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PI4K2A

Function

Membrane-bound phosphatidylinositol-4 kinase (PI4-kinase) that catalyzes the phosphorylation of phosphatidylinositol (PI) to phosphatidylinositol 4-phosphate (PI4P), a lipid that plays important roles in endocytosis, Golgi function, protein sorting and membrane trafficking and is required for prolonged survival of neurons. Besides, phosphorylation of phosphatidylinositol (PI) to phosphatidylinositol 4-phosphate (PI4P) is the first committed step in the generation of phosphatidylinositol 4,5-bisphosphate (PIP2), a precursor of the second messenger inositol 1,4,5-trisphosphate (InsP3).

Involvement in disease

Neurodevelopmental disorder with hyperkinetic movements, seizures, and structural brain abnormalities

NEDMSB

An autosomal recessive disorder characterized by failure to thrive, global developmental delay with intellectual disability and absent speech, seizures, hypotonia, inability to walk, orofacial dyskinesia, involuntary movements, and structural brain abnormalities.

None

The disease is caused by variants affecting the gene represented in this entry.

Post-translational modifications

Palmitoylated by ZDHHC3 and ZDHHC7 in the CCPCC motif. Palmitoylation is cholesterol-dependent, and required for TGN localization.

Ubiquitinated by ITCH; this does not lead to proteasomal degradation.

Sequence Similarities

Belongs to the PI3/PI4-kinase family. Type II PI4K subfamily.

Tissue Specificity

Widely expressed. Highest expression is observed in kidney, brain, heart, skeletal muscle, and placenta and lowest expression is observed in colon, thymus, and small intestine.

Cellular localization

Alternative names

Phosphatidylinositol 4-kinase type 2-alpha, Phosphatidylinositol 4-kinase type II-alpha, PI4K2A

swissprot:Q9BTU6 omim:609763 entrezGene:55361