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Hereditary neuropathy v1.497 ARHGAP19 Shahryar Alavi gene: ARHGAP19 was added
gene: ARHGAP19 was added to Hereditary neuropathy. Sources: Research
Mode of inheritance for gene: ARHGAP19 was set to BIALLELIC, autosomal or pseudoautosomal
Publications for gene: ARHGAP19 were set to https://www.medrxiv.org/content/10.1101/2024.05.10.24306768v1
Phenotypes for gene: ARHGAP19 were set to neuropathy; Charcot-Marie-Tooth disease
Penetrance for gene: ARHGAP19 were set to Complete
Mode of pathogenicity for gene: ARHGAP19 was set to Other
Review for gene: ARHGAP19 was set to GREEN
Added comment: A comprehensive study on loss of function variants of the ARHGAP19 gene from several unrelated families shows that homozygous mutations cause early-onset motor-predominant neuropathy.
Biochemical assays revealed that GAP domain variants cause loss of protein function. Fruit fly and Zebrafish loss of function models also showed movement deficits.
RNA-Seq analysis confirmed downregulation of ARHGAP19 as well as cell cycle, motor and muscular cytoskeleton pathways in patients compared to controls.
The paper is under review.
Sources: Research
Hereditary neuropathy v1.63 BRAF Louise Daugherty reviewed gene: BRAF: Rating: AMBER; Mode of pathogenicity: ; Publications: ; Phenotypes: ; Mode of inheritance:
Hereditary neuropathy v1.60 BRAF Louise Daugherty Added phenotypes Cardiomyopathy for gene: BRAF
Hereditary neuropathy v1.58 BRAF Natalie Forrester reviewed gene: BRAF: Rating: RED; Mode of pathogenicity: ; Publications: ; Phenotypes: Cardiomyopathy; Mode of inheritance:
Hereditary neuropathy v1.57 BRAF Louise Daugherty Source NHS GMS was added to BRAF.
Hereditary neuropathy v1.56 BRAF Louise Daugherty Source South West GLH was added to BRAF.