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Lysosomal Storage Disorder v1.21 SPNS1 Sangavi Sivagnanasundram gene: SPNS1 was added
gene: SPNS1 was added to Lysosomal Storage Disorder. Sources: Literature
Mode of inheritance for gene: SPNS1 was set to BIALLELIC, autosomal or pseudoautosomal
Publications for gene: SPNS1 were set to 40608416
Phenotypes for gene: SPNS1 were set to Lysosomal disorder, SPNS1-related, MONDO:0002561
Review for gene: SPNS1 was set to AMBER
Added comment: Patient 1 and 2 - Proband and younger brother prolonged, transient neonatal unconjugated hyperbilirubinemia followed by persistently elevated transaminases, serum creatine kinase and myoglobin levels since 6 months and 12 months of age
Compound het - Ser416Cys; Ile50Alafs*48 confirmed in trans - both absent from gnomAD v4.1

Patient 3 - 8M from consanguineous parents with elevated transaminase and failure to thrive at 2.5years. Liver transaminase, serum creatinine kinase were elevated. Diagnosed with DD and presented with neonatal cardiac abnormalities
Homozygous variant - Thr287Met - NFE PopMax AF 0.0008474%

Supportive functional assay conducted on patient fibroblasts showed accumulated lysophospholipids including lysoplasmalogens and cholesterol in lysosomes with reduced cellular plasmalogens.
Sources: Literature
Lysosomal Storage Disorder v1.9 IDS Zornitza Stark Tag treatable tag was added to gene: IDS.
Lysosomal Storage Disorder v1.9 IDS Zornitza Stark Mode of inheritance for gene: IDS was changed from X-LINKED: hemizygous mutation in males, biallelic mutations in females to X-LINKED: hemizygous mutation in males, biallelic mutations in females
Lysosomal Storage Disorder v1.8 IDS Zornitza Stark Mode of inheritance for gene: IDS was changed from BIALLELIC, autosomal or pseudoautosomal to X-LINKED: hemizygous mutation in males, biallelic mutations in females
Lysosomal Storage Disorder v1.7 IDS Zornitza Stark edited their review of gene: IDS: Changed mode of inheritance: X-LINKED: hemizygous mutation in males, biallelic mutations in females
Lysosomal Storage Disorder v0.138 IDS Zornitza Stark Marked gene: IDS as ready
Lysosomal Storage Disorder v0.138 IDS Zornitza Stark Gene: ids has been classified as Green List (High Evidence).
Lysosomal Storage Disorder v0.138 IDS Zornitza Stark Phenotypes for gene: IDS were changed from to Mucopolysaccharidosis II, MIM# 309900; MONDO:0010674; Hunter syndrome
Lysosomal Storage Disorder v0.137 IDS Zornitza Stark Publications for gene: IDS were set to
Lysosomal Storage Disorder v0.136 IDS Zornitza Stark Mode of inheritance for gene: IDS was changed from Unknown to BIALLELIC, autosomal or pseudoautosomal
Lysosomal Storage Disorder v0.135 IDS Zornitza Stark reviewed gene: IDS: Rating: GREEN; Mode of pathogenicity: None; Publications: 9921913, 9762601, 8940265, 1901826; Phenotypes: Mucopolysaccharidosis II, MIM# 309900, MONDO:0010674, Hunter syndrome; Mode of inheritance: BIALLELIC, autosomal or pseudoautosomal
Lysosomal Storage Disorder v0.0 IDS Zornitza Stark gene: IDS was added
gene: IDS was added to Storage Disorder_VCGS. Sources: Expert Review Green,Victorian Clinical Genetics Services
Mode of inheritance for gene: IDS was set to Unknown