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DOT1L

Function

Histone methyltransferase that methylates 'Lys-79' of histone H3 (PubMed:12123582, PubMed:38503750, PubMed:38129415). Nucleosomes are preferred as substrate compared to free histones (PubMed:12123582, PubMed:38503750). Binds to DNA (PubMed:12628190, PubMed:38503750).

Involvement in disease

Nil-Deshwar neurodevelopmental syndrome

NDNS

An autosomal dominant neurodevelopmental disorder characterized by global developmental delay, impaired intellectual development, poor or absent speech, hypotonia, and various heterogeneous congenital anomalies, including brain, cardiac and urogenital abnormalities.

None

The disease is caused by variants affecting the gene represented in this entry. Disease-causing variants may act through a gain-of-function effect leading to an increase in cellular H3K79 methylation levels.

Sequence Similarities

Belongs to the class I-like SAM-binding methyltransferase superfamily. DOT1 family.

Cellular localization

Alternative names

KIAA1814, KMT4, DOT1L, DOT1-like protein, Disruptor of telomeric silencing 1-like histone lysine methyltransferase, Histone H3-K79 methyltransferase, Lysine N-methyltransferase 4, H3-K79-HMTase

swissprot:Q8TEK3 entrezGene:84444 omim:607375