PGM1

Chr 1AR

phosphoglucomutase 1

Also known as: CDG1T, GSD14

Phosphoglucomutase 1 catalyzes the reversible conversion of glucose 1-phosphate to glucose 6-phosphate, participating in both glucose breakdown and synthesis and representing about 90% of total phosphoglucomutase activity in most cell types. Mutations cause congenital disorder of glycosylation type It, which is inherited in an autosomal recessive pattern. This gene shows tolerance to loss-of-function variants (LOEUF 1.319), suggesting that complete loss of function may be compatible with survival when biallelic mutations occur.

Summary from RefSeq, OMIM, UniProt
Research Assistant →

Primary Disease Associations & Inheritance

Congenital disorder of glycosylation, type ItMIM #614921
AR
0
Active trials
33
Pubs (1 yr)
77
P/LP submissions
10%
P/LP missense
1.32
LOEUF
LOF
Mechanism· G2P
Clinical SummaryPGM1
🧬
Gene-Disease Validity (ClinGen)
PGM1-congenital disorder of glycosylation · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
49 unique Pathogenic / Likely Pathogenic· 191 VUS of 500 total submissions
📖
GeneReview available — PGM1
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.32LOEUF
pLI 0.000
Z-score 0.32
OE 0.93 (0.671.32)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.23Z-score
OE missense 1.04 (0.951.13)
336 obs / 324.5 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.93 (0.671.32)
00.351.4
Missense OE1.04 (0.951.13)
00.61.4
Synonymous OE1.16
01.21.6
LoF obs/exp: 23 / 24.7Missense obs/exp: 336 / 324.5Syn Z: -1.36
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitivePGM1-related congenital disorder of glycosylationLOFAR

Predictions shown for reference only — model trained on dominant genes, not applicable to AR conditions.

DN
0.6552th %ile
GOF
0.4481th %ile
LOF
0.3744th %ile

The Badonyi & Marsh prediction model was trained exclusively on dominant disease genes. Predictions are not reliable for genes with autosomal recessive inheritance and are shown at reduced opacity for reference only.

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312.

ClinVar Variant Classifications

500 submitted variants in ClinVar

Classification Summary

Pathogenic35
Likely Pathogenic14
VUS191
Likely Benign182
Benign47
Conflicting12
35
Pathogenic
14
Likely Pathogenic
191
VUS
182
Likely Benign
47
Benign
12
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
15
0
20
0
35
Likely Pathogenic
3
5
6
0
14
VUS
0
162
26
3
191
Likely Benign
1
1
76
104
182
Benign
0
1
45
1
47
Conflicting
12
Total19169173108481

LoF = frameshift, stop gained/lost, canonical splice · Counts from ClinVar esearch · Updated hourly

View in ClinVar →

Protein Context — Lollipop Plot

PGM1 · protein map & ClinVar variants

Showing all ClinVar variants across the protein. Search a specific variant to highlight its position.

Clinical Trials

Active and recruiting trials from ClinicalTrials.gov

No active trials found for this gene.

Search ClinicalTrials.gov →
Clinical Literature
Open Research Assistant →
Key Publications
Landmark & review papers · by relevance
PubMed
Liver glucose metabolism in humans.
Adeva-Andany MM et al.·Biosci Rep
2016Review
Congenital Hyperinsulinism: Diagnosis and Treatment Update.
Demirbilek H et al.·J Clin Res Pediatr Endocrinol
2017Review
Central nervous involvement is common in PGM1-CDG.
Radenkovic S et al.·Mol Genet Metab
2018Review
Top 5 results · since 2015Search PubMed ↗