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Renal Tubulointerstitial Disease v1.4 JAG1 Chirag Patel gene: JAG1 was added
gene: JAG1 was added to Renal Tubulointerstitial Disease. Sources: Literature
Mode of inheritance for gene: JAG1 was set to MONOALLELIC, autosomal or pseudoautosomal, NOT imprinted
Publications for gene: JAG1 were set to PMID: 41061854
Phenotypes for gene: JAG1 were set to JAG1-related autosomal dominant tubulointerstitial kidney disease; Alagille syndrome, MONDO:0007318
Review for gene: JAG1 was set to GREEN
Added comment: Kidney malformations and chronic kidney disease are well established in Alagille syndrome.

3 large families (out of 203) with ADTKD and a (likely) pathogenic variant in JAG1 gene segregating with disease. JAG1 expression studies as well ER stress analysis suggested that the tubulointerstitial kidney disease was not due to cell toxicity of an abnormal protein, but rather to haploinsufficiency and loss of function.

None of the 23 adult patients with isolated kidney failure (and tubulointerstitial nephritis in individuals with available kidney biopsy) had syndromic manifestations of Alagille syndrome (i.e. liver, bile duct, heart, eye, or skeletal). Therefore, JAG1 variants should be considered in isolated tubulointerstitial kidney disease.
Sources: Literature
Renal Tubulointerstitial Disease v1.4 JAG1 Chirag Patel gene: JAG1 was added
gene: JAG1 was added to Renal Tubulointerstitial Disease. Sources: Literature
Mode of inheritance for gene: JAG1 was set to MONOALLELIC, autosomal or pseudoautosomal, NOT imprinted
Publications for gene: JAG1 were set to PMID: 41061854
Phenotypes for gene: JAG1 were set to JAG1-related autosomal dominant tubulointerstitial kidney disease; Alagille syndrome, MONDO:0007318
Review for gene: JAG1 was set to GREEN
Added comment: Kidney malformations and chronic kidney disease are well established in Alagille syndrome.

3 large families (out of 203) with ADTKD and a (likely) pathogenic variant in JAG1 gene segregating with disease. JAG1 expression studies as well ER stress analysis suggested that the tubulointerstitial kidney disease was not due to cell toxicity of an abnormal protein, but rather to haploinsufficiency and loss of function.

None of the 23 adult patients with isolated kidney failure (and tubulointerstitial nephritis in individuals with available kidney biopsy) had syndromic manifestations of Alagille syndrome (i.e. liver, bile duct, heart, eye, or skeletal). Therefore, JAG1 variants should be considered in isolated tubulointerstitial kidney disease.
Sources: Literature
Renal Tubulointerstitial Disease v1.0 FAN1 Zornitza Stark changed review comment from: Phenotypic overlap.
Sources: Expert Review; to: Phenotypic overlap.

Well established gene-disease association. Karyomegalic tubulointerstitial nephritis (KMIN) is a rare kidney disease characterized clinically by onset in the third decade of progressive renal failure. Renal biopsy shows chronic tubulointerstitial nephritis and interstitial fibrosis associated with enlarged and atypical tubular epithelial cell nuclei.
Sources: Expert Review
Renal Tubulointerstitial Disease v0.19 REN Zornitza Stark Marked gene: REN as ready
Renal Tubulointerstitial Disease v0.19 REN Zornitza Stark Gene: ren has been classified as Green List (High Evidence).
Renal Tubulointerstitial Disease v0.19 REN Zornitza Stark Phenotypes for gene: REN were changed from to Autosomal dominant tubulointerstitial disease
Renal Tubulointerstitial Disease v0.18 REN Zornitza Stark Publications for gene: REN were set to
Renal Tubulointerstitial Disease v0.17 REN Zornitza Stark Mode of inheritance for gene: REN was changed from Unknown to MONOALLELIC, autosomal or pseudoautosomal, NOT imprinted
Renal Tubulointerstitial Disease v0.16 REN Zornitza Stark reviewed gene: REN: Rating: GREEN; Mode of pathogenicity: None; Publications: 31586593, 31406136, 28701203, 21473025; Phenotypes: Autosomal dominant tubulointerstitial disease; Mode of inheritance: MONOALLELIC, autosomal or pseudoautosomal, NOT imprinted
Renal Tubulointerstitial Disease v0.16 HNF1B Zornitza Stark Phenotypes for gene: HNF1B were changed from to Diabetes mellitus, noninsulin-dependent 125853 AD; Renal cysts and diabetes syndrome 137920 AD; {Renal cell carcinoma} 144700
Renal Tubulointerstitial Disease v0.12 HNF1B Michelle Torres reviewed gene: HNF1B: Rating: GREEN; Mode of pathogenicity: Other; Publications: 25536396, 11845238, 15509593; Phenotypes: Diabetes mellitus, noninsulin-dependent 125853 AD, Renal cysts and diabetes syndrome 137920 AD, {Renal cell carcinoma} 144700; Mode of inheritance: MONOALLELIC, autosomal or pseudoautosomal, NOT imprinted
Renal Tubulointerstitial Disease v0.10 FAN1 Zornitza Stark gene: FAN1 was added
gene: FAN1 was added to Renal Tubulointerstitial Disease. Sources: Expert Review
Mode of inheritance for gene: FAN1 was set to MONOALLELIC, autosomal or pseudoautosomal, NOT imprinted
Phenotypes for gene: FAN1 were set to Interstitial nephritis, karyomegalic, MIM# 614817
Review for gene: FAN1 was set to GREEN
Added comment: Phenotypic overlap.
Sources: Expert Review
Renal Tubulointerstitial Disease v0.8 Zornitza Stark Panel name changed from Renal tubulointerstitial disease_KidGen_VCGS to Renal Tubulointerstitial Disease
Panel types changed to Victorian Clinical Genetics Services; KidGen
Renal Tubulointerstitial Disease v0.5 Zornitza Stark Panel name changed from Renal tubulointerstitial disease_KidGen to Renal tubulointerstitial disease_KidGen_VCGS
Renal Tubulointerstitial Disease v0.3 DNAJB11 Zornitza Stark gene: DNAJB11 was added
gene: DNAJB11 was added to Renal tubulointerstitial disease_KidGen. Sources: Expert list
Mode of inheritance for gene: DNAJB11 was set to MONOALLELIC, autosomal or pseudoautosomal, NOT imprinted
Publications for gene: DNAJB11 were set to 29706351; 29777155
Phenotypes for gene: DNAJB11 were set to Polycystic kidney disease 6 with or without polycystic liver disease, MIM#618061
Review for gene: DNAJB11 was set to GREEN
gene: DNAJB11 was marked as current diagnostic
Added comment: At least 7 unrelated families with variants reported in this gene; variable presentation including tubulointerstitial disease (histologically) in addition to polycystic disease (hybrid between PKD and ADTKD).
Sources: Expert list
Renal Tubulointerstitial Disease v0.1 ATP6V0A4 Zornitza Stark reviewed gene: ATP6V0A4: Rating: RED; Mode of pathogenicity: None; Publications: ; Phenotypes: Renal tubular acidosis, distal, autosomal recessive, MIM#602722; Mode of inheritance: None
Renal Tubulointerstitial Disease v0.0 ATP6V0A4 Zornitza Stark gene: ATP6V0A4 was added
gene: ATP6V0A4 was added to Renal tubulointerstitial disease_KidGen. Sources: KidGen_Tubulointerstitial v38.1.0,Expert Review Green
Mode of inheritance for gene: ATP6V0A4 was set to Unknown
Renal Tubulointerstitial Disease v0.0 AQP2 Zornitza Stark gene: AQP2 was added
gene: AQP2 was added to Renal tubulointerstitial disease_KidGen. Sources: KidGen_Tubulointerstitial v38.1.0,Expert Review Green
Mode of inheritance for gene: AQP2 was set to Unknown
Renal Tubulointerstitial Disease v0.0 UMOD Zornitza Stark gene: UMOD was added
gene: UMOD was added to Renal tubulointerstitial disease_KidGen. Sources: KidGen_Tubulointerstitial v38.1.0,Expert Review Green
Mode of inheritance for gene: UMOD was set to Unknown
Renal Tubulointerstitial Disease v0.0 SEC61A1 Zornitza Stark gene: SEC61A1 was added
gene: SEC61A1 was added to Renal tubulointerstitial disease_KidGen. Sources: KidGen_Tubulointerstitial v38.1.0,Expert Review Green
Mode of inheritance for gene: SEC61A1 was set to Unknown
Renal Tubulointerstitial Disease v0.0 REN Zornitza Stark gene: REN was added
gene: REN was added to Renal tubulointerstitial disease_KidGen. Sources: KidGen_Tubulointerstitial v38.1.0,Expert Review Green
Mode of inheritance for gene: REN was set to Unknown
Renal Tubulointerstitial Disease v0.0 MUC1 Zornitza Stark gene: MUC1 was added
gene: MUC1 was added to Renal tubulointerstitial disease_KidGen. Sources: KidGen_Tubulointerstitial v38.1.0,Expert Review Green
Mode of inheritance for gene: MUC1 was set to Unknown
Renal Tubulointerstitial Disease v0.0 HNF1B Zornitza Stark gene: HNF1B was added
gene: HNF1B was added to Renal tubulointerstitial disease_KidGen. Sources: KidGen_Tubulointerstitial v38.1.0,Expert Review Green
Mode of inheritance for gene: HNF1B was set to Unknown
Renal Tubulointerstitial Disease v0.0 Zornitza Stark Added panel Renal tubulointerstitial disease_KidGen