Activity
| Date | Panel | Item | Activity | ||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|
|
40 actions
|
|||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v9.102 | PSMB8 |
Achchuthan Shanmugasundram changed review comment from: There is already sufficient evidence available for the association of biallelic variants in this gene with a relevant phenotype - associated with MIM #256040 in OMIM and with PSMB8-related Nakajo syndrome (with 'definitive' rating on the DD panel) in Gene2Phenotype (records last accessed 13 August 2026). PMID:41253591 (2026) reported the identification of a novel de novo heterozygous PSMB8 variant, p.G209R (c.625G>A/C), identified in 8 unrelated patients (6/8 de novo, 2/8 assumed de novo) presenting with a chronic atypical neutrophilic dermatosis with lipodystrophy and elevated temperature/ proteasome-associated autoinflammatory syndrome (CANDLE/ PRAAS) phenotype featuring neonatal-onset systemic inflammation, panniculitis, lipodystrophy, cytopenias (anaemia 8/8, lymphopenia and hypogammaglobulinemia 7/8), recurrent viral/bacterial/fungal infections, and porto-sinusoidal vascular liver disease with nodular regenerative hyperplasia—phenotypically broader and more severe than biallelic PSMB8-CANDLE. Structural modelling, transfection, and knockout cell studies showed that the PSMB8 p.G209R variant sterically disrupts the β5i–β4 (PSMB2) interface, arresting propeptide processing and 20S maturation in ~75% of assembling immunoproteasomes—directly demonstrating a dominant-negative mechanism rather than simple haploinsufficiency. PMID:42167218 (2026) reported the identification of five distinct monoallelic missense variants in PSMB8 gene in seven patients from five unrelated families with proteasome-associated autoinflammatory syndromes with immunodeficiency (PRAAS-ID). Four of these variants were inherited de novo in the respective families (although patient I7 inherited from mother (I6), for whom it is de novo), while it is inherited from their mother and mosaic (3%) in one family (patients I2/ I3). These patients presented with neonatal-onset immunodeficiency characterized by recurrent infections, B cell lymphopenia, and hypogammaglobulinemia requiring immunoglobulin replacement. Inflammatory manifestations of variable severity included enteropathy, hepatitis, myositis, and inflammatory lung disease. Functional studies showed that monoallelic PSMB8 variants impair immunoproteasome assembly, causing ~50% loss of mature IP complexes and reduced IP-specific activity while unincorporated mutant subunits accumulate as a stalled, chaperone-bound ~440-kDa assembly intermediate, rather than simply being degraded. This structural disruption destabilizing key PSMB8-PSMB2/PSMB3 interfaces supports a dominant-negative mechanism that reduces functional immunoproteasome output and drives the combined immunodeficiency and autoinflammation seen in PRAAS-ID. Monoallelic variants are not yet associated with any phenotypes in OMIM, Gene2Phenotype or ClinGen (last accessed 13 August 2026).; to: There is already sufficient evidence available for the association of biallelic variants in this gene with a relevant phenotype - associated with MIM #256040 in OMIM and with PSMB8-related Nakajo syndrome (with 'definitive' rating on the DD panel) in Gene2Phenotype (records last accessed 13 August 2026). PMID:41253591 (2026) reported the identification of a novel de novo heterozygous PSMB8 variant, p.G209R (c.625G>A/C), identified in 8 unrelated patients (6/8 de novo, 2/8 assumed de novo) presenting with a chronic atypical neutrophilic dermatosis with lipodystrophy and elevated temperature/ proteasome-associated autoinflammatory syndrome (CANDLE/ PRAAS) phenotype featuring neonatal-onset systemic inflammation, panniculitis, lipodystrophy, cytopenias (anaemia 8/8, lymphopenia and hypogammaglobulinemia 7/8), recurrent viral/bacterial/fungal infections, and porto-sinusoidal vascular liver disease with nodular regenerative hyperplasia—phenotypically broader and more severe than biallelic PSMB8-CANDLE. Structural modelling, transfection, and knockout cell studies showed that the PSMB8 p.G209R variant sterically disrupts the β5i–β4 (PSMB2) interface, arresting propeptide processing and 20S maturation in ~75% of assembling immunoproteasomes, directly demonstrating a dominant-negative mechanism rather than simple haploinsufficiency. PMID:42167218 (2026) reported the identification of five distinct monoallelic missense variants in PSMB8 gene in seven patients from five unrelated families with proteasome-associated autoinflammatory syndromes with immunodeficiency (PRAAS-ID). Four of these variants were inherited de novo in the respective families (although patient I7 inherited from mother (I6), for whom it is de novo), while it is inherited from their mother and mosaic (3%) in one family (patients I2/ I3). These patients presented with neonatal-onset immunodeficiency characterized by recurrent infections, B cell lymphopenia, and hypogammaglobulinemia requiring immunoglobulin replacement. Inflammatory manifestations of variable severity included enteropathy, hepatitis, myositis, and inflammatory lung disease. Functional studies showed that monoallelic PSMB8 variants impair immunoproteasome assembly, causing ~50% loss of mature IP complexes and reduced IP-specific activity while unincorporated mutant subunits accumulate as a stalled, chaperone-bound ~440-kDa assembly intermediate, rather than simply being degraded. This structural disruption destabilizing key PSMB8-PSMB2/PSMB3 interfaces supports a dominant-negative mechanism that reduces functional immunoproteasome output and drives the combined immunodeficiency and autoinflammation seen in PRAAS-ID. Monoallelic variants are not yet associated with any phenotypes in OMIM, Gene2Phenotype or ClinGen (last accessed 13 August 2026). |
|||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v9.102 | PSMB8 | Achchuthan Shanmugasundram Publications for gene: PSMB8 were set to 21881205; 20159315; 21953331; 21129723; 20534754; 21852578; 42167218 | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v9.101 | PSMB8 |
Achchuthan Shanmugasundram changed review comment from: Comment on mode of inheritance: There is sufficient evidence available (>10 unrelated families and functional work) for the association of monoallelic PSMB8 variants with immunodeficiency and inflammation. Hence, the MOI should be updated to 'BOTH monoallelic and biallelic, autosomal or pseudoautosomal' in the next GMS update.; to: Comment on mode of inheritance: There is sufficient evidence available (>10 unrelated families and functional work) for the association of monoallelic PSMB8 variants with proteasome-associated autoinflammatory syndrome and immunodeficiency. Hence, the MOI should be updated to 'BOTH monoallelic and biallelic, autosomal or pseudoautosomal' in the next GMS update. |
|||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v9.101 | PSMB8 |
Achchuthan Shanmugasundram changed review comment from: Comment on mode of inheritance: There is sufficient evidence available (>3 unrelated families and functional work) for the association of monoallelic PSMB8 variants with immunodeficiency and inflammation. Hence, the MOI should be updated to 'BOTH monoallelic and biallelic, autosomal or pseudoautosomal' in the next GMS update.; to: Comment on mode of inheritance: There is sufficient evidence available (>10 unrelated families and functional work) for the association of monoallelic PSMB8 variants with immunodeficiency and inflammation. Hence, the MOI should be updated to 'BOTH monoallelic and biallelic, autosomal or pseudoautosomal' in the next GMS update. |
|||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v9.101 | PSMB8 |
Achchuthan Shanmugasundram changed review comment from: There is already sufficient evidence available for the association of biallelic variants in this gene with a relevant phenotype - associated with MIM #256040 in OMIM and with PSMB8-related Nakajo syndrome (with 'definitive' rating on the DD panel) in Gene2Phenotype (records last accessed 13 August 2026). PMID:41253591 (2026) reported the identification of a novel de novo heterozygous PSMB8 variant, p.G209R (c.625G>A/C), identified in 8 unrelated patients (6/8 de novo, 2/8 assumed de novo) presenting with a chronic atypical neutrophilic dermatosis with lipodystrophy and elevated temperature/ proteasome-associated autoinflammatory syndrome (CANDLE/ PRAAS) phenotype featuring neonatal-onset systemic inflammation, panniculitis, lipodystrophy, cytopenias (anaemia 8/8, lymphopenia and hypogammaglobulinemia 7/8), recurrent viral/bacterial/fungal infections, and porto-sinusoidal vascular liver disease with nodular regenerative hyperplasia—phenotypically broader and more severe than biallelic PSMB8-CANDLE. Structural modelling, transfection, and knockout cell studies showed that the PSMB8 p.G209R variant sterically disrupts the β5i–β4 (PSMB2) interface, arresting propeptide processing and 20S maturation in ~75% of assembling immunoproteasomes—directly demonstrating a dominant-negative mechanism rather than simple haploinsufficiency. PMID:42167218 (2026) reported the identification of five distinct monoallelic missense variants in PSMB8 gene in seven patients from five unrelated families with proteasome-associated autoinflammatory syndromes with immunodeficiency (PRAAS-ID). Four of these variants were inherited de novo in the respective families (although patient I7 inherited from mother (I6), for whom it is de novo), while it is inherited from their mother and mosaic (3%) in one family (patients I2/ I3). These patients presented with neonatal-onset immunodeficiency characterized by recurrent infections, B cell lymphopenia, and hypogammaglobulinemia requiring immunoglobulin replacement. Inflammatory manifestations of variable severity included enteropathy, hepatitis, myositis, and inflammatory lung disease. Functional studies showed that monoallelic PSMB8 variants impair immunoproteasome assembly, causing ~50% loss of mature IP complexes and reduced IP-specific activity while unincorporated mutant subunits accumulate as a stalled, chaperone-bound ~440-kDa assembly intermediate, rather than simply being degraded. This structural disruption destabilizing key PSMB8-PSMB2/PSMB3 interfaces supports a dominant-negative mechanism that reduces functional immunoproteasome output and drives the combined immunodeficiency and autoinflammation seen in PRAAS-ID. Monoallelic variants are not yet associated with any phenotypes in OMIM, Gene2Phenotype or ClinGen (last accessed 13 August 2026).; to: There is already sufficient evidence available for the association of biallelic variants in this gene with a relevant phenotype - associated with MIM #256040 in OMIM and with PSMB8-related Nakajo syndrome (with 'definitive' rating on the DD panel) in Gene2Phenotype (records last accessed 13 August 2026). PMID:41253591 (2026) reported the identification of a novel de novo heterozygous PSMB8 variant, p.G209R (c.625G>A/C), identified in 8 unrelated patients (6/8 de novo, 2/8 assumed de novo) presenting with a chronic atypical neutrophilic dermatosis with lipodystrophy and elevated temperature/ proteasome-associated autoinflammatory syndrome (CANDLE/ PRAAS) phenotype featuring neonatal-onset systemic inflammation, panniculitis, lipodystrophy, cytopenias (anaemia 8/8, lymphopenia and hypogammaglobulinemia 7/8), recurrent viral/bacterial/fungal infections, and porto-sinusoidal vascular liver disease with nodular regenerative hyperplasia—phenotypically broader and more severe than biallelic PSMB8-CANDLE. Structural modelling, transfection, and knockout cell studies showed that the PSMB8 p.G209R variant sterically disrupts the β5i–β4 (PSMB2) interface, arresting propeptide processing and 20S maturation in ~75% of assembling immunoproteasomes—directly demonstrating a dominant-negative mechanism rather than simple haploinsufficiency. PMID:42167218 (2026) reported the identification of five distinct monoallelic missense variants in PSMB8 gene in seven patients from five unrelated families with proteasome-associated autoinflammatory syndromes with immunodeficiency (PRAAS-ID). Four of these variants were inherited de novo in the respective families (although patient I7 inherited from mother (I6), for whom it is de novo), while it is inherited from their mother and mosaic (3%) in one family (patients I2/ I3). These patients presented with neonatal-onset immunodeficiency characterized by recurrent infections, B cell lymphopenia, and hypogammaglobulinemia requiring immunoglobulin replacement. Inflammatory manifestations of variable severity included enteropathy, hepatitis, myositis, and inflammatory lung disease. Functional studies showed that monoallelic PSMB8 variants impair immunoproteasome assembly, causing ~50% loss of mature IP complexes and reduced IP-specific activity while unincorporated mutant subunits accumulate as a stalled, chaperone-bound ~440-kDa assembly intermediate, rather than simply being degraded. This structural disruption destabilizing key PSMB8-PSMB2/PSMB3 interfaces supports a dominant-negative mechanism that reduces functional immunoproteasome output and drives the combined immunodeficiency and autoinflammation seen in PRAAS-ID. Monoallelic variants are not yet associated with any phenotypes in OMIM, Gene2Phenotype or ClinGen (last accessed 13 August 2026). |
|||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v9.101 | PSMB8 |
Achchuthan Shanmugasundram changed review comment from: PMID:42167218 (2026) reported the identification of five distinct monoallelic missense variants in PSMB8 gene in seven patients from five unrelated families with proteasome-associated autoinflammatory syndromes with immunodeficiency (PRAAS-ID). Four of these variants were inherited de novo in the respective families (although patient I7 inherited from mother (I6), for whom it is de novo), while it is inherited from their mother and mosaic (3%) in one family (patients I2/ I3). These patients presented with neonatal-onset immunodeficiency characterized by recurrent infections, B cell lymphopenia, and hypogammaglobulinemia requiring immunoglobulin replacement. Inflammatory manifestations of variable severity included enteropathy, hepatitis, myositis, and inflammatory lung disease. Functional studies showed that monoallelic PSMB8 variants impair immunoproteasome assembly, causing ~50% loss of mature IP complexes and reduced IP-specific activity while unincorporated mutant subunits accumulate as a stalled, chaperone-bound ~440-kDa assembly intermediate, rather than simply being degraded. This structural disruption destabilizing key PSMB8-PSMB2/PSMB3 interfaces supports a dominant-negative mechanism that reduces functional immunoproteasome output and drives the combined immunodeficiency and autoinflammation seen in PRAAS-ID. There is already sufficient evidence available for the association of biallelic variants in this gene with a relevant phenotype - associated with MIM #256040 in OMIM and with PSMB8-related Nakajo syndrome (with definitive rating on the DD panel) in Gene2Phenotype (records last accessed 13 August 2026).; to: There is already sufficient evidence available for the association of biallelic variants in this gene with a relevant phenotype - associated with MIM #256040 in OMIM and with PSMB8-related Nakajo syndrome (with 'definitive' rating on the DD panel) in Gene2Phenotype (records last accessed 13 August 2026). PMID:41253591 (2026) reported the identification of a novel de novo heterozygous PSMB8 variant, p.G209R (c.625G>A/C), identified in 8 unrelated patients (6/8 de novo, 2/8 assumed de novo) presenting with a chronic atypical neutrophilic dermatosis with lipodystrophy and elevated temperature/ proteasome-associated autoinflammatory syndrome (CANDLE/ PRAAS) phenotype featuring neonatal-onset systemic inflammation, panniculitis, lipodystrophy, cytopenias (anaemia 8/8, lymphopenia and hypogammaglobulinemia 7/8), recurrent viral/bacterial/fungal infections, and porto-sinusoidal vascular liver disease with nodular regenerative hyperplasia—phenotypically broader and more severe than biallelic PSMB8-CANDLE. Structural modelling, transfection, and knockout cell studies showed that the PSMB8 p.G209R variant sterically disrupts the β5i–β4 (PSMB2) interface, arresting propeptide processing and 20S maturation in ~75% of assembling immunoproteasomes—directly demonstrating a dominant-negative mechanism rather than simple haploinsufficiency. PMID:42167218 (2026) reported the identification of five distinct monoallelic missense variants in PSMB8 gene in seven patients from five unrelated families with proteasome-associated autoinflammatory syndromes with immunodeficiency (PRAAS-ID). Four of these variants were inherited de novo in the respective families (although patient I7 inherited from mother (I6), for whom it is de novo), while it is inherited from their mother and mosaic (3%) in one family (patients I2/ I3). These patients presented with neonatal-onset immunodeficiency characterized by recurrent infections, B cell lymphopenia, and hypogammaglobulinemia requiring immunoglobulin replacement. Inflammatory manifestations of variable severity included enteropathy, hepatitis, myositis, and inflammatory lung disease. Functional studies showed that monoallelic PSMB8 variants impair immunoproteasome assembly, causing ~50% loss of mature IP complexes and reduced IP-specific activity while unincorporated mutant subunits accumulate as a stalled, chaperone-bound ~440-kDa assembly intermediate, rather than simply being degraded. This structural disruption destabilizing key PSMB8-PSMB2/PSMB3 interfaces supports a dominant-negative mechanism that reduces functional immunoproteasome output and drives the combined immunodeficiency and autoinflammation seen in PRAAS-ID. Monoallelic variants are not yet associated with any phenotypes in OMIM, Gene2Phenotype or ClinGen (last accessed 13 August 2026). |
|||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v9.101 | PSMB8 | Achchuthan Shanmugasundram edited their review of gene: PSMB8: Changed publications to: 41253591, 42167218 | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v9.101 | PSMB8 |
Achchuthan Shanmugasundram Added comment: Comment on mode of inheritance: There is sufficient evidence available (>3 unrelated families and functional work) for the association of monoallelic PSMB8 variants with immunodeficiency and inflammation. Hence, the MOI should be updated to 'BOTH monoallelic and biallelic, autosomal or pseudoautosomal' in the next GMS update. |
|||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v9.101 | PSMB8 | Achchuthan Shanmugasundram Mode of inheritance for gene: PSMB8 was changed from BIALLELIC, autosomal or pseudoautosomal to BIALLELIC, autosomal or pseudoautosomal | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v9.100 | PSMB8 | Achchuthan Shanmugasundram Mode of pathogenicity for gene: PSMB8 was changed from None to Other | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v9.99 | PSMB8 |
Achchuthan Shanmugasundram changed review comment from: PMID:42167218 (2026) reported the identification of five distinct monoallelic missense variants in PSMB8 gene in seven patients from five unrelated families with proteasome-associated autoinflammatory syndromes with immunodeficiency (PRAAS-ID). Four of these variants were inherited de novo in the respective families (although patient I7 inherited from mother (I6), for whom it is de novo), while it is inherited from their mother and mosaic (3%) in one family (patients I2/ I3). These patients presented with neonatal-onset immunodeficiency characterized by recurrent infections, B cell lymphopenia, and hypogammaglobulinemia requiring immunoglobulin replacement. Inflammatory manifestations of variable severity included enteropathy, hepatitis, myositis, and inflammatory lung disease. There is already sufficient evidence available for the association of biallelic variants in this gene with a relevant phenotype - associated with MIM #256040 in OMIM and with PSMB8-related Nakajo syndrome (with definitive rating on the DD panel) in Gene2Phenotype (records last accessed 13 August 2026).; to: PMID:42167218 (2026) reported the identification of five distinct monoallelic missense variants in PSMB8 gene in seven patients from five unrelated families with proteasome-associated autoinflammatory syndromes with immunodeficiency (PRAAS-ID). Four of these variants were inherited de novo in the respective families (although patient I7 inherited from mother (I6), for whom it is de novo), while it is inherited from their mother and mosaic (3%) in one family (patients I2/ I3). These patients presented with neonatal-onset immunodeficiency characterized by recurrent infections, B cell lymphopenia, and hypogammaglobulinemia requiring immunoglobulin replacement. Inflammatory manifestations of variable severity included enteropathy, hepatitis, myositis, and inflammatory lung disease. Functional studies showed that monoallelic PSMB8 variants impair immunoproteasome assembly, causing ~50% loss of mature IP complexes and reduced IP-specific activity while unincorporated mutant subunits accumulate as a stalled, chaperone-bound ~440-kDa assembly intermediate, rather than simply being degraded. This structural disruption destabilizing key PSMB8-PSMB2/PSMB3 interfaces supports a dominant-negative mechanism that reduces functional immunoproteasome output and drives the combined immunodeficiency and autoinflammation seen in PRAAS-ID. There is already sufficient evidence available for the association of biallelic variants in this gene with a relevant phenotype - associated with MIM #256040 in OMIM and with PSMB8-related Nakajo syndrome (with definitive rating on the DD panel) in Gene2Phenotype (records last accessed 13 August 2026). |
|||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v9.99 | PSMB8 | Achchuthan Shanmugasundram edited their review of gene: PSMB8: Changed mode of pathogenicity: Other | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v9.99 | PSMB8 | Achchuthan Shanmugasundram Publications for gene: PSMB8 were set to 21881205; 20159315; 21953331; 21129723; 20534754; 21852578 | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v9.98 | PSMB8 | Achchuthan Shanmugasundram Phenotypes for gene: PSMB8 were changed from Proteasome-associated autoinflammatory syndrome 1 and digenic forms, OMIM:256040; Autoinflammation, lipodystrophy, and dermatosis syndrome; Contractures, panniculitis, ICC, fevers; Autoinflammatory Disorders; CANDLE syndrome to Proteasome-associated autoinflammatory syndrome 1 and digenic forms, OMIM:256040; proteasome-associated autoinflammatory syndrome 1, MONDO:0054698; Immunodeficiency, HP:0002721 | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v9.97 | PSMB8 | Achchuthan Shanmugasundram Tag Q3_26_MOI tag was added to gene: PSMB8. | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v9.97 | PSMB8 | Achchuthan Shanmugasundram reviewed gene: PSMB8: Rating: GREEN; Mode of pathogenicity: None; Publications: 42167218; Phenotypes: Proteasome-associated autoinflammatory syndrome 1 and digenic forms, OMIM:256040, proteasome-associated autoinflammatory syndrome 1, MONDO:0054698, Immunodeficiency, HP:0002721; Mode of inheritance: BOTH monoallelic and biallelic, autosomal or pseudoautosomal | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v2.516 | PSMB8 | Arina Puzriakova Phenotypes for gene: PSMB8 were changed from Autoinflammation, lipodystrophy, and dermatosis syndrome 256040; Other autoinflammatory diseases with known genetic defect; CANDLE syndrome; chronic atypical neutrophilic dermatosis with lipodystrophy and elevated temperature syndrome (CANDLE); Contractures, panniculitis, ICC, fevers; Autoinflammatory Disorders to Proteasome-associated autoinflammatory syndrome 1 and digenic forms, OMIM:256040; Autoinflammation, lipodystrophy, and dermatosis syndrome; Contractures, panniculitis, ICC, fevers; Autoinflammatory Disorders; CANDLE syndrome | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v2.297 | PSMB8 | Eleanor Williams Classified gene: PSMB8 as Green List (high evidence) | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v2.297 | PSMB8 | Eleanor Williams Added comment: Comment on list classification: Changed rating of gene from Red to Green. This gene was rated as Green in v2.208 and incorrectly automatically demoted to Red in v2.209. This was due to a defect in the automatic PanelApp uploading tool when a set of publications was added to the panel with the source ‘Other’, and under certain conditions associated to previous sources listed, resulted in the rating of the gene being automatically changed when it should not have been. | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v2.297 | PSMB8 | Eleanor Williams Gene: psmb8 has been classified as Green List (High Evidence). | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v2.209 | PSMB8 |
Eleanor Williams Source Other was added to PSMB8. Publications for gene PSMB8 were updated from 21129723; 21953331; 21881205; 21852578; 21953331 to 21881205; 20159315; 21953331; 21129723; 20534754; 21852578 Rating Changed from Green List (high evidence) to Red List (low evidence) |
|||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v2.208 | PSMB8 | Eleanor Williams reviewed gene: PSMB8: Rating: ; Mode of pathogenicity: ; Publications: 20159315, 20534754; Phenotypes: ; Mode of inheritance: | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v1.130 | PSMA3 |
Louise Daugherty commented on gene: PSMA3: ?Proteasome-associated autoinflammatory syndrome 1, digenic. Two unrelated children het for this and another gene (PSMB8) hence ?digenic - amber on association |
|||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v1.94 | PSMB8 | Louise Daugherty commented on gene: PSMB8: Gene rating submitted by Kimberly Gilmour and Austen Worth on behalf of London North GLH for the GMS Immunology specialist test group. As discussed with the GMS Immunology Specialist Test Group during webex call 28th March 2019 and confirmed in follow up email 6th September the Specialist Test Group all agreed there is enough evidence to rate this gene Green. | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v1.94 | PSMB8 | Louise Daugherty commented on gene: PSMB8: Gene rating submitted by Tracy Briggs, David Gokhale and Abigal Rousseau on behalf of North West GLH for the GMS Immunology specialist test group. As discussed with the GMS Immunology Specialist Test Group during webex call 28th March 2019 and confirmed in follow up email on 20th June the Specialist Test Group all agreed there is enough evidence to rate this gene Green. | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v1.94 | PSMB8 | Kimberly Gilmour reviewed gene: PSMB8: Rating: GREEN; Mode of pathogenicity: ; Publications: ; Phenotypes: ; Mode of inheritance: | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v1.94 | PSMB8 | Tracy Briggs reviewed gene: PSMB8: Rating: GREEN; Mode of pathogenicity: ; Publications: ; Phenotypes: ; Mode of inheritance: | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v1.60 | PSMB8 | Louise Daugherty Source NHS GMS was added to PSMB8. | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v1.59 | PSMB8 | Louise Daugherty Source North West GLH was added to PSMB8. | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease v1.58 | PSMB8 | Louise Daugherty Source London North GLH was added to PSMB8. | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease | PSMB8 | Louise Daugherty edited their review of gene: PSMB8 | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease | PSMB8 | Louise Daugherty marked gene: PSMB8 as ready | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease | PSMB8 | Sophie Hambleton reviewed gene: PSMB8 | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease | PSMB8 | Sarah Leigh classified PSMB8 as Green List (high evidence) | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease | PSMB8 | Louise Daugherty commented on PSMB8 | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease | PSMB8 | Louise Daugherty commented on PSMB8 | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease | PSMB8 | Louise Daugherty commented on PSMB8 | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease | PSMB8 | Louise Daugherty reviewed PSMB8 | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease | PSMB8 | Louise Daugherty Added gene to panel | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
| Primary immunodeficiency or monogenic inflammatory bowel disease | PSMB8 | Louise Daugherty Added gene to panel | |||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||