Limb girdle muscular dystrophies, myofibrillar myopathies and distal myopathies

Gene: GUK1

Amber List (moderate evidence)

GUK1 (guanylate kinase 1)
EnsemblGeneIds (GRCh38): ENSG00000143774
EnsemblGeneIds (GRCh37): ENSG00000143774
OMIM: 139270, Gene2Phenotype
GUK1 is in 4 panels

2 reviews

Arina Puzriakova (Genomics England Curator)

Comment on list classification: There is sufficient evidence to promote this gene to Green at the next GMS panel update - 4 individuals from 3 unrelated families with biallelic variants leading to GUK1 deficiency. Muscle biopsies showed mtDNA depletion and/or deletions and reduced activities of mitochondrial respiratory chain enzymes.
Created: 29 Aug 2025, 4:03 p.m. | Last Modified: 29 Aug 2025, 4:03 p.m.
Panel Version: 9.26

Hannah Knight (NIHR BioResource - University of Cambridge)

Green List (high evidence)

Four patients from three families reported with mitochondrial disorders and biallelic GUK1 variants. Different variants in all three families.
Key symptoms include ptosis, ophthalmoparesis, and myopathic proximal limb weakness, as well as variable hepatopathy
Sources: Literature
Created: 22 Jul 2025, 9:07 a.m.

Mode of inheritance
BIALLELIC, autosomal or pseudoautosomal

Phenotypes
Mitochondrial DNA depletion syndrome 21

Publications

Details

Mode of Inheritance
BIALLELIC, autosomal or pseudoautosomal
Sources
  • Expert Review Amber
  • Literature
Phenotypes
  • Mitochondrial DNA depletion syndrome 21, OMIM:621071
Tags
Q3_25_promote_green
OMIM
139270
Clinvar variants
Variants in GUK1
Penetrance
None
Publications
Panels with this gene

History Filter Activity

29 Aug 2025, Gel status: 2

Removed Tag

Arina Puzriakova (Genomics England Curator)

Tag Q3_25_NHS_review was removed from gene: GUK1.

29 Aug 2025, Gel status: 2

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

Arina Puzriakova (Genomics England Curator)

gene: GUK1 was added gene: GUK1 was added to Limb girdle muscular dystrophies, myofibrillar myopathies and distal myopathies. Sources: Literature,Expert Review Amber Q3_25_promote_green, Q3_25_NHS_review tags were added to gene: GUK1. Mode of inheritance for gene: GUK1 was set to BIALLELIC, autosomal or pseudoautosomal Publications for gene: GUK1 were set to 39230499 Phenotypes for gene: GUK1 were set to Mitochondrial DNA depletion syndrome 21, OMIM:621071