rs1348967263

This variant is located in the TPP1 gene.

ClinVar annotation

Uncertain Significance
1 submitter1 publication

Neuronal ceroid lipofuscinosis 2

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Research that mentions this SNP (1)

Update of the mutation spectrum and clinical correlations of over 360 mutations in eight genes that underlie the neuronal ceroid lipofuscinoses
ReviewMaria Kousi et al.(2012)· Human Mutation

A comprehensive mutation update of neuronal ceroid lipofuscinoses (NCLs), cataloging 365 NCL-causing mutations across eight genes (PPT1/CLN1, TPP1/CLN2, CLN3, CLN5, CLN6, MFSD8/CLN7, CLN8, CTSD/CLN10), with 91 novel mutations reported. The review emphasizes complex genotype-phenotype correlations in these autosomal recessive neurodegenerative disorders and demonstrates how different mutations can cause phenotypic convergence or divergence, including variable disease severity.

Traits studied:Adult onset ceroid-lipofuscinosisBatten diseaseCLN1 diseaseCLN10 diseaseCLN2 diseaseCLN3 diseaseCLN5 diseaseCLN6 diseaseCLN7 diseaseCLN8 diseaseInfantile ceroid-lipofuscinosisJuvenile ceroid-lipofuscinosisLate infantile ceroid-lipofuscinosisNeuronal ceroid lipofuscinosisProgressive epilepsy with mental retardation

About TPP1

This gene encodes a member of the sedolisin family of serine proteases. The protease functions in the lysosome to cleave N-terminal tripeptides from substrates, and has weaker endopeptidase activity. It is synthesized as a catalytically-inactive enzyme which is activated and auto-proteolyzed upon acidification. Mutations in this gene result in late-infantile neuronal ceroid lipofuscinosis, which is associated with the failure to degrade specific neuropeptides and a subunit of ATP synthase in the lysosome. [provided by RefSeq, Jul 2008]

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Gene information from NCBI Gene. Variant classifications from ClinVar.

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