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Genetic Epilepsy

Gene: COQ5

Green List (high evidence)

COQ5 (coenzyme Q5, methyltransferase)
EnsemblGeneIds (GRCh38): ENSG00000110871
EnsemblGeneIds (GRCh37): ENSG00000110871
OMIM: 616359, ClinGen, DECIPHER
COQ5 is in 5 panels

1 review

Lucy Spencer (Victorian Clinical Genetics Services)

Green List (high evidence)

PMID: 29044765 3 siblings with cerebellar ataxia, encephalopathy, seizures, developmental delay, and oculomotor apraxia or nystagmus.. All homozygous for a tandem duplication of 9.59kb encompassing the last 4 exons. Parents both shown to be carriers and 3 siblings were either het or did not have the dup. qRT-PCR in patient fibroblasts showed COQ5 mRNA levels were at 2-4% compared to controls. Reduced COQ5 protein was also shown on western blots.

PMID: 37599337 1 patient compound heterozygous for c.681+1G>A and Gly118Ser. Cerebellar ataxia, encephalopathy, developmental delay, short stature, and microcephaly. COQ5 mRNA levels were markedly diminished in patient cells, and different mis-splicing events were seen on both alleles (confirmed in carrier parents). This paper also referenced another patient in PMID: 21937992, a large ID cohort with limited phenotypic information who was homozygous for the same missense variant in their patient Gly118Ser.

PMID: 41199775 2 siblings with a different missense at the recurring amino acid Gly118, Gly118Asp, in trans with an NMD predicted frameshift. Both siblings had severe developmental delay, microcephaly, spasticity, ataxia, seizures, and feeding difficulties. Recurrent strokes and other brain anomalies commonly seen in mitochondrial disorders were seen on brain MRI, but there was no cerebellar atrophy. 1 sibling also had retinitis pigmentosa. Yeast modelling of this missense variant showed yeast harboring p.Gly118Asp (yeast equivalent) had decreased CoQ6 levels and an accumulation of the COQ5 yeast-equivalent substrate, DDMQ suggesting LOF.

PMID: 36266294 2 probands with retinitis pigmentosa from 2 families, 1 individual had symptoms onset at 49 years old with no other features while the 2nd had onset at 5 years old and also had muscular weakness and hypertelorism. Both patients had COQ5 c.682-7T>G which has 285 hets but no homs in gnomad v4. RT-PCR on patient cells showed majority exon 5 skipping, and minor exon 4+5 skipping, both out of frame and predicted to cause NMD. The major splicing event was seen in 56% of transcripts for that allele, so normal splicing is still occurring and its not a complete LOF allele. Both patients had different 2nd hits, Tyr311* and Arg123Trp.

The two patients in PMID: 36266294 are phenotypic outliers, PMID: 41199775 suggests you need a mutation affecting Gly118 to cause severe multisystem disease. Gly118 is in a might conserved DVAGGSG motif which is essential for binding S-adenosylmethionine. All other cases have a missense at this residue except for the family with the large duplication.
Created: 26 Nov 2025, 2:31 p.m. | Last Modified: 26 Nov 2025, 2:31 p.m.
Panel Version: 1.3660

Mode of inheritance
BIALLELIC, autosomal or pseudoautosomal

Phenotypes
Coenzyme Q10 deficiency, primary, 9 MIM#619028

Publications

Details

Mode of Inheritance
BIALLELIC, autosomal or pseudoautosomal
Sources
  • Expert Review Green
  • Victorian Clinical Genetics Services
  • Expert list
Phenotypes
  • Coenzyme Q10 deficiency, primary 9, MIM#619028
OMIM
616359
ClinGen
COQ5
DECIPHER
COQ5
Clinvar variants
Variants in COQ5
Penetrance
None
Publications
Panels with this gene

History Filter Activity

26 Nov 2025, Gel status: 3

Entity classified by Genomics England curator

Zornitza Stark (Victorian Clinical Genetics Services)

Gene: coq5 has been classified as Green List (High Evidence).

26 Nov 2025, Gel status: 3

Created, Added New Source, Set mode of inheritance, Set publications, Set Phenotypes

Zornitza Stark (Victorian Clinical Genetics Services)

gene: COQ5 was added gene: COQ5 was added to Genetic Epilepsy. Sources: Expert Review Green,Expert list,Victorian Clinical Genetics Services Mode of inheritance for gene: COQ5 was set to BIALLELIC, autosomal or pseudoautosomal Publications for gene: COQ5 were set to 29044765; 37599337; 21937992; 41199775; 36266294 Phenotypes for gene: COQ5 were set to Coenzyme Q10 deficiency, primary 9, MIM#619028