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PMM2
phosphomannomutase 2
The PMM2 gene provides instructions for producing the phosphomannomutase 2 enzyme, which plays a critical role in glycosylation, a vital biological process for modifying proteins. PMM2 is integral to the initial steps of glycosylation, a process that attaches sugar molecules to proteins, enabling them to perform diverse functions within the body.
PMM2 is located on the short (p) arm of chromosome 16, at band 16p13.2. Arm ratio per GRCh38 - banding schematic.
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Overview
The PMM2 gene, also known as phosphomannomutase 2, is crucial for human health, primarily due to its involvement in glycosylation. This fundamental biological process involves the attachment of sugar molecules, known as oligosaccharides, to proteins. Proper glycosylation is essential for many proteins to function correctly and for overall cellular health. Dysregulation of this process, often due to variants in genes like PMM2, can lead to a range of inherited disorders.
What the gene does
The PMM2 gene encodes the enzyme phosphomannomutase 2, which acts as a catalyst in a specific step of the glycosylation pathway. This enzyme facilitates the conversion of mannose-6-phosphate to mannose-1-phosphate. Subsequently, mannose-1-phosphate is transformed into GDP-mannose. This GDP-mannose then serves as a donor molecule, transferring a mannose sugar unit to a growing oligosaccharide chain. Once the oligosaccharide reaches its correct size and structure, it can be attached to a protein, enabling the protein to perform its specialised biological roles. This modification is critical for a wide variety of protein functions across different physiological systems.
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Chromosome location
The PMM2 gene is located on chromosome 16, specifically at position 16p13.2. This location refers to the short (p) arm of chromosome 16, within region 13 and band 2. The precise genomic coordinates help in identifying the gene's exact position within the human genome.
Protein structure
Domain architecture has not been experimentally characterised in detail for this protein.
Key variants
Genetic variations within the PMM2 gene can alter the function of the phosphomannomutase 2 enzyme. These changes can range from single nucleotide substitutions to larger deletions or insertions, impacting the enzyme's ability to participate in glycosylation. Over 115 distinct pathogenic variants have been identified that are associated with clinical conditions.
Sample of pathogenic variants
10 pathogenic / likely-pathogenic variants from ClinVar, ranked by review status (expert-panel-reviewed first). This is a sample; recurrent founder variants in a specific population may not appear here - see the full ClinVar listing via the link above.
| Variant (HGVS) | Protein change | Classification | Evidence | Associated condition |
|---|---|---|---|---|
c.179-1G>C | - | Pathogenic/Likely pathogenic | ★★☆☆ | PMM2-congenital disorder of glycosylation |
c.193G>A | p.Asp65Asn | Pathogenic/Likely pathogenic | ★★☆☆ | PMM2-congenital disorder of glycosylation |
c.253C>T | p.Gln85Ter | Pathogenic/Likely pathogenic | ★★☆☆ | PMM2-congenital disorder of glycosylation |
c.34_36delinsTAA | p.Asp12Ter | Pathogenic/Likely pathogenic | ★★☆☆ | PMM2-congenital disorder of glycosylation |
c.385G>C | p.Val129Leu | Pathogenic/Likely pathogenic | ★★☆☆ | PMM2-congenital disorder of glycosylation |
c.64C>T | p.Gln22Ter | Pathogenic | ★★☆☆ | PMM2-congenital disorder of glycosylation |
c.667G>A | p.Asp223Asn | Pathogenic/Likely pathogenic | ★★☆☆ | PMM2-congenital disorder of glycosylation |
c.669C>A | p.Asp223Glu | Pathogenic/Likely pathogenic | ★★☆☆ | PMM2-congenital disorder of glycosylation |
c.715A>T | p.Arg239Trp | Pathogenic | ★★☆☆ | PMM2-congenital disorder of glycosylation |
c.79G>T | p.Glu27Ter | Pathogenic/Likely pathogenic | ★★☆☆ | PMM2-congenital disorder of glycosylation |
Evidence stars indicate ClinVar review status. Individual variant interpretation should always be performed by a qualified clinical laboratory - many variants remain classified as Variants of Uncertain Significance (VUS) pending more research.
Associated conditions
Variants in the PMM2 gene are primarily associated with Congenital disorder of glycosylation Ia, also known as PMM2-CDG. This is an autosomal recessive condition, meaning an individual must inherit two copies of a pathogenic variant (one from each parent) to develop the disorder. It is characterised by a range of symptoms including developmental delay, reduced muscle tone, and an unusual fat distribution.
Inheritance pattern
Conditions caused by pathogenic PMM2 variants typically follow autosomal recessive inheritance.
When both parents are carriers, each child has a 25% chance of being affected, 50% of being a carrier, and 25% of being unaffected.
UK clinical status
The PMM2 gene is included in several NHS England Genomic Medicine Service clinical panels, indicating its recognised clinical significance within the UK healthcare system. These panels include 'Congenital disorders of glycosylation', 'Congenital hyperinsulinism', 'Intellectual disability', and 'Undiagnosed metabolic disorders', among others, all listed with a 'green' status, signifying that variants in PMM2 are considered causally associated with these conditions.
Sources: NHS GMS PanelApp · Genomics England PanelApp · NHS National Genomic Test Directory
Frequently asked questions
What is the role of the PMM2 gene?
The PMM2 gene provides instructions for the phosphomannomutase 2 enzyme, which is vital for glycosylation. This enzyme helps attach sugar molecules to proteins, a process essential for proteins to function correctly throughout the body.
What condition is associated with PMM2 gene variants?
Pathogenic variants in the PMM2 gene are primarily associated with Congenital disorder of glycosylation Ia (PMM2-CDG). This is an inherited condition affecting development and various bodily functions.
How is Congenital disorder of glycosylation Ia inherited?
Congenital disorder of glycosylation Ia follows an autosomal recessive inheritance pattern. This means an individual must inherit two copies of a pathogenic PMM2 variant, one from each parent, to develop the condition.
References
- de la Morena-Barrio ME, Hernández-Caselles T, Corral J. GPI-anchor and GPI-anchored protein expression in PMM2-CDG patients. Orphanet journal of rare diseases. 2013. PMID: 24139637
- Grünewald S. The clinical spectrum of phosphomannomutase 2 deficiency (CDG-Ia). Biochimica et biophysica acta. 2009. PMID: 19272306
- Freeze HH. Towards a therapy for phosphomannomutase 2 deficiency, the defect in CDG-Ia patients. Biochimica et biophysica acta. 2009. PMID: 19339218
- Haeuptle MA, Hennet T. Congenital disorders of glycosylation: an update on defects affecting the biosynthesis of dolichol-linked oligosaccharides. Human mutation. 2009. PMID: 19862844
- Gao N, Shang J, Lehrman MA. Analysis of glycosylation in CDG-Ia fibroblasts by fluorophore-assisted carbohydrate electrophoresis: implications for extracellular glucose and intracellular mannose 6-phosphate. The Journal of biological chemistry. 2005. PMID: 15708848
- Adam MP, Bick S, Mirzaa GM. PMM2-CDG. 1993. PMID: 20301289