA2B and ITGB3 genes and extended 300-500 bp into the introns as well as including the UTR. They sequenced 416 bp upstream of the ITGA2B promoter and 463 bp upstream of the ITGB3 promoter. In total they sequenced 51.2% of the ITGA2B gene and 19.4% of the larger ITGB3 gene
Gene: ITGB3
Variant: NM_000212.2:c.565C>T (p.Pro189Ser)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA291224669
gnomAD: MAF in the European (non-Finnish) population is 0.00002638 https://gnomad.broadinstitute.org/variant/17-45362012-C-T
Patients With Glanzmann Thrombasthenia Lacking Platelet Glycoprotein αIIbβ3 (GPIIb/IIIa) and αvβ3 Receptors Are Not Protected From Atherosclerosis
PMID: 11877352
Gene: ITGA2B and/or ITGB3
HGNC: 6138 and 6156
InsufficientEvidence: This article mentions GT patients and 2 variants in ITGA2B and/or ITGB3 that have been seen in these patients; however, there is no direct correlation for which patients harbor which variant; therefore there is insufficient evidence
"Five were homozygous for the 11-bp deletion in the β3 gene11 and 2 were homozygous for a splice site mutation of the αIIb gene"
M/11
Case7: Male, 1 years old, West Bengal India
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: HP:0000225, HP:0006298, HP:0000967
(Gingival bleeding, prolonged bleeding after tooth extraction, petechiae)
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: See Table 1 for values
PlateletAggregation: See Table 1 for values
CasePreviousTesting: PCR amplification of ITAG2B and ITGB3
Gene: ITGA2B
Variant: NM_000419.3:c.1651C>T (p.Arg551Trp)
LegacyVariant: (p.Arg520Trp)
ClinVar: N/A
CAiD: CA399802348
gnomAD: MAF in the East Asian population is 0.00005045https://gnomad.broadinstitute.org/variant/17-42457471-G-A
Diversity of Glanzmann thrombasthenia in southern India: 10 novel mutations identified among 15 unrelated patients E. J. R. NELSON Departments of HematologySearch for more papers by this author S. C. NAIR Clinical Pathology, Christian Medical College, Vellore, IndiaSearch for more papers by this author H. PERETZ Clinical Biochemistry Laboratory, Tel Aviv Sourasky Medical Center, Tel‐Aviv, IsraelSearch for more papers by this author B. S. COLLER Laboratory of Blood and Vascular Biology, The Rockefeller University, New York, NY, USASearch for more papers by this author U. SELIGSOHN Institute of Thrombosis and Hemostasis, The Chaim Sheba Medical Center, Tel‐Hashomer, IsraelSearch for more papers by this author M. CHANDY Departments of HematologySearch for more papers by this author A. SRIVASTAVA Departments of HematologySearch for more papers by this author
PMID: 16879215
Gene: ITGA2B and ITBG3
HGNC: 6138 and 6156
Molecular basis ofGlanzmann’sThrombasthenia andcurrent strategies intreatment
PMID: 12163005
Gene: ITGA2B and/or ITGB3
HGNC: 6138 and 6156
InsufficientEvidence: This article is a review on GT.
GT 11
Case19: GT11, Female, India, age of onset is 12 years old
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Patients were recruited as they presented consecutively from 1996 to 1998 for evaluation of a mucocutaneous bleeding disorder
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: N/A
PlateletAggregation: All patients examined had defective ADP, epinephrine, and collagen induced aggregation and normal ristocetin induced aggregation
CasePreviousTesting: Mutations were detected by the method of single strand conformation polymorphism (SSCP) analysis, followed by nucleotide sequencing and confirmation by restriction fragment length polymorphism (RFLP) assays. The sequences of the primers employed and the reaction conditions for PCR amplification of the exons, adjacent intronic regions, and 50 and 30 untranslated regions of the aIIb and b3 gene
Gene: ITGB3
Variant: NM_000212.2:c.115T>G (p.Cys39Gly)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA400031666
gnomAD: N/A
GT 04
Case15: GT04, Female, India, age of onset is 11 years old
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: Menorrhagia HP:0000132
CaseHPOFreeText: Patients were recruited as they presented consecutively from 1996 to 1998 for evaluation of a mucocutaneous bleeding disorder
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: N/A
PlateletAggregation: All patients examined had defective ADP, epinephrine, and collagen induced aggregation and normal ristocetin induced aggregation
CasePreviousTesting: Mutations were detected by the method of single strand conformation polymorphism (SSCP) analysis, followed by nucleotide sequencing and confirmation by restriction fragment length polymorphism (RFLP) assays. The sequences of the primers employed and the reaction conditions for PCR amplification of the exons, adjacent intronic regions, and 50 and 30 untranslated regions of the aIIb and b3 gene
Gene: ITGB3
Variant: NM_000212.2:c.262C>T (p.Arg88Ter) and NM_000212.2:c.356G>A (p.Arg119Gln)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA400020703 and CA8622925
gnomAD: MAF in the Other population is 0.0001385 https://gnomad.broadinstitute.org/variant/17-45360816-C-T and MAF in the African population is 0.0002470 https://gnomad.broadinstitute.org/variant/17-45360910-G-A
GT 07
Case16: GT07, Male, India, age of onset is 3 years old
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Patients were recruited as they presented consecutively from 1996 to 1998 for evaluation of a mucocutaneous bleeding disorder
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: N/A
PlateletAggregation: All patients examined had defective ADP, epinephrine, and collagen induced aggregation and normal ristocetin induced aggregation
CasePreviousTesting: Mutations were detected by the method of single strand conformation polymorphism (SSCP) analysis, followed by nucleotide sequencing and confirmation by restriction fragment length polymorphism (RFLP) assays. The sequences of the primers employed and the reaction conditions for PCR amplification of the exons, adjacent intronic regions, and 50 and 30 untranslated regions of the aIIb and b3 gene
Gene: ITGB3
Variant: NM_000212.2:c.153del (p.Trp51CysfsTer5)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA915940728
gnomAD: N/A
GT 55
Case34: GT55, Male, India, age of onset is 21 years old
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Patients were recruited as they presented consecutively from 1996 to 1998 for evaluation of a mucocutaneous bleeding disorder
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: N/A
PlateletAggregation: All patients examined had defective ADP, epinephrine, and collagen induced aggregation and normal ristocetin induced aggregation
CasePreviousTesting: Mutations were detected by the method of single strand conformation polymorphism (SSCP) analysis, followed by nucleotide sequencing and confirmation by restriction fragment length polymorphism (RFLP) assays. The sequences of the primers employed and the reaction conditions for PCR amplification of the exons, adjacent intronic regions, and 50 and 30 untranslated regions of the aIIb and b3 gene
Gene: ITGB3
Variant: NM_000212.2:c.921C>A (p.Tyr307Ter)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA400025209
gnomAD: N/A
Molecular Diversity of Glanzmann Thrombastheniain Southern India: New Insights Into mRNASplicing and Structure–Function CorrelationsofaIIbb3 Integrin (ITGA2B, ITGB3)
PMID: 16463284
Gene: ITGA2B and ITGB3
HGNC: 6156 and 6156
GT 24
Case6: GT24, Male, India, age of onset is 10 years old
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Patients were recruited as they presented consecutively from 1996 to 1998 for evaluation of a mucocutaneous bleeding disorder
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: N/A
PlateletAggregation: All patients examined had defective ADP, epinephrine, and collagen induced aggregation and normal ristocetin induced aggregation
CasePreviousTesting: Mutations were detected by the method of single strand conformation polymorphism (SSCP) analysis, followed by nucleotide sequencing and confirmation by restriction fragment length polymorphism (RFLP) assays. The sequences of the primers employed and the reaction conditions for PCR amplification of the exons, adjacent intronic regions, and 50 and 30 untranslated regions of the aIIb and b3 gene
Gene: ITGA2B
Variant: NM_000419.3:c.2898_2902dup
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA915940725
gnomAD: N/A
3
Case3: Gender or age not specified, Thai ethnicity
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Parental information available in Table 2
ParentalGenotype: Father: p.Arg782Ter/WT and Mother: Unavailable
Note: No parental consanguinity
CasePresentingHPOs: N/A
CaseHPOFreeText: The article does not mention specific bleeding phenotypes for each of the 4 probands in the article, rather, there is a general statement that says: "The common presentations included recurrent epistaxis, bleeding in oral cavity, and easy-bruising spontaneously or after trauma"
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinEpression: General statement from the article that states that flow cytometry to assess the surface GPIIb/IIIa complex showed reduced expression for all probands.
PlateletAggregation: The platelet aggregation test was typically defective with all physiological agonists except ristocetin, for all patients.
CasePreviousTesting: Whole exome sequencing was performed on the 30 exons of ITGA2B and 15 exons in ITGB3. PCR and direct sequencing were then performed in the DNA samples of the patients and their available parents to verify the presence of the identified mutations.
Gene: ITGA2B
Variant: NM_000419.3:c.2344C>T (p.Arg782Ter) and NM_000419.3:c.2915dup (p.Leu973Alafs*63)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA399796586 (p.Arg782Ter) and CA8602531 (p.Leu973Alafs*63)
gnomAD: MAF for (p.Leu973Alafs*63) in East Asian population 0.0001088 http://gnomad.broadinstitute.org/variant/17-42452054-C-CG
2
Case2: Gender or age not specified, Thai ethnicity
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Parental information available in Table 2
ParentalGenotype: Father: p.Val206Asp/WT and Mother: also p.Val206Asp/WT (See Table 2)
Note: Yes to parental consanguinity
CasePresentingHPOs: N/A
CaseHPOFreeText: The article does not mention specific bleeding phenotypes for each of the 4 probands in the article, rather, there is a general statement that says: "The common presentations included recurrent epistaxis, bleeding in oral cavity, and easy-bruising spontaneously or after trauma"
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinEpression: General statement from the article that states that flow cytometry to assess the surface GPIIb/IIIa complex showed reduced expression for all probands.
PlateletAggregation: The platelet aggregation test was typically defective with all physiological agonists except ristocetin, for all patients.
CasePreviousTesting: Whole exome sequencing was performed on the 30 exons of ITGA2B and 15 exons in ITGB3. PCR and direct sequencing were then performed in the DNA samples of the patients and their available parents to verify the presence of the identified mutations.
Gene: ITGA2B
Variant: NM_000419.3:c.617T>A (p.Val206Asp)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA399805578
gnomAD: N/A
Supplementary Tables 1 and 2 combined information:
Case22: GT22, Female, 16 years old
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Not available per supplementary table 1
ParentalGenotype: Parents are not consanguineous, no other genotype information available
CasePresentingHPOs: HP:0000132 (Menorrhagia)
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Showed <5% of αIIbβ3 expression measured by flow cytometry
PlateletAggregation: Platelet aggregation showed no response to ADP and collagen but normal response to ristocetin
CasePreviousTesting: All exons from ITGA2B and ITGB3 genes were amplified by PCR using intronic primers. DNA sequencing was determined by Sanger sequencing.
Gene: ITGA2B
Variant: NM_000419.3:c.1750C>T (p.Arg584Ter)
LegacyVariant: N/A
ClinVar: 2892
CAiD: CA115834
gnomAD: MAF in the East Asian population: 0.0002176 https://gnomad.broadinstitute.org/variant/17-42457372-G-A
Supplementary Tables 1 and 2 combined information:
Case4: GT4, Female, 22 years old
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Not available per supplementary table 1
ParentalGenotype: Parents are consanguineous, no other genotype information available
CasePresentingHPOs: HP:0000978, HP:0000979, HP:0031364, HP:0000421, HP:0000225, HP:0000132
(Bruising, purpura, ecchymosis, epistaxis, gingival bleeding, menorrhagia)
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Showed <5% of αIIbβ3 expression measured by flow cytometry
PlateletAggregation: Platelet aggregation showed no response to ADP and collagen but normal response to ristocetin
CasePreviousTesting: All exons from ITGA2B and ITGB3 genes were amplified by PCR using intronic primers. DNA sequencing was determined by Sanger sequencing.
Gene: ITGA2B
Variant: NM_000419.3:c.2671C>T (p.Gln891Ter)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA8602626
gnomAD: N/A
Supplementary Tables 1 and 2 combined information:
Case41: GT41, Male, 5 years old
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Not available per supplementary table 1
ParentalGenotype: Parents are not consanguineous, no other genotype information available
CasePresentingHPOs: HP:0000978, HP:0000979, HP:0031364, HP:0000421
(Bruising, purpura, ecchymosis, epistaxis)
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Showed 10-20% of αIIbβ3 expression measured by flow cytometry
PlateletAggregation: Platelet aggregation showed no response to ADP and collagen but normal response to ristocetin
CasePreviousTesting: All exons from ITGA2B and ITGB3 genes were amplified by PCR using intronic primers. DNA sequencing was determined by Sanger sequencing.
Gene: ITGB3
Variant: NM_000212.2:c.1736G>A (p.Trp579Ter) and NM_000212.2:c.1784_1802delinsGTCACA
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA400032429 (p.Trp579Ter) and CA915940255 (c.1784_1802delinsGTCACA)
gnomAD: N/A
Supplementary Tables 1 and 2 combined information:
Case8: GT8, Male, 9 years old
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Not available per supplementary table 1
ParentalGenotype: Parents are not consanguineous, no other genotype information available
CasePresentingHPOs: HP:0000978, HP:0000979, HP:0031364, HP:0000421
(Bruising, purpura, ecchymosis, epistaxis)
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Showed <5% of αIIbβ3 expression measured by flow cytometry
PlateletAggregation: Platelet aggregation showed no response to ADP and collagen but normal response to ristocetin
CasePreviousTesting: All exons from ITGA2B and ITGB3 genes were amplified by PCR using intronic primers. DNA sequencing was determined by Sanger sequencing.
Gene: ITGA2B
Variant: NM_000419.3:c.1651C>T (p.Arg551Trp) and NM_000419.3:c.2236G>T (p.Glu746Ter)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA399802348 (p.Arg551Trp) and CA399798321 (p.Glu746Ter)
gnomAD: N/A
Supplementary Tables 1 and 2 combined information:
Case38: GT38, Female, 23 years old
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Not available per supplementary table 1
ParentalGenotype: Parents are not consanguineous, no other genotype information available
CasePresentingHPOs: HP:0000978, HP:0000979, HP:0031364, HP:0000132
(Bruising, purpura, ecchymosis, menorrhagia)
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Showed <5% of αIIbβ3 expression measured by flow cytometry
PlateletAggregation: Platelet aggregation showed no response to ADP and collagen but normal response to ristocetin
CasePreviousTesting: All exons from ITGA2B and ITGB3 genes were amplified by PCR using intronic primers. DNA sequencing was determined by Sanger sequencing.
Gene: ITGA2B and ITGB3
Variant: NM_000419.3:c.2613del in ITGA2B (p.Leu872Cysfs*38) and NM_000212.2:c.1646G>C in ITGB3 (p.Cys549Ser)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA915940594 (p.Leu872Cysfs*38) and CA400030198 (p.Cys549Ser)
gnomAD: MAF of (p.Cys549Ser) in the East Asian population: 0.00005453 https://gnomad.broadinstitute.org/variant/17-45369890-G-C
Supplementary Tables 1 and 2 combined information:
Case33: GT33, Female, 12 years old
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Not available per supplementary table 1
ParentalGenotype: Parents are consanguineous, no other genotype information available
CasePresentingHPOs: HP:0000978, HP:0000979, HP:0031364, HP:0000421
(Bruising, purpura, ecchymosis, epistaxis)
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Showed <5% of αIIbβ3 expression measured by flow cytometry
PlateletAggregation: Platelet aggregation showed a small response to ADP, no response to collagen but normal response to ristocetin
CasePreviousTesting: All exons from ITGA2B and ITGB3 genes were amplified by PCR using intronic primers. DNA sequencing was determined by Sanger sequencing.
Gene: ITGB3
Variant: NM_000212.2:c.719G>A (p.Arg240Gln)
LegacyVariant: N/A
ClinVar: 13553
CAiD: CA123224
gnomAD: N/A
Supplementary Tables 1 and 2 combined information:
Case31: GT31, Female, 13 years old
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Not available per supplementary table 1
ParentalGenotype: Parents are not consanguineous, no other genotype information available
CasePresentingHPOs: HP:0000978, HP:0000979, HP:0031364, HP:0000421, HP:0000225
(Bruising, purpura, ecchymosis, epistaxis, gingival bleeding)
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Showed <5% of αIIbβ3 expression measured by flow cytometry
PlateletAggregation: Platelet aggregation showed no response to ADP and collagen but normal response to ristocetin
CasePreviousTesting: All exons from ITGA2B and ITGB3 genes were amplified by PCR using intronic primers. DNA sequencing was determined by Sanger sequencing.
Gene: ITGA2B and ITGB3
Variant: NM_000419.3:c.1022C>T (p.Ala341Val) in ITGA2B and NM_000212.2:c.412G>A (p.Val138Met) in ITGB3
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA399804629 (p.Ala341Val) and CA400021826 (p.Val139Met)
gnomAD: N/A
Supplementary Tables 1 and 2 combined information:
Case23: GT23, Female, 11 years old
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Not available per supplementary table 1
ParentalGenotype: Parents are consanguineous, no other genotype information available
CasePresentingHPOs: HP:0000978, HP:0000979, HP:0031364, HP:0000421, HP:0000225, HP:0000132
(Bruising, purpura, ecchymosis, epistaxis, gingival bleeding, menorrhagia)
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Showed <5% of αIIbβ3 expression measured by flow cytometry
PlateletAggregation: Platelet aggregation showed a small response to ADP, no response to collagen but normal response to ristocetin
CasePreviousTesting: All exons from ITGA2B and ITGB3 genes were amplified by PCR using intronic primers. DNA sequencing was determined by Sanger sequencing.
Gene: ITGB3
Variant: NM_000212.2:c.1980C>A (p.Tyr660Ter) and NM_000212.2:c.2147G>T (p.Gly716Val)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA400032983 (p.Tyr660Ter) and CA400033923 (p.Gly716Val)
gnomAD: N/A
A rare case of Glanzmann's thrombasthenia and factor VII deficiency due to a combination of pathogenic and non-pathogenic gene variants
PMID: 31777146
Disease: Glazmann's Thrombasthenia type 1 and factor 7 deficiency
Case: 13 year old male, from non-consanguineous
CasePresentingHPOs: HP:0011889, HP:0000978, HP:0031364,
GenotypingMethod: Direct sequencing of all coding, splice site and promoter regions of F7 and ITGA2B and ITGB3 genes. Heterozygous for two ITGB3 variants predicted to be deleterious in silico.
First heterozygous mutation:
ClinVar: Not found
CAID: CA400022111
Variant: NM_000212.3:c.473A>T
GnomAD: Not found
Second heterozygous mutation:
ClinVar: Not found
CAID: CA400033092
Variant: NM_000212.3:c.2027A>C
GnomAD: Not found
PlateletAggregation: Platelet aggregation studies showed absent aggregation with adenosine diphosphate (ADP), collagen and was reduced with ristocetin
ExpressionEvidence: flow cytometry studies for platelet receptors showed absence of αIIbβ3 integrin and normal levels of GPIb and GPIX receptors
Patient 2
Case2: Male, 38 years old, Korean
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: Figure 1 shows haplotype analysis
CasePresentingHPOs: HP:0004406, HP:0000225
(Recurrent epistaxis, gingival bleeding)
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry analyses revealed markedly decreasedCD41/CD61 expression (<1%) in Patient 2 (type I GT).
PlateletAggregation: Platelet aggregation tests were performed to evaluate responses to ADP, collagen, epinephrine, and ristocetin agonists
CasePreviousTesting: Polymerase chain reaction for all exons ofITGA2Band/orITGB3were performed using primers. Sequence analysis was performed with reference to M_000419.3 andNP_000410.2 forITGA2Band NM_000212.2 and NP_000203.2 for ITGB3.
Gene: ITGB3
Variant: NM_000212.2:c.1913+5G>T
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA8623377
gnomAD: MAF in the East Asian population is 0.0001633 http://gnomad.broadinstitute.org/variant/17-45376901-G-T
Independent adjudicator assessments of platelet refractorinessand rFVIIa efficacy in bleeding episodes and surgeries from themultinational Glanzmann’s thrombasthenia registry
PMID: 28370369
Gene: ITGA2B and ITGB3
HGNC: 6138 and 6156
NoEvidence: No GT probands or variants within ITGA2B or ITGB3 mentioned in this article
CW
Case12: Patient CW (can be found in GT database) gender and age are not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: HP:0031364, HP:0000979, HP:0000967, (Ecchymosis, Purpura/petechiae)
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: <1% of normal GPIIIa and GPIIb surface expression
PlateletAggregation: N/A
CasePreviousTesting: N/A
Gene: ITGA2B
Variant: NM_000419.3:c.1750C>T (p.Arg584Ter) and NM_000419.3:c.480C>G (p.Ser160_102del)
LegacyVariant: N/A
ClinVar: 2892 (Arg584Ter)
CAiD: CA115834 and CA399805892
gnomAD: MAF in the East Asian population is 0.0002176 for the Arg584Ter variant and MAF in the East Asian population is 0.00005507 for the other variant https://gnomad.broadinstitute.org/variant/17-42463013-G-C
PreviouslyPublished: Reference number 23 in this article
I-J1
Case38: Patient IJ12
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: N/A
CaseNotHPOs: HP:0000421, HP:0000225, HP:0002239, HP:0000979, HP:0000967, HP:0006298
(Epistaxis, gingival bleeding, GI bleeding, purpura/petechiae, prolonged bleeding after tooth extraction)
CaseNotHPOsFreeText: N/A
ProteinExpression: <2% GPIIIa and GPIIb surface expression
PlateletAggregation: N/A
CasePreviousTesting: N/A
Gene: ITGB3
Variant: NM_000212.2:c.2031_2041del
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA915940605
gnomAD: N/A
PreviouslyPublished: References 8-12
Arab
Case2: Arab Patient (found in the GT database) gender and age are not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: HP:0000421, HP:0000225, HP:0002239, HP:0000132, HP:0000967, HP:0000979, HP:0006298
(Epistaxis, gingival bleeding, GI bleeding, menorrhagia, petechiae/purpura, prolonged , bleeding after tooth extraction)
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: <2% of normal GPIIIa and GPIIb surface expression
PlateletAggregation: N/A
CasePreviousTesting: N/A
Gene: ITGA2B
Variant: IVS(-3)-418del (May be registered; however, need to consult senior biocurator)
LegacyVariant: ^
ClinVar: ^
CAiD: ^
gnomAD: ^
PreviouslyPublished: References 8-12
PDJ
Case28: Patient PDJ
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: N/A
PlateletAggregation: N/A
CasePreviousTesting: N/A
Gene: ITGB3
Variant: NM_000212.2:c.709_710del
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA8623029
gnomAD: MAF in the European non Finnish population is 0.00001796 https://gnomad.broadinstitute.org/variant/17-45363717-CAG-C
PreviouslyPublished: Reference 45
NR
Case19: Patient NR (found in the GT database as well) gender and age are not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: Menorrhagia HP:0000132
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: N/A Undetectable
PlateletAggregation: N/A
CasePreviousTesting: N/A
Gene: ITGA2B
Variant: NM_000419.3:c.2941C>T
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA399790399
gnomAD: N/A
PreviouslyPublished: Reference numbers 32,22 in this article
Glanzmann Thrombasthenia in a Newborn with Heterozygous Factor VLeiden and Heterozygous MTHFR C677T Gene Mutations
PMID: 30819996
Gene: ITGA2B and/or ITGB3 (Proband has GT; therefore, one of these variants may be implicated)
HGNC: 6138 and 6156
InsufficientEvidence: Case report presented in this article with Glanzmann thrombasthenia; however, sequencing reveals differential diagnosis to GT, with mutations in other genes.
J
Case10: Patient ID J, Female, 5 years old, ethnicity not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Related to Patient's I and K (See Table 1)
ParentalGenotype: N/A (Parents are consangenous)
CasePresentingHPOs: HP:0000978, HP:0004406, HP:0000225, HP:0002239
(Easy bruising, recurrent epistaxis, gingival bleeding, GI bleeding
CaseHPOFreeText: Hemorrhage after trauma, hematoma
CaseNotHPOs: HP:0000967, HP:0000979, HP:0002239, HP:0000790, HP:0005261, HP:0002248
(Petechiae, purpura, intracranial hemorrhage, hematuria, hemarthrosis, hematemesis)
CaseNotHPOsFreeText: Hemorrhage after dental, ear bleeding
ProteinExpression: N/A
PlateletAggregation: Defective response to ADP, adrenaline, collagen, arachidonic acid, and normal response to ristocetin
CasePreviousTesting: Exon PCR was used to amplify coding regions of the ITGA2B and ITGB3 genes. PCR products were purified and Sanger sequenced. Segregation using parental samples was undertaken when available.
Gene: ITGA2B
Variant: NM_000419.3:c.1652G>A
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA8602986
gnomAD: MAF in South Asian population is 0.00006536 http://gnomad.broadinstitute.org/variant/17-42457470-C-T
PreviouslyPublished: PMID: 19172520
I
Case9: Patient ID I, Female, 3 years old, ethnicity not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Related to Patient's J and K (See Table 1)
ParentalGenotype: N/A (Parents are consangenous)
CasePresentingHPOs: HP:0000967, HP:0000979, HP:0000978, HP:0004406
(Petechiae, purpura, easy bruising, recurrent epistaxis)
CaseHPOFreeText: N/A
CaseNotHPOs: HP:0000225, HP:0002239, HP:0002239, HP:0000790, HP:0005261, HP:0002248
(Gingival bleeding, GI bleeding, intracranial hemorrhage, hematuria, hemarthrosis, hematemesis)
CaseNotHPOsFreeText: Hemorrhage after dnetal, hemorrhage after trauma, ear bleeding, hematoma
ProteinExpression: N/A
PlateletAggregation: Defective response to ADP, adrenaline, collagen, arachidonic acid, and normal response to ristocetin
CasePreviousTesting: Exon PCR was used to amplify coding regions of the ITGA2B and ITGB3 genes. PCR products were purified and Sanger sequenced. Segregation using parental samples was undertaken when available.
Gene: ITGA2B
Variant: NM_000419.3:c.1652G>A
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA8602986
gnomAD: MAF in South Asian population is 0.00006536 http://gnomad.broadinstitute.org/variant/17-42457470-C-T
PreviouslyPublished: PMID: 19172520
Acquired Glanzmann's thrombasthenia caused by glycoprotein IIb/IIIa autoantibodies of the immunoglobulin G1 (IgG1), IgG2 or IgG4 subclass: a study in six cases.
PMID: 19138263
Gene: ITGA2B and/or ITGB3
HGNC: 6138 and 6156
InsufficientEvidence: This article mentions probands with GT; however, no mention of variants in ITGA2B and/or ITGB3
Note: Full-text found on UNC library website-- was not able to annotate on that document therefore annotation found here
23.
Case23: Female, age not specified
DiseaseAssertion: Glanzmann thrombasthenia type 1
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: N/A
PlateletAggregation: Minimal information, not specific enough
CasePreviousTesting: Sanger sequencing identified disease-causing variants in 18 out of 20 patients (Probands 6 and 14 excluded) with istoantibodies to αIIbβ3. The mutations were evenly distributed between ITGB3 and ITGA2B.
Gene: ITGB3
Variant: All Table 1 states is "missense mutation" not enough information provided
LegacyVariant: N/A
ClinVar: N/A
CAiD: N/A
gnomAD: N/A
1.
Case1: Male, now deceased
DiseaseAssertion: Glanzmann thrombasthenia type I
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: HP:0002239
(Gastrointestinal bleeding)
CaseHPOFreeText: Severe bleeding when young
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: N/A
PlateletAggregation: Minimal information, not specific at all
CasePreviousTesting: Sanger sequencing identified disease-causing variants in 18 out of 20 patients (Probands 6 and 14 excluded) with istoantibodies to αIIbβ3. The mutations were evenly distributed between ITGB3 and ITGA2B.
Gene: ITGA2B
Variant: NM_000419.3:c.1413C>G (p.Tyr471Ter) Codon change confirmed from PMID: 20492470 and NM_000419.3:c.1882C>T (p.Arg628Ter)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA290950376 and CA399801310
gnomAD: MAF in European non Finnish population 0.000008791 http://gnomad.broadinstitute.org/variant/17-42457994-G-C and MAF in Latino population 0.00003983 http://gnomad.broadinstitute.org/variant/17-42456075-G-A
22.
Case22: Female, age not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: N/A
PlateletAggregation: Minimal information, not specific enough
CasePreviousTesting: Sanger sequencing identified disease-causing variants in 18 out of 20 patients (Probands 6 and 14 excluded) with istoantibodies to αIIbβ3. The mutations were evenly distributed between ITGB3 and ITGA2B.
Gene: ITGB3
Variant: NM_000212.2:c.166-2A>G (IVS2-2A>G)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA291224483
gnomAD: N/A
21
Case21: Male, age not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Mild bleeding tendency (See Table 1 for Clinical and Biological data for selected genotyped patients
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: N/A
PlateletAggregation: Minimal information, not specific enough
CasePreviousTesting: Sanger sequencing identified disease-causing variants in 18 out of 20 patients (Probands 6 and 14 excluded) with istoantibodies to αIIbβ3. The mutations were evenly distributed between ITGB3 and ITGA2B.
Gene: ITGA2B
Variant: NM_000419.3:c.2421G>A (p.Trp807Ter)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA399796017
gnomAD: N/A
Case13: Patient BM, gender and age are not specified, all of these patients are Caucasian (either Italian or European)
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: All patients presented with a current or past history of bleeding and a bleeding time >15 min. Bleeding symptoms were evaluated by examining available hospital records. Mild bleeders were defined as those who bled only after trauma or surgery or had minor symptoms, such as epistaxis. Moderate/severe bleeders were defined as those with a history of spontaneous or life threatening hemorrhages, such as gastrointestinal bleeding, or who had repeated episodes requiring platelet transfusions
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: See Table 1
PlateletAggregation: See Table 1
CasePreviousTesting: To identify point mutations responsible for GT, we have sequenced all coding regions of Ib and 3 genes and intron/exon boundaries.
Gene: ITGA2B
Variant: NM_000419.3:c.2113T>C (p.Cys705Arg)
LegacyVariant: (p.Arg674Arg)
ClinVar: N/A
CAiD: CA8602816
gnomAD: MAF in the European (non-Finnish) population is 0.000008792 https://gnomad.broadinstitute.org/variant/17-42455140-A-G
Case19: Patient SM, gender and age are not specified, all of these patients are Caucasian (either Italian or European)
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: All patients presented with a current or past history of bleeding and a bleeding time >15 min. Bleeding symptoms were evaluated by examining available hospital records. Mild bleeders were defined as those who bled only after trauma or surgery or had minor symptoms, such as epistaxis. Moderate/severe bleeders were defined as those with a history of spontaneous or life threatening hemorrhages, such as gastrointestinal bleeding, or who had repeated episodes requiring platelet transfusions
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: See Table 1
PlateletAggregation: See Table 1
CasePreviousTesting: To identify point mutations responsible for GT, we have sequenced all coding regions of Ib and 3 genes and intron/exon boundaries.
Gene: ITGB3
Variant: NM_000212.2:c.355C>T and NM_000212.2:c.1801T>C
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA8622924 and CA8623361
gnomAD: MAF in the South Asian population is 0.0001307 https://gnomad.broadinstitute.org/variant/17-45360909-C-T and MAF in the European non Finnish pop is 0.00001758 https://gnomad.broadinstitute.org/variant/17-45376784-T-C
Case25: Patient CD, gender and age are not specified, all of these patients are Caucasian (either Italian or European)
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: All patients presented with a current or past history of bleeding and a bleeding time >15 min. Bleeding symptoms were evaluated by examining available hospital records. Mild bleeders were defined as those who bled only after trauma or surgery or had minor symptoms, such as epistaxis. Moderate/severe bleeders were defined as those with a history of spontaneous or life threatening hemorrhages, such as gastrointestinal bleeding, or who had repeated episodes requiring platelet transfusions
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: See Table 1
PlateletAggregation: See Table 1
CasePreviousTesting: To identify point mutations responsible for GT, we have sequenced all coding regions of Ib and 3 genes and intron/exon boundaries.
Gene: ITGA2B
Variant: NM_000212.2:c.1801T>C
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA8623361
gnomAD: MAF in the European non Finnish population is 0.00001758 https://gnomad.broadinstitute.org/variant/17-45376784-T-C
Case5: Patient DRP, gender and age are not specified, all of these patients are Caucasian (either Italian or European)
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: All patients presented with a current or past history of bleeding and a bleeding time >15 min. Bleeding symptoms were evaluated by examining available hospital records. Mild bleeders were defined as those who bled only after trauma or surgery or had minor symptoms, such as epistaxis. Moderate/severe bleeders were defined as those with a history of spontaneous or life threatening hemorrhages, such as gastrointestinal bleeding, or who had repeated episodes requiring platelet transfusions
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: See Table 1
PlateletAggregation: See Table 1
CasePreviousTesting: To identify point mutations responsible for GT, we have sequenced all coding regions of Ib and 3 genes and intron/exon boundaries.
Gene: ITGA2B
Variant: This variant has a typo in the paper the real variant is this: NM_000419.3:c.800 G>A and NM_000419.3:c.2930del
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA399805147 and CA915940386
gnomAD: N/A
GT-02
Case2: (GT-02) Male, age or ethnicity not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Probands 1-9 are related to GT-02 see Table 1
ParentalGenotype: Parents may be included in Table 1; however, not specified so no information
CasePresentingHPOs: N/A
CaseHPOFreeText: Bleeding severity: N/A for proband 2 (See Supplemental Table 3)
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry performed and CD41 and CD61 not observed (See Supplementary Table 3)
PlateletAggregation: Minimal information (See Supplementary Table 3)
CasePreviousTesting: DNA was extracted from peripheral blood samples from patients and family members, and Sanger sequencing and PCR was performed. Samples were processed for targeted sequencing using the available SHGp heme panel of 393 genes.
Gene: ITGA2B
Variant: NM_000419.3:c.1210+5G>A
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA915940251
gnomAD: N/A
GT-43
Case33: (GT-43) Male, age or ethnicity not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Proband GT43-GT44 are related
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Bleeding severity: Severe (See Supplementary Table 3)
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry performed and CD41 and CD61 were observed (See Supplementary Table 3)
PlateletAggregation: Minimal information (See Supplementary Table 3)
CasePreviousTesting: DNA was extracted from peripheral blood samples from patients and family members, and Sanger sequencing and PCR was performed. Samples were processed for targeted sequencing using the available SHGp heme panel of 393 genes.
Gene: ITGA2B
Variant: NM_000419.3:c.1620G>C (p.Gln540His) and NM_000419.3:c.1616T>G (p.Leu539Arg)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA399802407 and CA399802416
gnomAD: N/A
GT-12
Case10: (GT-12) Male, age or ethnicity not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Probands GT11 -GT14 are related to GT-12 see Table 1
ParentalGenotype: Parents may be included in Table 1; however, not specified so no information
CasePresentingHPOs: N/A
CaseHPOFreeText: Bleeding severity: not available (See Supplemental Table 3)
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry performed and CD41 and CD61 were observed (See Supplementary Table 3)
PlateletAggregation: Minimal information (See Supplementary Table 3)
CasePreviousTesting: DNA was extracted from peripheral blood samples from patients and family members, and Sanger sequencing and PCR was performed. Samples were processed for targeted sequencing using the available SHGp heme panel of 393 genes.
Gene: ITGB3
Variant: NM_000212.2:c.727G>C (p.Asp243His)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA400023600
gnomAD: N/A
GT-62
Case50: (GT-62) Female, age or ethnicity not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Bleeding severity: Severe (See Supplementary Table 3)
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry performed and CD41 and CD61 were observed (See Supplementary Table 3)
PlateletAggregation: Minimal information (See Supplementary Table 3)
CasePreviousTesting: DNA was extracted from peripheral blood samples from patients and family members, and Sanger sequencing and PCR was performed. Samples were processed for targeted sequencing using the available SHGp heme panel of 393 genes.
Gene: ITGA2B and ITGB3
Variant: NM_000419.3:c.985G>T (p.Val329Phe) and NM_000212.2:c.1265G>A (p.Ser422Asn)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA399804719 (p.Val329Phe) and CA291225740 (p.Ser422Asn)
gnomAD: N/A
GT-20b
Case18: (GT-20) Female, age or ethnicity not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Probands GT15-GT19 are related to GT-20 see Table 1
ParentalGenotype: Parents may be included in Table 1; however, not specified so no information
CasePresentingHPOs: N/A
CaseHPOFreeText: Bleeding severity: Information not available on bleeding phenotype/severity (See Supplementary Table 3)
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry performed and CD41 and CD61 were observed (See Supplementary Table 3)
PlateletAggregation: Minimal information (See Supplementary Table 3)
CasePreviousTesting: DNA was extracted from peripheral blood samples from patients and family members, and Sanger sequencing and PCR was performed. Samples were processed for targeted sequencing using the available SHGp heme panel of 393 genes.
Gene: ITGB3
Variant: NM_000212.2:c.727G>C (p.Asp243His)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA400023600
gnomAD: N/A
GT-19b
Case17: (GT-19) Male, carrier status, age or ethnicity not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Probands GT15-GT20 are related to GT-19 see Table 1
ParentalGenotype: Parents may be included in Table 1; however, not specified so no information
CasePresentingHPOs: N/A
CaseHPOFreeText: Bleeding severity: Information not available on bleeding phenotype/severity (See Supplementary Table 3)
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry performed and CD41 and CD61 were observed (See Supplementary Table 3)
PlateletAggregation: Minimal information (See Supplementary Table 3)
CasePreviousTesting: DNA was extracted from peripheral blood samples from patients and family members, and Sanger sequencing and PCR was performed. Samples were processed for targeted sequencing using the available SHGp heme panel of 393 genes.
Gene: ITGB3
Variant: NM_000212.2:c.727G>C (p.Asp243His)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA400023600
gnomAD: N/A
GT-72c
Case53: (GT-72) Gender, age, or ethnicity not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Bleeding severity: Information not given (See Supplementary Table 3)
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry performed and CD41 and CD61 were observed (See Supplementary Table 3)
PlateletAggregation: Minimal information (See Supplementary Table 3)
CasePreviousTesting: DNA was extracted from peripheral blood samples from patients and family members, and Sanger sequencing and PCR was performed. Samples were processed for targeted sequencing using the available SHGp heme panel of 393 genes.
Gene: ITGB3
Variant: NM_000212.2:c.662C>T (p.Thr221Met)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA8623017
gnomAD: MAF in European Finnish population 0.0001386 http://gnomad.broadinstitute.org/variant/17-45363673-C-T
GT-56
Case45: (GT-56) Female, age or ethnicity not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Bleeding severity: Moderate (See Supplementary Table 3)
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry performed and CD41 and CD61 were observed (See Supplementary Table 3)
PlateletAggregation: Minimal information (See Supplementary Table 3)
CasePreviousTesting: DNA was extracted from peripheral blood samples from patients and family members, and Sanger sequencing and PCR was performed. Samples were processed for targeted sequencing using the available SHGp heme panel of 393 genes.
Gene: ITGA2B
Variant: NM_000419.3:c.985G>T (p.Val329Phe)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA399804719
gnomAD: N/A
GT-16
Case14: (GT-16) Male, age or ethnicity not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Probands GT15-GT20 are related to GT-16 see Table 1
ParentalGenotype: Parents may be included in Table 1; however, not specified so no information
CasePresentingHPOs: N/A
CaseHPOFreeText: Bleeding severity: Severe (See Supplementary Table 3)
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry performed and CD41 and CD61 were observed (See Supplementary Table 3)
PlateletAggregation: Minimal information (See Supplementary Table 3)
CasePreviousTesting: DNA was extracted from peripheral blood samples from patients and family members, and Sanger sequencing and PCR was performed. Samples were processed for targeted sequencing using the available SHGp heme panel of 393 genes.
Gene: ITGB3
Variant: NM_000212.2:c.727G>C (p.Asp243His)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA400023600
gnomAD: N/A
GT-46b
Case36: (GT-46) Female, age or ethnicity not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Proband GT45-GT48 are related
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Bleeding severity: Information not available (See Supplementary Table 3)
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry performed and CD41 and CD61 were observed (See Supplementary Table 3)
PlateletAggregation: Minimal information (See Supplementary Table 3)
CasePreviousTesting: DNA was extracted from peripheral blood samples from patients and family members, and Sanger sequencing and PCR was performed. Samples were processed for targeted sequencing using the available SHGp heme panel of 393 genes.
Gene: ITGA2B
Variant: NM_000419.3:c.1879-2A>G
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA290949031
gnomAD: N/A
GT-52
Case42: (GT-52) Male, age or ethnicity not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Proband GT49-GT52 are related
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Bleeding severity: Moderate (See Supplementary Table 3)
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry performed and CD41 and CD61 were observed (See Supplementary Table 3)
PlateletAggregation: Minimal information (See Supplementary Table 3)
CasePreviousTesting: DNA was extracted from peripheral blood samples from patients and family members, and Sanger sequencing and PCR was performed. Samples were processed for targeted sequencing using the available SHGp heme panel of 393 genes.
Gene: ITGB3
Variant: NM_000212.2:c.2113del (p.Leu705Cysfs*4)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA8623443
gnomAD: MAF in East Asian population: 0.0001087 http://gnomad.broadinstitute.org/variant/17-45380183-TC-T
Characterisation of patients with Glanzmann thrombasthenia and identification of 17 novel mutations
PMID: 25373348
Genes: ITGA2B and ITGB3
HGNC: 6138 and 6156
GT7
Case7: GT-7, German and Jugoslav, gender or age not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: Mother: c.1126-72C>T heterozygous and Father: c.1126-72C>T heterozygous
CasePresentingHPOs: N/A
CaseHPOFreeText: Bleeding symptoms are severe and bleeding time is pathogenic (See Table 1) Abstract mentions patients' bleeding signs include epistaxis, mucocutaneous bleeding, haematomas, petechiae, GI bleeding and menorrhagia; however, not specified per proband.
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry was performed and expression is stated as pathogenic in Table 1
PlateletAggregation: Platelet aggregation was performed and is stated as pathogenic in Table 1; however, no specific agonists mentioned
CasePreviousTesting: All exons of ITGA2B and ITGB3 genes as well as surrounding intron regions were amplified by PCR using intronic primers as pre-viously described (12). The purified DNA fragments were then di-rectly sequenced.
Gene: ITGB3
Variant: NM_000212.2:c.1030dup and an Unregistered Variant (c.1126_72C>T) (Does not show up in CAR)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA658820875 and N/A
gnomAD: N/A
GT1
Case1: GT-1, Turkish, gender or age not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: Mother: c.2374delG heterozygous and Father: c.2374delG heterozygous
CasePresentingHPOs: N/A
CaseHPOFreeText: Bleeding symptoms are severe and bleeding time is pathogenic (See Table 1) Abstract mentions patients' bleeding signs include epistaxis, mucocutaneous bleeding, haematomas, petechiae, GI bleeding and menorrhagia; however, not specified per proband.
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry was performed and expression is stated as pathogenic in Table 1
PlateletAggregation: Platelet aggregation was performed and is stated as pathogenic in Table 1; however, no specific agonists mentioned
CasePreviousTesting: All exons of ITGA2B and ITGB3 genes as well as surrounding intron regions were amplified by PCR using intronic primers as pre-viously described (12). The purified DNA fragments were then di-rectly sequenced.
Gene: ITGA2B
Variant: NM_000419.3:c.2374del
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA915940267
gnomAD: N/A
GT12
Case12: GT-12, Caucasian, gender or age not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: Mother: c.2326_2331dupGAGGCC and Father: deceased
CasePresentingHPOs: N/A
CaseHPOFreeText: Bleeding symptoms are severe and bleeding time is pathogenic (See Table 1) Abstract mentions patients' bleeding signs include epistaxis, mucocutaneous bleeding, haematomas, petechiae, GI bleeding and menorrhagia; however, not specified per proband.
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry was performed and expression is stated as pathogenic in Table 1
PlateletAggregation: Platelet aggregation was performed and is stated as pathogenic in Table 1; however, no specific agonists mentioned
CasePreviousTesting: All exons of ITGA2B and ITGB3 genes as well as surrounding intron regions were amplified by PCR using intronic primers as pre-viously described (12). The purified DNA fragments were then di-rectly sequenced.
Gene: ITGA2B
Variant: NM_000419.3:c.957T>A and NM_000419.3:c.2326_2331dupGAGGCC
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA399804780 and CA915940324
gnomAD: N/A
GT5
Case5: GT-5, Caucasian, gender or age not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Bleeding symptoms are severe and bleeding time is pathogenic (See Table 1) Abstract mentions patients' bleeding signs include epistaxis, mucocutaneous bleeding, haematomas, petechiae, GI bleeding and menorrhagia; however, not specified per proband.
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry was performed and expression is stated as pathogenic in Table 1
PlateletAggregation: Platelet aggregation was performed and is stated as pathogenic in Table 1; however, no specific agonists mentioned
CasePreviousTesting: All exons of ITGA2B and ITGB3 genes as well as surrounding intron regions were amplified by PCR using intronic primers as pre-viously described (12). The purified DNA fragments were then di-rectly sequenced.
Gene: ITGA2B
Variant: NM_000419.3:c.408G>C and NM_000419.3:c.1787T>C
LegacyVariant: N/A
ClinVar: 2900
CAiD: CA500624244 and CA115846
gnomAD: MAF in the European (non-Finnish) population 0.00006484 http://gnomad.broadinstitute.org/variant/17-42463192-C-G and MAF in European (non-Finnish) population is 0.0001325 http://gnomad.broadinstitute.org/variant/17-42457148-A-G
GT6
Case6: GT-6, Croats, gender or age not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Bleeding symptoms are severe and bleeding time is pathogenic (See Table 1) Abstract mentions patients' bleeding signs include epistaxis, mucocutaneous bleeding, haematomas, petechiae, GI bleeding and menorrhagia; however, not specified per proband.
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry was performed and expression is stated as pathogenic in Table 1
PlateletAggregation: Platelet aggregation was performed and is stated as pathogenic in Table 1; however, no specific agonists mentioned
CasePreviousTesting: All exons of ITGA2B and ITGB3 genes as well as surrounding intron regions were amplified by PCR using intronic primers as pre-viously described (12). The purified DNA fragments were then di-rectly sequenced.
Gene: ITGB3
Variant: NM_000212.2:c.166–2A>G
LegacyVariant: N/A
ClinVar: N/A
CAiD: Unregistered Variant
gnomAD: N/A
cases 3
Case3: Female, 32 years old, Japanese
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A per Table 1
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Prolonged ivy bleeding times, bleeding diathesis since early childhood
CaseNotHPOs: N/A
CaseNotHPOFreeText: N/A
ProteinExpression: Surface expression of αIIbβ3 was analyzed by flow cytometry which was 10-20% of normal
PlateletAggregation: Aggregation in response to ADP, epinephrine and collagen was absent but normal in response to ristocetin
CasePreviousTesting: Nucleotide sequences of the αIIb and β3 coding regions were determined by direct sequencing of RT-PCR products from platelet RNA.
Gene: ITGA2B
Variant: NM_000419.3:c.2333A>C (p.Gln778Pro)
LegacyVariant: (p.Gln747Pro)
ClinVar: 225393
CAiD: CA290947544
gnomAD: MAF in the East Asian population is 0.0001087 https://gnomad.broadinstitute.org/variant/17-42453691-T-G
2
Case2: Female, 67 years old, Japanese
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A per Table 1
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Prolonged ivy bleeding times, bleeding diathesis since early childhood
CaseNotHPOs: N/A
CaseNotHPOFreeText: N/A
ProteinExpression: Surface expression of αIIbβ3 was analyzed by flow cytometry which was < 5% of normal
PlateletAggregation: Aggregation in response to ADP, epinephrine and collagen was absent but normal in response to ristocetin
CasePreviousTesting: Nucleotide sequences of the αIIb and β3 coding regions were determined by direct sequencing of RT-PCR products from platelet RNA.
Gene: ITGA2B
Variant: NM_000419.3:c.1063G>A (p.Glu355Lys)
LegacyVariant: (p.Glu324Lys)
ClinVar: 2899
CAiD: CA115844
gnomAD: MAF in the European (non-Finnish) population is 0.00001978 https://gnomad.broadinstitute.org/variant/17-42461008-C-T
Acquired Glanzmann’s Thrombasthenia Associatedwith Hodgkin’s Lymphoma
PMID: 9576300
Gene: ITGA2B and/or ITGB3
HGNC: 6138 and 6156
InsufficientEvidence: This mentions a Glanzmann thrombasthenia patient; however, no variants in ITGA2B and/or ITGB3 are mentioned in this patient; therefore, we have insufficient evidence
Bone marrow transplantation in severe Glanzmann's thrombasthenia with antiplatelet alloimmunization
PMID: 10673707
Gene: ITGA2B and/or ITGB3
HGNC: 6138 and 6156
InsufficientEvidence: This article mentions a Glanzmann thrombasthenia patient; however, there is no mention of variants in ITGA2B and/or ITGB3; therefore, there is insufficient evidence
13
Case12: No assertion made on age, gender or ethnicity
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: The identified variants were heterozygote in parents.
CasePresentingHPOs: N/A
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: N/A
CasePreviousTesting: Genetic study of patient was conducted using direct sequencing of entire coding and exon-intron boundaries of the ITGA2B and ITGB3 genes.
Gene: ITGA2B
Variant: NM_000419.3:c.1555C>T (p.Gln519Ter)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA399802559
gnomAD: N/A
11
Case9: No assertion made on age, gender or ethnicity
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Table 1 mentions family history available for this proband.
ParentalGenotype: The identified variants were heterozygote in parents.
CasePresentingHPOs: HP:0030402, HP:0005542
(Abnormal platelet aggregation, prolonged clotting time)
CaseHPOFreeText: Bleeding time greater than 20 minutes and hematocrit of 38.1%.
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: N/A
CasePreviousTesting: Genetic study of patient was conducted using direct sequencing of entire coding and exon-intron boundaries of the ITGA2B and ITGB3 genes.
Gene: ITGA2B
Variant: NM_000419.3:c.286T>A (p.Cys96Ser)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA8603504
gnomAD: N/A
Recently, we reported the case of an 8-year-old boy with a variant form of GT
Case1: Male, 8 years old, ethnicity not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Sister had no bleeding tendency (all the information given in the previously published article cited in this annotation)
ParentalGenotype: Parental mutations shown in Fig. 3 A and B
CasePresentingHPOs: HP:0000421
(Epistaxis)
CaseHPOFreeText: Tonsilar bleeding
CaseNotHPOs: HP:0000979
(Purpura)
CaseNotHPOFreeText: N/A
ProteinExpression: Western blotting indicated that the proband's platelets contained no normal GPIIb, but showed a trace amount of an abnormal GPIIb (-6% with respect to the GPIIb level in a normal individual).
PlateletAggregation: Decreased aggregation in response to ADP, collagen and epinephrine but a normal ristocetin-induced aggregation (this information was obtained from the previously published article cited at in this annotation)
CasePreviousTesting: Genomic DNA fragments containing exons of the GPIIb gene were amplified by PCR
Gene: ITGA2B
Variant: NM_000419.3:c.1750C>T (p.Arg584Ter) and NM_000419.3:c.2602-3C>G
LegacyVariant: N/A
ClinVar: 2892 and 2893
CAiD: CA115834 and CA10575472
gnomAD: MAF in the East Asian population is 0.0002176 https://gnomad.broadinstitute.org/variant/17-42457372-G-A and MAF in the East Asian population is 0.00006523 https://gnomad.broadinstitute.org/variant/17-42453087-G-C
PreviouslyPublished: PMID: 1891767
GT72
Case17: (GT72) Gender, age and ethnicity not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Inclusion criteria were patients with a history of mucocutaneous bleeding (Figure 1 shows a bar diagram of all the clinical manifestations in the patient cohort in this article)
CaseNotHPOs: N/A
ProteinExpression: Platelet receptor alphaIIbbeta3 protein was analyzed by flow cytometry followed by western blotting
Platelet Aggregation: Inclusion criteria for probands was absent or reduced platelet aggregation with agonists such as ADP, adrenaline, arachidonic acid, and collagen (no mention of ristocetin here-- this article does not tell us the specific aggregation for each proband in the paper)
CasePreviousTesting: For mutation analysis of ITGA2B and ITGB3 genes, DNA extraction from whole blood, PCR, heteroduplex PCR followed by CSGE and DNA sequencing were performed. All 30 exons of ITGA2B and 15 exons of ITGB3, as well as their promoters, were analyzed by CSGE following heteroduplex PCR.
Gene: ITGB3
Variant: NM_000212.2:c.1641C>G (p.Cys547Trp)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA400030171
gnomAD N/A
58
Case9: (GT58), Gender, age and ethnicity not reported
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Inclusion criteria were patients with a history of mucocutaneous bleeding (Figure 1 shows a bar diagram of all the clinical manifestations in the patient cohort in this article)
CaseNotHPOs: N/A
ProteinExpression: Platelet receptor alphaIIbbeta3 protein was analyzed by flow cytometry followed by western blotting
Platelet Aggregation: Inclusion criteria for probands was absent or reduced platelet aggregation with agonists such as ADP, adrenaline, arachidonic acid, and collagen (no mention of ristocetin here-- this article does not tell us the specific aggregation for each proband in the paper)
CasePreviousTesting: For mutation analysis of ITGA2B and ITGB3 genes, DNA extraction from whole blood, PCR, heteroduplex PCR followed by CSGE and DNA sequencing were performed. All 30 exons of ITGA2B and 15 exons of ITGB3, as well as their promoters, were analyzed by CSGE following heteroduplex PCR.
Gene: ITGA2B
Variant: NM_000419.3:c.1028T>C (p.Leu343Pro)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA399804619 (p.Leu343Pro)
gnomAD N/A
69
Case4: (GT69), Gender, age and ethnicity not reported
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Inclusion criteria were patients with a history of mucocutaneous bleeding (Figure 1 shows a bar diagram of all the clinical manifestations in the patient cohort in this article)
CaseNotHPOs: N/A
ProteinExpression: Platelet receptor alphaIIbbeta3 protein was analyzed by flow cytometry followed by western blotting
Platelet Aggregation: Inclusion criteria for probands was absent or reduced platelet aggregation with agonists such as ADP, adrenaline, arachidonic acid, and collagen (no mention of ristocetin here-- this article does not tell us the specific aggregation for each proband in the paper)
CasePreviousTesting: For mutation analysis of ITGA2B and ITGB3 genes, DNA extraction from whole blood, PCR, heteroduplex PCR followed by CSGE and DNA sequencing were performed. All 30 exons of ITGA2B and 15 exons of ITGB3, as well as their promoters, were analyzed by CSGE following heteroduplex PCR.
Gene: ITGA2B
Variant: NC_000017.11:g.44393555C>T (g.951G>A promoter variant) and NM_000419.3:c.1028T>C (p.Leu343Pro)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA913189168 (promoter variant) and CA399804619 (p.Leu343Pro)
gnomAD N/A
67
Case23: (GT67) Gender, age and ethnicity are not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Inclusion criteria were patients with a history of mucocutaneous bleeding (Figure 1 shows a bar diagram of all the clinical manifestations in the patient cohort in this article)
CaseNotHPOs: N/A
ProteinExpression: Platelet receptor alphaIIbbeta3 protein was analyzed by flow cytometry followed by western blotting
Platelet Aggregation: Inclusion criteria for probands was absent or reduced platelet aggregation with agonists such as ADP, adrenaline, arachidonic acid, and collagen (no mention of ristocetin here-- this article does not tell us the specific aggregation for each proband in the paper)
CasePreviousTesting: For mutation analysis of ITGA2B and ITGB3 genes, DNA extraction from whole blood, PCR, heteroduplex PCR followed by CSGE and DNA sequencing were performed. All 30 exons of ITGA2B and 15 exons of ITGB3, as well as their promoters, were analyzed by CSGE following heteroduplex PCR.
Gene:
Variant: NM_000419.3:c.1919_1920del
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA915940685
gnomAD N/A
GT31
Case30: (GT31) Gender, age and ethnicity are not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Inclusion criteria were patients with a history of mucocutaneous bleeding (Figure 1 shows a bar diagram of all the clinical manifestations in the patient cohort in this article)
CaseNotHPOs: N/A
ProteinExpression: Platelet receptor alphaIIbbeta3 protein was analyzed by flow cytometry followed by western blotting
Platelet Aggregation: Inclusion criteria for probands was absent or reduced platelet aggregation with agonists such as ADP, adrenaline, arachidonic acid, and collagen (no mention of ristocetin here-- this article does not tell us the specific aggregation for each proband in the paper)
CasePreviousTesting: For mutation analysis of ITGA2B and ITGB3 genes, DNA extraction from whole blood, PCR, heteroduplex PCR followed by CSGE and DNA sequencing were performed. All 30 exons of ITGA2B and 15 exons of ITGB3, as well as their promoters, were analyzed by CSGE following heteroduplex PCR.
Gene: ITGB3
Variant: NM_000212.2:c.887_901del
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA626379036
gnomAD MAF in the South Asian population is 0.00003266 https://gnomad.broadinstitute.org/variant/17-45364541-AATGACGGGCAGTGTC-A
GT12
Case6: (GT12) Gender, age and ethnicity not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Inclusion criteria were patients with a history of mucocutaneous bleeding (Figure 1 shows a bar diagram of all the clinical manifestations in the patient cohort in this article)
CaseNotHPOs: N/A
ProteinExpression: Platelet receptor alphaIIbbeta3 protein was analyzed by flow cytometry followed by western blotting
Platelet Aggregation: Inclusion criteria for probands was absent or reduced platelet aggregation with agonists such as ADP, adrenaline, arachidonic acid, and collagen (no mention of ristocetin here-- this article does not tell us the specific aggregation for each proband in the paper)
CasePreviousTesting: For mutation analysis of ITGA2B and ITGB3 genes, DNA extraction from whole blood, PCR, heteroduplex PCR followed by CSGE and DNA sequencing were performed. All 30 exons of ITGA2B and 15 exons of ITGB3, as well as their promoters, were analyzed by CSGE following heteroduplex PCR.
Gene: ITGA2B
Variant: NM_000419.3:c.641T>C (p.Leu214Pro)
LegacyVariant: N/A
ClinVar: 2901
CAiD: CA115848
gnomAD N/A
GT20
Case26: (GT20) Gender, age and ethnicity are not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Brother is homozygous for the mutation and the second brother is heterozygous
ParentalGenotype: Mother and father are heterozygous for the mutation
CasePresentingHPOs: N/A
CaseHPOFreeText: Inclusion criteria were patients with a history of mucocutaneous bleeding (Figure 1 shows a bar diagram of all the clinical manifestations in the patient cohort in this article)
CaseNotHPOs: N/A
ProteinExpression: Platelet receptor alphaIIbbeta3 protein was analyzed by flow cytometry followed by western blotting
Platelet Aggregation: Inclusion criteria for probands was absent or reduced platelet aggregation with agonists such as ADP, adrenaline, arachidonic acid, and collagen (no mention of ristocetin here-- this article does not tell us the specific aggregation for each proband in the paper)
CasePreviousTesting: For mutation analysis of ITGA2B and ITGB3 genes, DNA extraction from whole blood, PCR, heteroduplex PCR followed by CSGE and DNA sequencing were performed. All 30 exons of ITGA2B and 15 exons of ITGB3, as well as their promoters, were analyzed by CSGE following heteroduplex PCR.
Gene: ITGA2B
Variant: NM_000419.3:c.1424_1427dup
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA8603099
gnomAD MAF in the South Asian population is 0.0001960 https://gnomad.broadinstitute.org/variant/17-42457979-C-CACCT
PMA induces platelet activation of specificantigens (CD62/CD63) in GpIIb-IIIa deficientplatelets from Glanzmann's thrombasthenia
PMID: 16793673
Gene: ITGA2B and/or ITGB3
HGNC: 6138 and 6156
InsufficientEvidence: This article mentions individuals with Glanzmann thrombasthenia; however, the probands don't have variants in ITGAB and/or ITGB3 mentioned
F6.II.4
Case#: Female, 30 (age of diagnosis)
Disease: Glazmann's Thrombasthenia Like Syndrome
CasePresentingHPOs: HP:0000225, HP:0000978, HP:0006298
Gingival bleeding, Bruising susceptibility, Prolonged bleeding after dental extraction
CaseNotHPOs: NA
FamilyInfo: Pedigree figure 1. Father and two daughters affected.
PlateletAgreggation: Family member studied: Decreased PLT aggregation and diminished ATP release with the agonists ADP, collagen, and AA, with normal ristocetin agglutination
GenotypingMethod: Genetic analysis of ITGA2B and ITGB3 genes was performed by next-generation sequencing (NGS). Sanger sequencing used to confirm variants.
ClinVar: Not Found
CAID: CA400034251
Variant: NM_000212.3:c.2236A>C (ITGB3)
GnomAD: Not Found
ExpressionEvidence: Level of expression on PLT surface determined by FCM: CD41a (GPIIb/IIIa) 38%, CD61 (GPIIIa) 41%, and CD42b (GPIb) 85%. Healthy control value range: CD41a (GPIIb/IIIa) (85-115%); CD61 (GPIIIa) (80-120%); CD42b (GPIb) (70-130%).
F8.I.2
Case#: Female, 75 (age when studied in 1975)
Disease: Glazmann's Thrombasthenia Like Syndrome
CasePresentingHPOs: HP:0001873, HP:0006298
Thrombocytopenia, Prolonged bleeding after dental extraction
CaseNotHPOs: NA
FamilyInfo: Pedigree figure 1. Several affected family members.
PlateletAgreggation:Daughter studied: Decreased PLT aggregation and diminished ATP release with the agonists ADP, collagen, and AA, with normal ristocetin agglutination
GenotypingMethod: Genetic analysis of ITGA2B and ITGB3 genes was performed by next-generation sequencing (NGS). Sanger sequencing used to confirm variants.
From Daughter
ClinVar: Not Found
CAID: CA400034393
Variant: NM_000212.3:c.2278C>T (ITGB3)
GnomAD: Not Found
ExpressionEvidence: Level of expression on PLT surface determined by FCM: CD41a (GPIIb/IIIa) 52%, CD61 (GPIIIa) 77%, and CD42b (GPIb) 129%. Healthy control value range: CD41a (GPIIb/IIIa) (85-115%); CD61 (GPIIIa) (80-120%); CD42b (GPIb) (70-130%).
26CabGT-26
Case26: (Proband CabGT-26), Female, age not specified, North American
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
ProteinExpression: Flow cytometry showed 12% of both GPIIIa and GPIIb surface expression
Platelet Aggregation: N/A
CasePreviousTesting: Promoter and exon sequences of aIIb and b3 genes were amplified and directly sequence
Gene: ITGB3
Variant: "c.1143C>A" is incorrect in the CAR- so will need a senior biocurator to ask-- and NM_000212.2:c.1260G>A
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA8623200
gnomAD MA in the African population is 0.00004005 https://gnomad.broadinstitute.org/variant/17-45368454-G-A
25CabGT-25
Case25: (Proband CabGT-25), Gender, age and ethnicity not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
ProteinExpression: N/A
Platelet Aggregation: N/A
CasePreviousTesting: Promoter and exon sequences of aIIb and b3 genes were amplified and directly sequence
Gene: ITGB3
Variant: c.112513_112516del (Not showing up in the CAR) and they could not determine the second variant
LegacyVariant: N/A
ClinVar: N/A
CAiD: N/A
gnomAD N/A
6CabGT-6
Case6: (Proband, CabGT-6), Male, age not specified, Caucasian
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
ProteinExpression: Flow cytometry showed <5% surface expression of GPIIIa and GPIIb
Platelet Aggregation: N/A
CasePreviousTesting: Promoter and exon sequences of aIIb and b3 genes were amplified and directly sequence
Gene: ITGA2B
Variant: NM_000419.3:c.1413C>G (p.Tyr471Ter) and NM_000419.3:c.2800G>T (p.Val934Phe)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA290950376 (p.Tyr471Ter) and CA290946484 (p.Val934Phe)
gnomAD MAF in the European (non-Finnish) population is 0.000008791 https://gnomad.broadinstitute.org/variant/17-42457994-G-C
15CabGT-15
Case15: (Proband CabGT-15)
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
ProteinExpression: N/A
Platelet Aggregation: N/A
CasePreviousTesting: Promoter and exon sequences of aIIb and b3 genes were amplified and directly sequence
Gene: ITGA2B
Variant: NM_000419.3:c.2800G>T (p.Val934Phe)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA290946484
gnomAD N/A
Identification of Clinicopathological Spectrum, Platelet Glycoprotein IIb/IIIa complex and Platelet Antibodies in Egyptian Children with Glanzmann’s Thrombasthenia
PMID: 28161816
Gene: ITGA2B and ITGB3
HGNC 6138 and 6156
NoEvidence: The aim of this article's study was to evaluate the clinicopathological spectrum of this syndrome and to study the platelet glycoprotein IIb/IIIa complex and platelet antibodies by flow cytometry in a cohort of children with GT in a tertiary care center in Upper Egypt. There is no mention of individual probands with variants in ITGA2B or ITGB3
The proband
Case#: Female, 29 yo
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Figure 2 Father and brother, unaffected carriers. Mother, unaffected negative. Sister, affected: Epistaxis, Cutaneous bleeding, Menorrhagia, Hematuria, Postpartum bleeding, Other bleeding symptoms (Ruptured corpus luteum bleeding)
ParentalGenotype: Father carrier, unaffected. Mother negative, unaffected.
CasePresentingHPOs: HP:0000421, Epistaxis. HP:0000132, Menorrhagia. HP:0000790, Hematuria. HP:0003540, Impaired platelet aggregation. HP:0001892, Abnormal bleeding. HP:0003010, Prolonged bleeding time. HP:0034263, Abnormal vaginal bleeding.
PlateletAggregation: Normal platelet morphology, count 159 (125-350), MPV 10.6 fL (6.5-12.0), and PDW 12% (19-17). Decreased platelet aggregation: ADP 29.7& (30-70), Collagen 14.5% (30-70), AA 14.5% (40-80). Normal response to ristocetin, 88.7% (40-80). Thromboelastography (TEG) examination: hypocoagulable.
CaseHPOFreeText: Cutaneous bleeding, Postpartum bleeding, Blood accumulation in the uterine cavity, Bleeding in ovaries, Vaginal bleeding during pregnancy,
CaseNotHPOs: HP:0011875, Abnormal platelet morphology. HP:0011873, Abnormal platelet count.
ProteinExpression: Protein structure model: destroyed local beta folding structure and molecular changes to structure of GPII beta helical region reduces the expression of GP IIb/IIIa. Proband refused further testing (flow cytometry).
Gene: ITGA2B
Variant: c.2822G>A, p. (Trp941Ter)
HGVS: NM_000419.5:c.2822G>A
ClinVar: absent
CAID: CA399791662
gnomAD: 0.00 https://gnomad.broadinstitute.org/variant/chr17-44375017-C-T?dataset=gnomad_r4
An Exon 28 Mutation Resulting in Alternative Splicing of the Glycoprotein IIb Transcript and Glanzmann's Thrombasthenia
PMID: 8111043
Gene: ITGA2B
HGNC: 6138
F5P1
Case1: Patient F1P1, Male, 11 years old, Tunisian
DiseaseAssertion: Glanzmann thrombasthenia type I
FamilyInfo: Table 1 states there is a family history of bleeding (we have minimal to no specific info on this)
ParentalGenotype: Mother and father are heterozygous for mutation
CasePresentingHPOs:
CaseHPOFreeText:
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: N/A
PlateletAggregation: GT diagnosis was based on absent platelet aggregation induced by ADP, collagen, and TRAP and subnormal or cyclical ristocetin induced agglutination
CasePreviousTesting: Using PCR they successfully amplified 20 DNA fragments for the ITGA2B gene (covering more than 48% of the ITGA2B gene constituting 30 exons) and15 DNA fragments of theITGB3gene (covering 15 exons)
Gene: ITGA2B
Variant: NM_000419.3:c.2702C>A (p.Ser901X) another variant this patient has is g.13944G>A --> I am noting this as Unknown Variant as I will need guidance curating this one
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA399792888
gnomAD: N/A
F2P1
Case2: Patient F2P1, Male, 34 years old, Tunisian
DiseaseAssertion: Glanzmann thrombasthenia type I
FamilyInfo: Table 1 states there is a family history of bleeding (we have minimal to no specific info on this)
ParentalGenotype: Mother and father are heterozygotes for the mutation here
CasePresentingHPOs, : HP:0000225, HP:0004406, HP:0000978,HP:0000790
(Gingival bleeding, recurrent epistaxis, easy bruising, hematuria)
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: N/A
PlateletAggregation: GT diagnosis was based on absent platelet aggregation induced by ADP, collagen, and TRAP and subnormal or cyclical ristocetin induced agglutination
CasePreviousTesting: Using PCR they successfully amplified 20 DNA fragments for the ITGA2B gene (covering more than 48% of the ITGA2B gene constituting 30 exons) and15 DNA fragments of theITGB3gene (covering 15 exons)
Gene: ITGA2B
Variant: NM_000419.3:c.2702C>A (p.Ser901X) another variant this patient has is g.13944G>A --> I am noting this as Unknown Variant as I will need guidance curating this one
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA399792888
gnomAD: N/A
A Glanzmann patient, LeM
Case2: Patient LeM
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs:
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Platelets lack surface αIIbβ3 expression
PlateletAggregation: N/A
CasePreviousTesting: N/A
Gene: ITGA2B
Variant: NM_000419.3:c.1366_1371del V(425)D(426)del and other other variant is stated as "Unknown" in the GT database. See GT database if questions arise, this is where I found the variants
LegacyVariant: N/A
ClinVar: 2898
CAiD: CA8603160
gnomAD: MAF in the European (non-Finnish) population is 0.000008791 http://reg.clinicalgenome.org/redmine/projects/registry/genboree_registry/allele?hgvsOrDescriptor=NM_000419.3%3Ac.1366_1371del
Patient MK
Case1: (Patient MK), Female, Pakistani, age not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: HP:0000967, HP:0000979
(Petechiae, purpura)
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOFreeText: N/A
ProteinExpression: Figure 1 shows protein expression by flow cytometry
PlateletAggregation: Absence of platelet aggregation in response to ADP, epinephrine, and collagen but a normal response to ristocetin
CasePreviousTesting: Genomic DNA containing all of the exons of the aIIb and b3 genes and 500 bp of DNA immediately upstream of each transcriptional start site was isolated from the patient and normal controls. Screening for mutations was performed by using single stranded conformation polymorphism analysis. PCR amplification was performed as well.
Gene: ITGB3
Variant: NM_000212.2:c.428T>G (p.Leu143Trp)
LegacyVariant: (p.Leu117Trp)
ClinVar: 13567
CAiD: CA123252
gnomAD: MAF in the Other population is 0.0001629 https://gnomad.broadinstitute.org/variant/17-45361875-T-G
A Leu117rTrp Mutation Within the RGD-Peptide Cross-Linking Region ofb3 Results in Glanzmann Thrombasthenia by PreventingaIIbb3 Export tothe Platelet Surface
PMID: 9376589
Gene: ITGB3
HGNC: 6156
A novel Pro126His β propeller mutation in integrin αIIb causes Glanzmann thrombasthenia by impairing progression of pro-αIIbβ3 from endoplasmic reticulum to Golgi
PMID: 18976939
Gene: ITGA2B
HGNC: 6138
Acquired Glanzmann’s thrombasthenia associated with Hairy cell leukaemia M. Kannan All India Institute of Medical Sciences, New Delhi, IndiaSearch for more papers by this author T. Chatterjee All India Institute of Medical Sciences, New Delhi, IndiaSearch for more papers by this author F. Ahmad All India Institute of Medical Sciences, New Delhi, IndiaSearch for more papers by this author R. Kumar DR BRA IRCH, New Delhi, IndiaSearch for more papers by this author V. P. Choudhry All India Institute of Medical Sciences, New Delhi, IndiaSearch for more papers by this author R. Saxena All India Institute of Medical Sciences, New Delhi, IndiaSearch for more papers by this author
PMID: 19594615
Gene: ITGA2B and/or ITGB3
HGNC: 6138 and 6156
InsufficientEvidence: This article mentions an individual with Hairy cell leukemia who presented with bleeding diathesis diagnosed to have acquired GT. There is no mention of ITGA2B/ ITGB3 variants in this individual.
Patient 1
Case1: Male, 14 years old, ethnicity not specified
DiseaseAssertion: Glanzmann thrombasthenia type 1
FamilyInfo: Sister is also heterozygous for this mutation but shows no bleeding symptoms
ParentalGenotype: Both parents are heterozygous for this mutation
Note: There is parental consanguinity
CasePresentingHPOs: HP:0000967, HP:0000225, HP:0000421
(Petechiae, ginigival bleeding, epistaxis)
CaseHPOFreeText: Haematoma,
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry analysis revealed 3.8 % platelet surface presentation of the αIIbβ3 complex for patient 1 and αIIb was not detected and β3 was significantly reduced in Western blots. Platelets from GT patients showed less than 5 % relative levels of the integrin subunit IIb and 5–9 % fibrinogen compared to controls.
PlateletAggregation: Article states agonist-induced platelet aggregation and binding of exogenous fibrinogen was pathological, too but does not mention specific agonists used or response to ristocetin
CasePreviousTesting: Molecular genetic analysis was done; however, no other information mentioned. In addition, the global proteome of highlight purified washed platelets was quantified by liquid chromatography-mass spectrometry and targeted MS-methods.
Gene: ITGA2B
Variant: NM_000419.3:c.175del
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA915940266
gnomAD: N/A
A novel amino acid substitution of integrin αIIb in Glanzmann thrombasthenia confirms that the N-terminal region of the receptor plays a role in maintaining β-propeller structure
PMID: 22394243
Gene: ITGA2B
HGNC: 6138
GT3
Case3: GT-3, Male, Catalan, age not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Affected sisters (GT-1) and (GT-2)
ParentalGenotype: N/A
CasePresentingHPOs: HP:0000421
(Epistaxis)
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Standard glycoprotein analysis confirmed the absence of αIIbβ3 in their platelets but while western blotting failed to reveal αIIb, residual β3 was at about 10% of usual levels and of normal migration (illustrated for GT1 in supplemental Figure 1).
PlateletAggregation: Platelets of all 3 patients failed to aggregate in response to a range of physiologic agonists but were agglutinated by ristocetin. Clot retraction was also defective.
CasePreviousTesting: Screened ITGA2B and ITGB3 genes of all three probands. Direct sequencing of all coding regions and splice sites of ITGA2B (30 exons) showed mutation.
Gene: ITGA2B
Variant: NM_000419.3:c.131G>T and NM_000419.3:c.1440-13_1440-1del
LegacyVariant: (p.Gly13Val)
ClinVar: N/A
CAiD: CA399806881 and CA913184940
gnomAD: N/A
GT2
Case2: GT-2, Female, Catalan, age not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Affected sister (GT-1) and brother (GT-3)
ParentalGenotype: N/A
CasePresentingHPOs: HP:0011891
(Post-partum hemorrhage)
CaseHPOFreeText: N/A
CaseNotHPOs:
CaseNotHPOsFreeText:
ProteinExpression: Standard glycoprotein analysis confirmed the absence of αIIbβ3 in their platelets but while western blotting failed to reveal αIIb, residual β3 was at about 10% of usual levels and of normal migration (illustrated for GT1 in supplemental Figure 1).
PlateletAggregation: Platelets of all 3 patients failed to aggregate in response to a range of physiologic agonists but were agglutinated by ristocetin. Clot retraction was also defective.
CasePreviousTesting: Screened ITGA2B and ITGB3 genes of all three probands. Direct sequencing of all coding regions and splice sites of ITGA2B (30 exons) showed mutation.
Gene: ITGA2B
Variant: NM_000419.3:c.131G>T and NM_000419.3:c.1440-13_1440-1del
LegacyVariant: (p.Gly13Val)
ClinVar: N/A
CAiD: CA399806881 and CA913184940
gnomAD: N/A
ANEWMETHODFORQUANTITATIVEANALYSISOFWHOLEBLOODPLATELETINTERACTIONWITHEXTRACELLULARMATRIXUNDERFLOWCONDITIONS
PMID: 9062952
Gene: ITGA2B and ITGB3
HGNC: 6138 and 6156
NoEvidence: This article does not mention Glanzmann thrombasthenia patients/probands with variants in ITGA2B and/or ITGB3; therefore, there is no evidence to report
LB
Case18: Patient LB, gender, age and ethnicity not specified
DiseaseAssertion: Glanzmann thrombasthenia type I
FamilyInfo: Child is patient LN, significant other is patient LH
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Bleeding symptoms were evaluated by examining available hospital records. Mild bleeders were defined as those who had minor symptoms, such as epistaxis or gum bleeds, and moderate bleeders suffered, in addition, from bleeding complications after surgery and trauma. Severe bleeders were defined as those with a history of spontaneous or life-threatening hemorrhages, such as gastrointestinal bleeding, or who had repeated episodes requiring platelet transfusion. See Table 1.
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: See Table 6
PlateletAggregation: See Table 6
CasePreviousTesting: To ascertain the mutations, we sequenced the coding regions of the αIIb and β3 subunits including the exon/intron boundaries
Gene: ITGA2B
Variant: NM_000419.3:c.3062T>C
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA399787956
gnomAD: N/A
TN
Case14: Patient TN, gender, age and ethnicity not specified
DiseaseAssertion: Glanzmann thrombasthenia type I
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Bleeding symptoms were evaluated by examining available hospital records. Mild bleeders were defined as those who had minor symptoms, such as epistaxis or gum bleeds, and moderate bleeders suffered, in addition, from bleeding complications after surgery and trauma. Severe bleeders were defined as those with a history of spontaneous or life-threatening hemorrhages, such as gastrointestinal bleeding, or who had repeated episodes requiring platelet transfusion. See Table 1.
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: See Table 6
PlateletAggregation: See Table 6
CasePreviousTesting: To ascertain the mutations, we sequenced the coding regions of the αIIb and β3 subunits including the exon/intron boundaries
Gene: ITGA2B
Variant: NM_000419.3:c.3060G>A
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA500261104
gnomAD: N/A
MU
Case3: Patient MU, gender age and ethnicity not specified
DiseaseAssertion: Glanzmann thrombasthenia type I
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Bleeding symptoms were evaluated by examining available hospital records. Mild bleeders were defined as those who had minor symptoms, such as epistaxis or gum bleeds, and moderate bleeders suffered, in addition, from bleeding complications after surgery and trauma. Severe bleeders were defined as those with a history of spontaneous or life-threatening hemorrhages, such as gastrointestinal bleeding, or who had repeated episodes requiring platelet transfusion. See Table 1.
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: See Table 6
PlateletAggregation: See Table 6
CasePreviousTesting: To ascertain the mutations, we sequenced the coding regions of the αIIb and β3 subunits including the exon/intron boundaries
Gene: ITGA2B
Variant: NM_000419.3:c.641T>C (p.Leu214Pro)
LegacyVariant: N/A
ClinVar: 2901
CAiD: CA115848
gnomAD: N/A
LH
Case17: Patient LH, gender, age and ethnicity not specified
DiseaseAssertion: Glanzmann thrombasthenia type I
FamilyInfo: Child is patient LN, significant other is patient LB
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Bleeding symptoms were evaluated by examining available hospital records. Mild bleeders were defined as those who had minor symptoms, such as epistaxis or gum bleeds, and moderate bleeders suffered, in addition, from bleeding complications after surgery and trauma. Severe bleeders were defined as those with a history of spontaneous or life-threatening hemorrhages, such as gastrointestinal bleeding, or who had repeated episodes requiring platelet transfusion. See Table 1.
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: See Table 6
PlateletAggregation: See Table 6
CasePreviousTesting: To ascertain the mutations, we sequenced the coding regions of the αIIb and β3 subunits including the exon/intron boundaries
Gene: ITGA2B
Variant: NM_000419.3:c.3062T>C
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA399787956
gnomAD: N/A
Fetal‐neonatal alloimmune thrombocytopenia and unexpected Glanzmann thrombasthenia carrier: report of two cases
PMID: 15819676
Gene: ITGA2B and ITGB3
HGNC: 6138 and 6156
Mother L
Case1: Mother L
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Assessment of surface PTL expression of GPIIIa-IIb was performed
PlateletAggregation: N/A
CasePreviousTesting: DNA sequence analysis was performed -- direct sequence analysis of the PCR products corresponding to exon 10 was performed here
Gene: ITGA2B
Variant: NM_000212.2:c.1447T>C
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA400029321
gnomAD: N/A
ORIGINAL ARTICLEMutations in GPIIIa molecule as a cause for Glanzmannthrombasthenia in Indian patients
PMID: 15748237
Gene: ITGA2B and ITGB3
HGNC: 6138 and 1656
InsufficientEvidence: This article mentions 22 GT patients and variants, which these patients harbor; however, the article does not specify which patient harbors which variants therefore there is insufficient evidence
The β3 subunit of the integrin αIIbβ3 regulates αIIb-mediated outside-in signaling
PMID: 15701721
Gene: ITGB3
HGNC: 6156
Defects in Glanzmann thrombasthenia and LAD-III (LAD-1/v) syndrome: the role of integrin β1 and β3 in platelet adhesion to collagen
PMID: 22065596
Genes: ITGA2B and/or ITGB3
HGNC: 6138 and 6156
NoEvidence: This article does not mention probands or variants in ITGA2B or ITGB3; however, there are some experimental assays for consideration
Supplemental Table S1:
Case5: Male, India, age not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Bleeding from multiple sites
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: 8% αIIbβ3 expression
PlateletAggregation: Absent platelet aggregation (all the information that is provided in Supp. Table S1)
CasePreviousTesting: Sanger sequencing of exons, splice sites and untranslated regions of the ITGA2B and ITGB3 genes was performed
Gene: ITGA2B
Variant: NM_000419.3:c.1651C>T (p.Arg551Trp)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA399802348
gnomAD: MAF in the East Asian population is 0.00005045 https://gnomad.broadinstitute.org/variant/17-42457471-G-A
PreviouslyPublished: PMID: 16879215
In silico analysis of structural modifications in and around the integrin αIIb genu caused by ITGA2B variants in human platelets with emphasis on Glanzmann thrombasthenia.
PMID: 29385657
Gene: ITGA2B
HGNC: 6138
See Page Notes for Supplemental Proband Data
Supplemental Table S1:
Case12: Male, Italy, age not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: Moderate bleeding
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: <5% αIIbβ3 expression
PlateletAggregation: Absent platelet aggregation (all the information that is provided in Supp. Table S1)
CasePreviousTesting: Sanger sequencing of exons, splice sites and untranslated regions of the ITGA2B and ITGB3 genes was performed
Gene: ITGA2B
Variant: NM_000419.3:c.1742C>A (p.Ala581Asp)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA399802112
gnomAD: N/A
PreviouslyPublished: PMID: 12083483
Upregulation of osteoclast α2β1 integrin compensates for lack of αvβ3 vitronectin receptor in Iraqi‐Jewish‐type Glanzmann thrombasthenia
PMID: 12956766
Gene: ITGA2B and/or ITGB3
HGNC: 6138 and 6156
InsufficientEvidence: This article mentions GT patients; however, there are no variants presented in ITGA2B and/or ITGB3
L718P mutation in the membrane-proximal cytoplasmic tail of β3 promotes abnormal αIIbβ3 clustering and lipid microdomain coalescence, and associates with a thrombasthenia-like phenotype
PMID: 20081061
Gene: ITGB3
HGNC: 6156
Severe and Relapsing Upper GastrointestinalBleeding in a Patient withGlanzmann’s Thrombasthenia
PMID: 10749344
Gene: ITGA2B and/or ITGB3
HGNC: 6138 and 6156
InsufficientEvidence: This article mentions a Glanzmann thrombasthenia patient (Patient DM, 55 year old); however, no variants in ITGA2B and/or ITGB3 were mentioned in this proband therefore there is insufficient evidence
NM_000419:c.1947-9T>C
NM_001098670:c.920A>T
Unable to identify variant for following reasons: 1) NM_001098670 is for RASBRP2 not ITGA2B
allele registry query for ITGA2B c.920A>T yields the possible ID of CA399799125 which has a transcript of ENST00000592462.5:n.920A>T equivalent to the Mane transcript of NM_000419.5:c.2125A>T but that is in exon 21 not exon 9 and this is unlikely to be a match
A Novel Leukocyte Adhesion Deficiency III Variant:Kindlin-3 Deficiency Results in Integrin- andNonintegrin-Related Defects in Different Steps of LeukocyteAdhesion
PMID: 21441448
Gene: ITGA2B
HGNC: 6138
GTa, a 37-year-old female IrishCaucasian
Case1: (GTa) Female, 27 years old, Irish Caucasian
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: No family history of bleeding
ParentalGenotype: N/A
CasePresentingHPOs: HP:0000978
(Bruising)
CaseHPOFreeText: Excessive bleeding after a normal delivery
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Reduced expression of GPIIb/IIIa
PlateletAggregation: Platelet aggregation to ADP, thrombin receptor activating peptide, thrombin and U46619 was less than 10% and ristocetin induced agglutination was normal
CasePreviousTesting: Total RNA isolated from the proband's platelets was used as a template for RT PCR. Seven primer pairs were used to amplify overlapping regions of the entire GPIIIa coding sequence using cDNA template
Gene: ITGB3
Variant: NM_000212.2:c.655G>A (p.Val219Met)
LegacyVariant: (p.Val193Met)
ClinVar: N/A
CAiD: CA8623016
gnomAD: MAF in the South Asian population is 0.00003266 http://gnomad.broadinstitute.org/variant/17-45363666-G-A
Therapeutic expression of the platelet-specific integrin, αIIbβ3, in a murine model for Glanzmann thrombasthenia
PMID:* 15972454
Gene: ITGA2B and/or ITGB3
HGNC: 6138 and 6156
NoEvidence: This article does not mention Glanzmann thrombasthenia patients/probands with variants in ITGA2B and/or ITGB3; therefore, there is no evidence to report
Note: This article investigated the practicality of gene therapy for correcting molecular defects in platelets, and targeted expression of the human integrin β3-subunit in megakaryocytes of β3–/– mice affected with GT. Although, this does not apply to the Platelet Disorder VCEP functional criteria, this could be useful experimental assay information as this deals with mouse models
M5
Case5: Patient M5, Female, French Gypsy, age is not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: See Figure 2 for pedigree
ParentalGenotype: Unaffected parents see pedigree Fig 2
CasePresentingHPOs: N/A
CaseHPOFreeText: France, GT is particularly frequent in the Gypsy population, mainly represented by the Manouche tribe. Their clinical phenotype is the same as for classic type I disease, with a moderate and occasional severe bleeding syndrome
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry analysis has shown that these patients' surface expresses <5% of normal GPIIIa and GPIIb levels (we are not given an exact value for each proband)
PlateletAggregation: "Absent platelet aggregation" this is all the information we are given for platelet aggregation
CasePreviousTesting: N/A
Gene: ITGA2B
Variant: NM_000419.3:c.1544+1G>A
LegacyVariant: N/A
ClinVar: 2895
CAiD: CA10575473
gnomAD: N/A
M10
Case10: Patient M10, Male, French Gypsy, age is not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: See Figure 2 for pedigree
ParentalGenotype: Unaffected parents see pedigree Fig 2
CasePresentingHPOs: N/A
CaseHPOFreeText: France, GT is particularly frequent in the Gypsy population, mainly represented by the Manouche tribe. Their clinical phenotype is the same as for classic type I disease, with a moderate and occasional severe bleeding syndrome
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry analysis has shown that these patients' surface expresses <5% of normal GPIIIa and GPIIb levels (we are not given an exact value for each proband)
PlateletAggregation: "Absent platelet aggregation" this is all the information we are given for platelet aggregation
CasePreviousTesting: N/A
Gene: ITGA2B
Variant: NM_000419.3:c.1544+1G>A
LegacyVariant: N/A
ClinVar: 2895
CAiD: CA10575473
gnomAD: N/A
M1
Case1: Patient M1, Female, French Gypsy, age is not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: See Figure 2 for pedigree
ParentalGenotype: Unaffected parents see pedigree Fig 2
CasePresentingHPOs: N/A
CaseHPOFreeText: France, GT is particularly frequent in the Gypsy population, mainly represented by the Manouche tribe. Their clinical phenotype is the same as for classic type I disease, with a moderate and occasional severe bleeding syndrome
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry analysis has shown that these patients' surface expresses <5% of normal GPIIIa and GPIIb levels (we are not given an exact value for each proband)
PlateletAggregation: "Absent platelet aggregation" this is all the information we are given for platelet aggregation
CasePreviousTesting: N/A
Gene: ITGA2B
Variant: NM_000419.3:c.1544+1G>A
LegacyVariant: N/A
ClinVar: 2895
CAiD: CA10575473
gnomAD: N/A
A variant thrombasthenic phenotype associated with compound heterozygosity of integrinβ3-subunit:(Met124Val)β3 alters the subunit dimerization rendering adecreased number of constitutive active αIIbβ3 receptors
PMID: 15583747
Gene: ITGB3
HGNC: 6156
Low Concentrations of Recombinant Factor VIIaMay Improve the Impaired Thrombin Generationof Glanzmann Thrombasthenia Patients
PMID: 30597506
Gene: ITGA2B and ITGB3
HGNC: 6138 and 6156
23
Case23: Female, Arab, age not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: Table 1 mentions family history available for the proband, however no specific details about family provided in paper
ParentalGenotype: N/A
CasePresentingHPOs: HP:0000421, HP:0000132)
(Epistaxis, menorrhagia)
CaseHPOFreeText: Haematuria
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: N/A
PlateletAggregation: N/A
CasePreviousTesting: N/A
Gene: ITGA2B
Variant: NM_000419.3:c.409-2_419del
LegacyVariant: N/A
ClinVar: 2889
CAiD: CA10575471
gnomAD: N/A
2
Case1: 39 years old, Female, Jewish Iraqi
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Mucocutaneous bleeding
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: All patients demonstrated lack of αIIbβ3 surface expression." No specifics are given on the percentage, but test was performed
PlateletAggregation: Presented with mucocutaneous bleeding, absence of platelet aggregation in response to all physiological agonists in contrast to normal aggregation with ristocetin
CasePreviousTesting: "Molecular analysis disclosed that patient 2 was homozygous for an out of frame mutation (c.2031–2041 del, exon 13) in ITGB3, and patient 4 was compound heterozygote for the same deletion and a large deletion of 11.2Kb del"
Gene: ITGB3
Variant: NM_000212.2:c.2051_2061del (c.2051_2061del)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA915940226
gnomAD: N/A
patient 9
Case1: Patient 9, Female, 10 years old
DiseaseAssertion: Glanzmann thrombasthenia III (Diagnosed at 3 months of age)
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: N/A
CaseHPOFreeText: Bleeding phenotypes are not specified per patient in this article, rather, it states: "Petechia, epistaxis, and prolonged bleeding after trauma were the most common symptoms among studied patients"
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: This patient has type III Glanzmann thrombasthenia; therefore, according to this article, this proband showed normal or near-nromal levels of the GPIIb/IIIa complex via flow cytometry.
PlateletAggregation: Prolonged bleeding time, abnormal clot retraction, impaired platelet aggregation induced by most physiological agonists (ADP, epinephrine, and collagen) except ristocetin, mucosal bleeding, easy bruising, epistaxis, as well as normal prothrombin/acivated partial thromboplastin time, and normal platelet count. (This is among inclusion criteria for this study)
CasePreviousTesting: To identify the underlying molecular mechanism of GPIIb/IIIa complex abnormalities among probands in this study, all the 30 exons of ITGA2B and the 15 exons of ITGB3 were analyzed using PCR–CSGE and DNA sequencing.
Gene: ITGB3
Variant: NM_000212.2:c.719G>A (p.Arg240Gln)
LegacyVariant: N/A
ClinVar: 13553
CAiD: CA123224
gnomAD: N/A
Interesting variations on how a disease is defined: comparisonsof von Willebrand disease and Glanzmann thrombasthenia
PMID: 17166245
Gene: ITGA2B and/or ITGB3
HGNC: 6138 and 6156
Glanzmann Thrombasthenia Due to a Two Amino Acid Deletion in the Fourth Calcium-Binding Domain of αIIb: Demonstration of the Importance of Calcium-Binding Domains in the Conformation of αIIbβ3
PMID: 8704171
Gene: ITA2B
HGNC: 6138
Recognition and management of platelet-refractory bleeding in patients with Glanzmann’s thrombasthenia and other severe platelet function disorders
PMID: 28435309
Gene: ITGA2B and ITGB3
HGNC: 6138 and 6156
NoEvidence: This article reviews the rFVIIa clinical data in patients with GT and platelet refractoriness and discusses clinical implications relevant to the hospital-based physician. This article does not mention probands with variants in ITGA2B or ITGB3
Bilateral linkage between a new deletion polymorphism in intron 21 of the GP IIb gene and the HPA-3b (Bakb) determinant
PMID: 8555087
Gene: ITGA2B
HGNC: 6138
InsufficientEvidence: This article mentions a variant in ITGA2B; however, there are no specific case reports in this article, rather a large cohort that was tested
Modeling Glanzmann thrombasthenia using patient specific iPSCs and restoring platelet aggregation function by CD41 overexpression
PMID: 28232155
Gene: ITGA2B
HGNC: 6138
GT2
Case#: GT2 male, 7 years old
Disease: Glazmann's Thrombasthenia
CasePresentingHPOs: (found in appendix) HP:0000967, HP:0000979, HP:0031364, HP:0000978, HP:0002239
ExpressionEvidence: Flow Cytometry of platelet αIIbβ3: 0%CD41, 0% CD61
FamilyInfo: Positive consanguinity
GenotypingMethod: Sanger DNA sequencing to identify causative mutations in each case
ClinVar: 1691476
CAID: CA400030171
Variant: NM_000212.3:c.1641C>G
GnomAD: Not Found
The platelet fibrinogen receptor: from megakaryocyte to the mortuary
PMID: 24175064
Gene: ITGA2B and ITGB3
HGNC: 6138 and 6156
InsufficientEvidence: This article mentions a variant; however, not in a human.
Nutritional Zinc Increases Platelet Reactivity
PMID: 1951315
Gene: ITGA2B and ITBG3
HGNC: 6138 and 6156
NoEvidence: This article does not mention Glanzmann thrombasthenia probands with variants in ITGA2B and/or ITGB3; therefore, there is no evidence
Association of thrombospondin-1 with the actin cytoskeleton of humanthrombin-activated platelets through anaIIbb3- or CD36-independentmechanism
PMID: 11964147
Gene: ITGA2B and/or ITGB3
HGNC: 6138 and 6156
InsufficientEvidence: This article mentions a Glanzmann thrombasthenia patient who was studied; however, the patient did not have variants in ITGA2B and/or ITGB3 mentioned in this article, therefore there is insufficient evidence
A 3-year-old girl
Case1: Female, 3 year old, ethnicity not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: An analysis of family members indicated that the Gln517Stop and Arg553Stop mutations were located on different alleles and inherited from the patient’s mother and father, respectively
CasePresentingHPOs: HP:0004406, HP:0002248, HP:0000978, HP:0000225
(Recurrent epistaxis, hematemesis, easy bruising, gingival bleeding)
CaseHPOFreeText: Petechial hemorrhages
CaseNotHPOs: HP:0001945, HP:0002014, HP:0005261
Fever, diarreah, hemarthrosis
CaseNotHPOsFreeText: N/A
ProteinExpression: A flow cytometry evaluation revealed marked reduction in glycoprotein IIb/IIIa (See Table 1 for Patient's Lab results)
PlateletAggregation: Patients platelets did not aggregate in response to ADP, epinephrine, arachidonic acid, or collagen but platelets had normal ristocetin induced aggregation
CasePreviousTesting: Molecular genetic analysis of ITGA2B was performed which revealed mutations
Gene: ITGA2B
Variant: NM_000419.3:c.1642C>T (p.Gln548Ter) and NM_000419.3:c.1750C>T (Arg584Ter)
LegacyVariant: Gln517Ter and Arg553Ter
ClinVar: 2892
CAiD: CA399802364 and CA115834
gnomAD: MAF in the East Asian population is 0.0002176
Real-Time Analysis of Mural Thrombus Formation in Various Platelet Aggregation Disorders: Distinct Shear-Dependent Roles of Platelet Receptors and Adhesive Proteins Under Flow
PMID: 10419888
Gene: ITGA2B and/or ITGB3
HGNC: 6138 and 6156
InsufficientEvidence: This article mentions 3Glanzmann thrombasthenia patients; however, there is no mention of variants in ITGA2B and/or ITGB3 within these patients; therefore, there is insufficient evidence. In the present study, they observed the real-time process of mural thrombogenesis on a type 1 collagen coated surface under flow conditions with various shear rates in the blood of patients with major congenital platelet aggregation disorders
proband‐III2
Case3: Male, Israeli-Arab, (age is either 10 or 14)
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: See Fig 1C for pedigree
ParentalGenotype: See Fig 1C for pedigree
CasePresentingHPOs: HP:0000421, HP:0031364, HP:0000967
(Severe epistaxis, ecchymosis, petechiae)
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOFreeText: N/A
ProteinExpression: No surface expression of αIIbβ3 was found in patients’ platelets (See Fig 3)
PlateletAggregation: Absent platelet aggregation in response to ADP, epinephrine, collagen and normal ristocetin-induced platelet agglutination
CasePreviousTesting: DNA fragments containing exons and flanking intronic regions of the aIIb and B3 genes were amplified by PCR using primers
Gene: ITGA2B
Variant: NM_000419.3:c.97A>G (p.Asn2Asp)
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA399806951
gnomAD: N/A
CC
Case9: Patient CC
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs:
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: αIIbβ3 expression
PlateletAggregation: N/A
CasePreviousTesting: The amplification of DNA fragments using PCR and their electrophoretic migration through a denaturing gradient was used to scan the entire coding region, splice site junctions and promoter regions of the GPIIb and GPIIIa genes for all 20 patients in this study
Gene: ITGA2B
Variant: NM_000419.3:c.666C>A (p.Phe222Leu)
LegacyVariant: (p.Phe191Leu)
ClinVar: N/A
CAiD: CA399805465
gnomAD: N/A
Congenital platelet disorders and understanding of plateletfunction
PMID: 24286193
Gene: ITGA2B and ITGB3
HGNC: 6138 and 6156
InsufficientEvidence: This article mentions variants in ITGA2B and ITGB3; however, there are no probands with these variants mentioned in this article. Article mentions, "Due to space restrictions, single case reports have mostly been omitted, as have many historical reports with no recent update"
The probands are two brothers, 18
Case1: Male, 18 years old, ethnicity not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: See Figure 4 for family pedigree of patients carrying the 288delC mutation in GPIIb
ParentalGenotype: Heterozygous for variant
CasePresentingHPOs: HP:0000978 (Bruising)
CaseHPOFreeText: Mucocutaneous bleeding
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry analysis shown in Fig 1
PlateletAggregation: Platelets failed to aggregate either spontaneously or in response to ADP, adrenaline, collagen or arachidonic acid, but they showed agglutination in the presence of ristocetin
CasePreviousTesting: ITGA2B and ITGB3 both were sequenced
Gene: ITGA2B
Variant: NM_000419.3:c.291del
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA915940464
gnomAD: N/A
PMID: 7492773
Gene: ITGA2B and/or ITGB3
HGNC: 6138 and 6156
NoEvidence: This article does not mention a GT case report with variants in ITGA2B and/or ITGB3; therefore, there is no evidence. This article has studied the role of vWF in the interaction of platelets with fibrin under flow conditions.
Table 3 Molecular Diagnosis of Patients with Glanzmann Thrombasthenia:
Case5: GT-5, age, gender, ethnicity not specified
DiseaseAssertion: Glanzmann thrombasthenia type I
FamilyInfo: Segregation analysis confirmed in presence of heterozygous mutations in this proband.
ParentalGenotype: Although segregation analysis confirmed, we do not know which variant in particular came from mom or dad.
CasePresentingHPOs: N/A
CaseHPOFreeText: Patients presented with a variable bleeding diathesis; most patients had epistaxis (54%), cutaneous bleeding (58%), and menorrhagia (100% of women) while other common symptoms (oral cavity, GI, following dental extraction of mild injuries, postpartum, or hematomas) were in general milder. There is no specific phenotypes attached to probands in this paper.
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometry showed that αIIbβ3 expression was less than 5%
PlateletAggregation: There was severely impaired aggregation in response to all physiological agonists, and ristocetin induced platelet agglutination was normal or reduced to some extent.
CasePreviousTesting: Total RNA from peripheral blood was used for cDNA synthesis for RT-PCR. Then, using specific oligonucleotides and conditions, they amplified and sequenced ITGA2B and ITGB3 by Sanger sequencing. Every mutation found in cDNA was confirmed in DNA samples.
Gene: ITGA2B
Variant: NM_000419.3:c.2637del (c.2637delC) and
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA915940297
gnomAD: N/A
56-year-old Algerian woman
Case1: Female, 56 years old, Algerian
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: N/A
CasePresentingHPOs: HP:0004406, HP:0000132
(Recurrent epistaxis, menorrhagia)
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: No platelet αIIbβ3 expression (mentioned briefly in abstract)
PlateletAggregation: The patient shows all the typical signs of GT with no platelet aggregation in response to physiologic agonists and an absence of clot retraction. No mention of ristocetin
CasePreviousTesting: N/A
Gene: ITGA2B
Variant: NM_000419.3:c.1063G>A (p.Glu355Lys)
LegacyVariant: p.Glu324Lys
ClinVar: 2899
CAiD: CA115844
gnomAD: MAF in European (non-Finnish population 0.00001978 http://gnomad.broadinstitute.org/variant/17-42461008-C-T
PreviouslyPublished: Proband 9 in PMID: 2180491
Click here for additional data file
Case Info Found in supplementary data:
Case#: Patient GT, Female 43 years old. Greek family.
FamilyInfo: Two sisters, one affected.
Disease: Glazmann's Thrombasthenia
CasePresentingHPOs: HP:0003540, HP:0000967, HP:0031364, HP:0000421, HP:0000225, HP:0000978, HP:0000132
Impaired platelet aggregation, Petechiae, Ecchymosis, Epistaxis, Gingival bleeding, bruising susceptibility, Menorrhagia,
PlateletAgreggation: Light transmission aggregometry studies demonstrated no aggregation in response to ADP, epinephrine, collagen, and arachidonic acid, but a normal response to ristocetin.
GenotypingMethod: Reverse transcription PCR of platelet RNA confirmed the presence of a homozygous β3 C575R mutation.
ClinVar: NA
PAID: PA2573320341 (almost certainly CA400032399 but no c. reported)
Variant: NA
GnomAD: NA
ExpressionEvidence: Flow cytometry demonstrated normal platelet binding of an antibody to GPIb, but virtually no binding of mAb 7E3. Immunoblot analysis demonstrated the presence of <20% of normal β3, with a faint band corresponding to pro-αIIb
Levonorgestrel-releasing intrauterine system for treatment of heavy menstrual bleeding in adolescents with Glanzmann’s Thrombasthenia: illustrated case series
PMID: 29486748
Gene: ITGA2B and ITGB3
HGNC: 6138 and 6156
InsufficientEvidence: There are two probands in this article with Glanzmann thrombasthenia; however, no mention of variants in these two patients and no mention of ITGA2B or ITBG3 genes
Glanzmann's Thrombasthenia
PMID: 11450390
Gene: ITGA2B and/or ITGB3
HGNC: 6138 and 6156
InsufficientEvidence: This article mentions two probands with Glanzmann thrombasthenia; however, no variants in ITGA2B and/or ITGB3 are mentioned in these probands therefore we have insufficient evidence
Protein A Sepharose immunoadsorption can restore the efficacy of platelet concentrates in patients with Glanzmann's thrombasthenia and anti‐glycoprotein IIb–IIIa antibodies Isabelle Martin Laboratoire d'Hématologie, Centre de Traitement de l'Hémophilie andSearch for more papers by this author Fayçal Kriaa Service de Néphrologie, CHU Kremlin Bicêtre, Assistance Publique‐Hôpitaux de Paris and Faculté de Médecine Paris XI, Le Kremlin‐Bicêtre,Search for more papers by this author Valérie Proulle Laboratoire d'Hématologie, Centre de Traitement de l'Hémophilie andSearch for more papers by this author Benoît Guillet Laboratoire d'Hématologie, Centre de Traitement de l'Hémophilie andSearch for more papers by this author Cécile Kaplan Laboratoire d'Immunologie Plaquettaire, INTS,Search for more papers by this author Roseline D'Oiron Laboratoire d'Hématologie, Centre de Traitement de l'Hémophilie andSearch for more papers by this author Marianne Debré Unité d'Hématologie et d'Immunologie Pédiatrique, Hôpital Necker‐Enfants Malades, Paris, andSearch for more papers by this author Edith Fressinaud Laboratoire d'Hématologie, Centre Hospitalier Régional de Nantes, FranceSearch for more papers by this author Yyes Laurian Laboratoire d'Hématologie, Centre de Traitement de l'Hémophilie andSearch for more papers by this author Gil Tchernia Laboratoire d'Hématologie, Centre de Traitement de l'Hémophilie andSearch for more papers by this author Bernard Charpentier Service de Néphrologie, CHU Kremlin Bicêtre, Assistance Publique‐Hôpitaux de Paris and Faculté de Médecine Paris XI, Le Kremlin‐Bicêtre,Search for more papers by this author Thierry Lambert Laboratoire d'Hématologie, Centre de Traitement de l'Hémophilie andSearch for more papers by this author Marie Dreyfus Laboratoire d'Hématologie, Centre de Traitement de l'Hémophilie andSearch for more papers by this author
PMID: 12472579
Gene: ITGA2B and/or ITGB3
HGNC: 6138 and 6156
InsufficientEvidence: This article mentions GT probands; however, there is no mention of these patients harboring variants in ITGA2B and/or ITGB3; therefore, we have insufficient evidence
Case 1
Case#: 11 yo, Female
DiseaseAssertion: GT
FamilyInfo: No family hx. No pedigree.
CasePresentingHPOs: (HP:0002249) Melena; (HP:0000421) Epistaxis; (HP:0000225) Gingival bleeding; (HP:0100769) Synovitis; (HP:0005261) Joint hemorrhage, right ankle; (HP:0001891) Iron deficiency anemia;
PlateletAggregation: Prothrombin time: 11, Partial thromboplastin time: 26, Platelet function analyzer-100: >300
CaseHPOFreeText: stable intestinal angiodysplasia
ProteinExpression: Flow cytometry: CD41 and 61 <50%
CasePreviousTesting: Molecular sequencing, Patients’ information was obtained from the medical record system of the Madinah Hereditary Blood Disorders Center, Maternity and Children Hospital, King Fahd Hospital, Al Madinah, Saudi Arabia.
Gene: ITGB3
HGVS: NM_000212.3:c.2111_2112del
ClinVar:
CAID: CA3062865178
gnomAD:
ADeletionintheGeneforGlycoproteinlibAssociatedwithGlanzmann'sThrombasthenia
PMID: 1702098
Gene: ITGA2B
HGNC: 6156
Truncation of the Cytoplasmic Domain of b3 in a Variant Form of Glanzmann Thrombasthenia Abrogates Signaling through the Integrin aIIbb3 Complex
PMID: 9351872
Gene: ITGB3
HGNC: 6156
GT5
Disease: Glazmann's Thrombasthenia
Case: 48 year old female French Gypsy
CasePresentingHPOs: HP:0001975
(Decreased platelet glycoprotein IIb-IIIa)
GenotypingMethod:Genotyping analysis of the HPA-1 system by high resolution melting analysis (methods not provided, cited article I don't have access too)
ClinVar: 2895
CAID: CA10575473
Variant: NM_000419.5:c.1544+1G>A
GnomAD: Not found
ExpressionEvidence: Failed to express αIIbβ3 integrin on their platelet surface (methods not provided cited article no access too)
AnInheritedBleedingDisorderLinkedtoaDefectiveInteractionbetweenADPandItsReceptoronPlatelets
PMID: 7706468
Gene: ITGA2B and/or ITGB3
HGNC: 6138 and 6156
InsufficientEvidence: This mentions a Glanzmann thrombasthenia patient; however, no variants in ITGA2B and/or ITGB3 are mentioned in this patient; therefore, we have insufficient evidence
Red blood cell transfusion in patients with type 1 Glanzmann’sthrombasthenia
PMID: 16194208
Gene: ITGA2B and/or ITGB3
HGNC: 6138 and 6156
InsufficientEvidence: This article mentions a proband with GT; however, there is no mention of variants in ITGA2B and/or ITGB3, therefore we have insufficient evidence
Alloimmunization in Congenital Deficiencies of Platelet Surface Glycoproteins: Focus on Glanzmann's Thrombasthenia and Bernard–Soulier's Syndrome
PMID: 29879742
Gene:ITGA2B
HGNC: 6138
InsufficientEvidence Variant IVS15(+1)G > A mutation in ITGA2B is mentioned in this article; however, no proband to tie variant to therefore we have insufficient evidence
Rapid diagnosis of the French gypsy mutation in Glanzmann thrombasthenia using high- resolution melting analysis
PMID: 20886184
Gene: ITGA2B
HGNC: 6138
InsufficientEvidence: Variant mentioned here; however, specific proband information is not provided here, probands are clumped together in cohort
Three novel mutations in the glycoprotein IIb gene in a patient with type II Glanzmann thrombasthenia
PMID: 17488698
Gene: ITGA2B
HGNC: 6138
Identification of the integrinb3 L718P mutation in a pedigreewith autosomal dominant thrombocytopenia with anisocytosis
PMID: 23253071
Gene: ITGB3
HGNC: 6156
Triplejeopardy:AcaseofGlanzmann'sthrombastheniawithanti-GPIIb-IIIaantibodiesandHPAincompatibilityresultinginstillbirth
PMID: 31404847
Gene: ITGA2B
HGNC: 6138
Perioperative Management of a Heterozygous Carrier of Glanzmann’s Thrombasthenia Submitted to Coronary Artery Bypass Grafting With Cardiopulmonary Bypass
PMID: 16861409
Gene: ITGA2B and/or ITGB3
HGNC: 6138 and 6156
InsufficientEvidence: This mentions a Glanzmann thrombasthenia patient; however, no variants in ITGA2B and/or ITGB3 are mentioned in this patient; therefore, we have insufficient evidence
Catheter Intervention in a Patient with Intracranial Aneurysms and Glanzmann Thrombasthenia Caused by a Novel Homozygous Likely Pathogenic Variant in the ITGA2B Gene
PMID: 39057107
Gene: ITGA2B
GNC: 6138
13
Case2: Female, 41 years old, ethnicity not specified
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: N/A
ParentalGenotype: "This mutation was confirmed in the proband and parents."
CasePresentingHPOs: HP:0000225 (Gingival bleeding)
CaseHPOFreeText: Site of bleeding: Skin
CaseNotHPOs: N/A
CaseNotHPOFreeText: N/A
ProteinExpression: See Table 1 (information provided)
PlateletAggregation: See Table 1 (information provided)
CasePreviousTesting: Screened mutations in ITGA2B and ITGB3 by multiplex polymerase chain reaction (PCR) amplification (MPCR) and conformation-sensitive gel electrophoresis
Gene: ITGA2B
Variant: NM_000419.3:c.1750C>T (p.Arg584Ter)
LegacyVariant: N/A
ClinVar: 2892
CAiD: CA115834
gnomAD: MAF in the East Asian population is 0.0002176 https://gnomad.broadinstitute.org/variant/17-42457372-G-A
A novel homozygous splice junction mutation in GPIIb associated with alternative splicing, nonsense‐mediated decay of GPIIb‐mRNA, and type II Glanzmann's thrombasthenia C. González‐Manchón Department of Pathophysiology and Human Molecular Genetics, Centro de Investigaciones Biológicas (CSIC), Madrid, Spain; andSearch for more papers by this author E. G. Arias‐Salgado Department of Pathophysiology and Human Molecular Genetics, Centro de Investigaciones Biológicas (CSIC), Madrid, Spain; andSearch for more papers by this author N. Butta Department of Pathophysiology and Human Molecular Genetics, Centro de Investigaciones Biológicas (CSIC), Madrid, Spain; andSearch for more papers by this author G. Martín Department of Pathophysiology and Human Molecular Genetics, Centro de Investigaciones Biológicas (CSIC), Madrid, Spain; andSearch for more papers by this author R. B. Rodríguez Department of Pathophysiology and Human Molecular Genetics, Centro de Investigaciones Biológicas (CSIC), Madrid, Spain; andSearch for more papers by this author I. Elalamy Service d'Hématologie Biologique, Hôpital d'Enfants A Trousseau, Paris, FranceSearch for more papers by this author R. Parrilla Department of Pathophysiology and Human Molecular Genetics, Centro de Investigaciones Biológicas (CSIC), Madrid, Spain; andSearch for more papers by this author R. Favier Service d'Hématologie Biologique, Hôpital d'Enfants A Trousseau, Paris, FranceSearch for more papers by this author
PMID: 12871379
Gene: ITGA2B
HGNC: 6138
Single Point Mutation in Human Glycoprotein IIIa Is Associated With a New Platelet-Specific Alloantigen (Mo) Involved in Neonatal Alloimmune Thrombocytopenia
PMID: 8093349
Gene: ITGB3
HGNC: 6156
A 62‐year‐old Egyptian male
Case#: Male, 62, Egyptian
DiseaseAssertion: GT with concurrent platelet CD36 deficiency (~1 in 100 million) and anti‐CD36 antibodies
CasePresentingHPOs: HP:0000421,Epistaxis. HP:0000978, Bruising susceptibility. HP:0000225, Gingival bleeding. HP:0001903, Anemia. HP:0001934, Persistent bleeding after trauma. HP:0001058, Poor wound healing. HP:0002573, Hematochezia. HP:0005261, Joint hemorrhage.
PlateletAggregation: See Table 1. Normal platelet count. Normal platelet size, modestly elevated mean platelet volume. Normal response to ristocetin.
ProteinExpression: Flow cytometry. GPIb/IX Expression 108% (range: 65-124%); GPIIb/IIIa expression 0% (range:65-124%).
Gene: ITGA2B
Variant: c.2148dup, p.Leu717Alafs3X
HGVS: NM_000419.5:c.2148dup
ClinVar: 627273
CAID: CA915940304
gnomAD: Absent
Variant: c.1234G>A, p.Gly412Arg
HGVS: NM_000419.5:c.1234G>A
ClinVar: 381748
CAID: CA8603182
gnomAD: 0.00003236, https://gnomad.broadinstitute.org/variant/chr17-44381038-C-T?dataset=gnomad_r4
ANewVariantofGlanzmann'sThrombasthenia(StrasbourgI)
PMID: 1602006
Gene: ITGB3
HGNC: 6156
In silico analysis of Glanzmann variants of Calf-1 domain of αIIbβ3 integrin revealed dynamic allosteric effect
PMID: 28808266
Gene: ITGA2B and ITGB3
HGNC: 6138 and 6156
InsufficientEvidence: This article mentions 7 Glanzmann thrombasthenia variants that were identified in a long independent molecular dynamics simulation. There are no probands with these variants mentioned.
Case 1
Case1: Female, 9 years old, Japanese
DiseaseAssertion: Macrothrombocytopenia
FamilyInfo: Information given in Table 1 and Fig 2
ParentalGenotype: Mother: ITGA2B (p.Arg422Ter heterozygous) and Father: ITGA2B (p.Gly991Cys heterozygous) See Table 1 for this information
Sequencing of parental DNA indicated parental mutations. CasePresentingHPOs: HP:0000979, HP:0000421, HP:0000978
(Purpura, epistaxis, bruising)
CaseHPOFreeText: Hemorrhage after dental
CaseNotHPOs: N/A
CaseNotHPOsFreeText: N/A
ProteinExpression: Flow cytometric analysis showed that surface αIIbβ3 expression in platelets (was markedly impaired 3-11%)
PlateletAggregation: N/A
CasePreviousTesting: Sequence analysis of entire coding regions of exons and exon-intron boundaries of ITGA2B and ITGB3 gene of the patient revealed two novel mutations in ITGA2B.
Gene: ITGA2B
Variant: NM_000419.3:c.3064G>T (p.Gly1022Cys) and NM_000419.3:c.1357C>T (p.Arg453Ter)
LegacyVariant: (p.Gly991Cys legacy nomenclature for p.Gly1022Cys) and (p.Arg422Ter legacy nomenclature for p.Arg453Ter)
ClinVar: 631773
CAiD: CA399787943 and CA399803529
gnomAD: N/A
Case1: Male, 61 years old, Iraqi-Jewish origin
DiseaseAssertion: Glanzmann thrombasthenia
FamilyInfo: His sister is the other proband in this article
ParentalGenotype: N/A
CasePresentingHPOs: HP:0000225, HP:0000225, HP:0031364, HP:0002249
(Gingival bleeding, epistaxis, ecchymosis, melena)
CaseHPOFreeText: N/A
CaseNotHPOs: N/A
CaseNotHPOFreeText: N/A
ProteinExpression: Surface expression GPIIb/IIIa was 9% of normal
PlateletAggregation: Platelet aggregation in response to thrombin, collagen, ADP and epinephrine was absent, whereas ristocetin induced aggregation was normal
CasePreviousTesting: DNA fragments containing exons and flanking intronic regions of the GPIIb gene were amplified by PCR
Gene: ITGA2B
Variant: NM_000419.3:c.1947-2A>G
LegacyVariant: N/A
ClinVar: N/A
CAiD: CA399801096
gnomAD: N/A