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Leukodystrophy v0.392 CNP Zornitza Stark Publications for gene: CNP were set to 32128616; 12590258
Leukodystrophy v0.391 CNP Zornitza Stark Classified gene: CNP as Green List (high evidence)
Leukodystrophy v0.391 CNP Zornitza Stark Gene: cnp has been classified as Green List (High Evidence).
Leukodystrophy v0.390 CNP Zornitza Stark edited their review of gene: CNP: Added comment: PMID 40396300 adds two affected siblings from an independent consanguineous family with a homozygous nonsense CNP variant (p.Glu99*) resulting in hypomyelinating leukodystrophy type 20.; Changed rating: GREEN; Changed publications: 40396300
Leukodystrophy v0.390 Zornitza Stark Added reviews for gene CNP from panel Mendeliome
Leukodystrophy v0.208 CNP Zornitza Stark Phenotypes for gene: CNP were changed from Hypomyelinating leukodystrophy to Leukodystrophy, hypomyelinating, 20, MIM# 619071
Leukodystrophy v0.207 CNP Zornitza Stark reviewed gene: CNP: Rating: AMBER; Mode of pathogenicity: None; Publications: ; Phenotypes: Leukodystrophy, hypomyelinating, 20, MIM# 619071; Mode of inheritance: BIALLELIC, autosomal or pseudoautosomal
Leukodystrophy v0.127 CNP Zornitza Stark Marked gene: CNP as ready
Leukodystrophy v0.127 CNP Zornitza Stark Gene: cnp has been classified as Amber List (Moderate Evidence).
Leukodystrophy v0.127 CNP Zornitza Stark Classified gene: CNP as Amber List (moderate evidence)
Leukodystrophy v0.127 CNP Zornitza Stark Gene: cnp has been classified as Amber List (Moderate Evidence).
Leukodystrophy v0.126 CNP Kristin Rigbye gene: CNP was added
gene: CNP was added to Leukodystrophy - paediatric. Sources: Literature
Mode of inheritance for gene: CNP was set to BIALLELIC, autosomal or pseudoautosomal
Publications for gene: CNP were set to 32128616; 12590258
Phenotypes for gene: CNP were set to Hypomyelinating leukodystrophy
Review for gene: CNP was set to AMBER
Added comment: Single consanguineous family described with homozygous missense in affected child (additional two affected deceased offspring unavailable for testing; healthy carrier parents and sibling).
Loss of protein by Western blot and defect in F-actin structure and organization observed in patient fibroblasts.
Deficiency of CNP in mouse has previously been shown to cause a lethal white matter neurodegenerative phenotype (PMID: 12590258), similar to the phenotype observed in this family.
Sources: Literature