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Mendeliome v2.0 CDK5 Gene migrated from ENSG00000164885 to ENSG00000164885 (gene set migration)
Mendeliome v1.4916 CDK5RAP3 Sangavi Sivagnanasundram changed review comment from: CDK5RAP3 functions as a regulator that restricts UFMylation which is important for protein function.
No pathogenic variants reported in ClinVar across the gene. Currently, no OMIM entry for this GDA.

This publication reports two probands with a severe neurodevelopmental disorder. Affected probands presented with foetal growth restriction, foetal akinesia, pontocerebellar hypoplasia, arthrogryposis and hepatic pathology. A homozygous deep‑intronic splice variant (c.334+243G>A) was identified in both probands.
Supportive function assay showed loss of CDK5RAP3 protein and rescue by antisense-oligonucleotides however only one patient-derived cell line was used.

Gene to be RED. More evidence is required to upgrade to AMBER.
Sources: Literature; to: CDK5RAP3 functions as a regulator that restricts UFMylation which is important for protein function.
No pathogenic variants reported in ClinVar across the gene. Currently, no OMIM entry for this GDA.

This publication reports two probands with a severe neurodevelopmental disorder. Affected probands presented with foetal growth restriction, foetal akinesia, pontocerebellar hypoplasia, arthrogryposis and hepatic pathology. A homozygous deep‑intronic splice variant (c.334+243G>A) was identified in both probands.
Supportive function assay showed loss of CDK5RAP3 protein and rescue by antisense-oligonucleotides however only one patient-derived cell line was used.

Gene to be RED. More evidence is required to upgrade to the GDA.
Sources: Literature
Mendeliome v1.4916 CDK5RAP3 Sangavi Sivagnanasundram gene: CDK5RAP3 was added
gene: CDK5RAP3 was added to Mendeliome. Sources: Literature
Mode of inheritance for gene: CDK5RAP3 was set to BIALLELIC, autosomal or pseudoautosomal
Publications for gene: CDK5RAP3 were set to 42045457
Phenotypes for gene: CDK5RAP3 were set to Neurodevelopmental disorder, MONDO:0700092
Review for gene: CDK5RAP3 was set to RED
Added comment: CDK5RAP3 functions as a regulator that restricts UFMylation which is important for protein function.
No pathogenic variants reported in ClinVar across the gene. Currently, no OMIM entry for this GDA.

This publication reports two probands with a severe neurodevelopmental disorder. Affected probands presented with foetal growth restriction, foetal akinesia, pontocerebellar hypoplasia, arthrogryposis and hepatic pathology. A homozygous deep‑intronic splice variant (c.334+243G>A) was identified in both probands.
Supportive function assay showed loss of CDK5RAP3 protein and rescue by antisense-oligonucleotides however only one patient-derived cell line was used.

Gene to be RED. More evidence is required to upgrade to AMBER.
Sources: Literature
Mendeliome v1.3146 CDK5 Zornitza Stark Publications for gene: CDK5 were set to 25560765; 32273484; 32097629; 28854363; 7490100
Mendeliome v1.3145 CDK5 Zornitza Stark Classified gene: CDK5 as Green List (high evidence)
Mendeliome v1.3145 CDK5 Zornitza Stark Gene: cdk5 has been classified as Green List (High Evidence).
Mendeliome v1.3143 CDK5 Arina Puzriakova reviewed gene: CDK5: Rating: GREEN; Mode of pathogenicity: None; Publications: 40186457; Phenotypes: Lissencephaly 7 with cerebellar hypoplasia, OMIM:616342; Mode of inheritance: BIALLELIC, autosomal or pseudoautosomal
Mendeliome v1.1212 CDK5R1 Zornitza Stark Phenotypes for gene: CDK5R1 were changed from Intellectual disability; autism to Neurodevelopmental disorder, MONDO:0700092, CDK5R1-related
Mendeliome v1.1211 CDK5R1 Zornitza Stark edited their review of gene: CDK5R1: Changed phenotypes: Neurodevelopmental disorder, MONDO:0700092, CDK5R1-related
Mendeliome v1.565 CDK5 Zornitza Stark Publications for gene: CDK5 were set to 25560765
Mendeliome v1.564 CDK5 Zornitza Stark Classified gene: CDK5 as Amber List (moderate evidence)
Mendeliome v1.564 CDK5 Zornitza Stark Gene: cdk5 has been classified as Amber List (Moderate Evidence).
Mendeliome v1.563 CDK5 Zornitza Stark edited their review of gene: CDK5: Added comment: Upgraded to Amber following GenCC discrepancy resolution: single family with four affected individuals but extensive supportive experimental evidence including mouse models.; Changed rating: AMBER; Changed publications: 25560765, 32273484, 32097629, 28854363, 7490100
Mendeliome v0.6646 CDK5RAP2 Zornitza Stark Marked gene: CDK5RAP2 as ready
Mendeliome v0.6646 CDK5RAP2 Zornitza Stark Gene: cdk5rap2 has been classified as Green List (High Evidence).
Mendeliome v0.6646 CDK5RAP2 Zornitza Stark Phenotypes for gene: CDK5RAP2 were changed from to Microcephaly 3, primary, autosomal recessive, MIM# 604804; MONDO:0011488
Mendeliome v0.6645 CDK5RAP2 Zornitza Stark Publications for gene: CDK5RAP2 were set to
Mendeliome v0.6644 CDK5RAP2 Zornitza Stark Mode of inheritance for gene: CDK5RAP2 was changed from Unknown to BIALLELIC, autosomal or pseudoautosomal
Mendeliome v0.6643 CDK5RAP2 Zornitza Stark reviewed gene: CDK5RAP2: Rating: GREEN; Mode of pathogenicity: None; Publications: 15793586, 22887808, 23995685, 23726037, 27761245, 20460369, 32677750, 32015000; Phenotypes: Microcephaly 3, primary, autosomal recessive, MIM# 604804, MONDO:0011488; Mode of inheritance: BIALLELIC, autosomal or pseudoautosomal
Mendeliome v0.2641 CDK5 Zornitza Stark changed review comment from: Single consanguineous family with multiple affected individuals reported.; to: Single consanguineous family with multiple affected individuals reported.
Mendeliome v0.2641 CDK5 Zornitza Stark Marked gene: CDK5 as ready
Mendeliome v0.2641 CDK5 Zornitza Stark Gene: cdk5 has been classified as Red List (Low Evidence).
Mendeliome v0.2641 CDK5 Zornitza Stark Phenotypes for gene: CDK5 were changed from to Lissencephaly 7 with cerebellar hypoplasia, MIM# 616342
Mendeliome v0.2640 CDK5 Zornitza Stark Publications for gene: CDK5 were set to
Mendeliome v0.2639 CDK5 Zornitza Stark Mode of inheritance for gene: CDK5 was changed from Unknown to BIALLELIC, autosomal or pseudoautosomal
Mendeliome v0.2638 CDK5 Zornitza Stark Classified gene: CDK5 as Red List (low evidence)
Mendeliome v0.2638 CDK5 Zornitza Stark Gene: cdk5 has been classified as Red List (Low Evidence).
Mendeliome v0.2637 CDK5 Zornitza Stark reviewed gene: CDK5: Rating: RED; Mode of pathogenicity: None; Publications: 25560765; Phenotypes: Lissencephaly 7 with cerebellar hypoplasia, MIM# 616342; Mode of inheritance: BIALLELIC, autosomal or pseudoautosomal
Mendeliome v0.1133 CDK5R1 Zornitza Stark Marked gene: CDK5R1 as ready
Mendeliome v0.1133 CDK5R1 Zornitza Stark Gene: cdk5r1 has been classified as Red List (Low Evidence).
Mendeliome v0.1133 CDK5R1 Zornitza Stark Mode of inheritance for gene: CDK5R1 was changed from Unknown to MONOALLELIC, autosomal or pseudoautosomal, NOT imprinted
Mendeliome v0.1132 CDK5R1 Zornitza Stark Phenotypes for gene: CDK5R1 were changed from to Intellectual disability; autism
Mendeliome v0.1131 CDK5R1 Zornitza Stark Publications for gene: CDK5R1 were set to
Mendeliome v0.1130 CDK5R1 Zornitza Stark Classified gene: CDK5R1 as Red List (low evidence)
Mendeliome v0.1130 CDK5R1 Zornitza Stark Gene: cdk5r1 has been classified as Red List (Low Evidence).
Mendeliome v0.1129 CDK5R1 Zornitza Stark reviewed gene: CDK5R1: Rating: RED; Mode of pathogenicity: None; Publications: 30733659; Phenotypes: Intellectual disability, autism; Mode of inheritance: MONOALLELIC, autosomal or pseudoautosomal, imprinted status unknown
Mendeliome v0.0 CDK5RAP2 Zornitza Stark gene: CDK5RAP2 was added
gene: CDK5RAP2 was added to Mendeliome_VCGS. Sources: Expert Review Green,Victorian Clinical Genetics Services
Mode of inheritance for gene: CDK5RAP2 was set to Unknown
Mendeliome v0.0 CDK5R1 Zornitza Stark gene: CDK5R1 was added
gene: CDK5R1 was added to Mendeliome_VCGS. Sources: Expert Review Green,Victorian Clinical Genetics Services
Mode of inheritance for gene: CDK5R1 was set to Unknown
Mendeliome v0.0 CDK5 Zornitza Stark gene: CDK5 was added
gene: CDK5 was added to Mendeliome_VCGS. Sources: Expert Review Green,Victorian Clinical Genetics Services
Mode of inheritance for gene: CDK5 was set to Unknown