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Function

3'-5' RNA exonuclease that trims the 3' end of oligo(U) and oligo(A) tracts of the pre-U6 small nuclear RNA (snRNA) molecule, leading to the formation of a mature U6 snRNA 3' end-terminated with a 2',3'-cyclic phosphate (PubMed:22899009, PubMed:23022480, PubMed:23190533, PubMed:26213367, PubMed:28887445, PubMed:30215753, PubMed:31832688). Participates in the U6 snRNA 3' end processing that prevents U6 snRNA degradation (PubMed:22899009, PubMed:23022480, PubMed:23190533, PubMed:26213367, PubMed:28887445, PubMed:30215753, PubMed:31832688). In addition also removes uridines from the 3' end of U6atac snRNA and possibly the vault RNA VTRNA1-1 (PubMed:26213367).

Involvement in disease

Poikiloderma with neutropenia

PN

A genodermatosis characterized by poikiloderma, pachyonychia and chronic neutropenia. The disorder starts as a papular erythematous rash on the limbs during the first year of life. It gradually spreads centripetally and, as the papular rash resolves, hypo- and hyperpigmentation result, with development of telangiectasias. Another skin manifestation is pachyonychia, but alopecia and leukoplakia are distinctively absent. Patients have recurrent pneumonias that usually result in reactive airway disease and/or chronic cough. One of the most important extracutaneous symptoms is an increased susceptibility to infections, mainly affecting the respiratory system, primarily due to a chronic neutropenia and to neutrophil functional defects. Bone marrow abnormalities account for neutropenia and may evolve into myelodysplasia associated with the risk of leukemic transformation. Poikiloderma with neutropenia shows phenotypic overlap with Rothmund-Thomson syndrome.

None

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

Sequence similarities

Belongs to the 2H phosphoesterase superfamily. USB1 family.

Cellular localization

  • Nucleus

Alternative names

C16orf57, Mpn1, USB1, U6 snRNA phosphodiesterase 1, hUsb1, 3'-5' RNA exonuclease USB1, Mutated in poikiloderma with neutropenia protein 1, Mutated in PN protein 1, hMpn1

Target type

Proteins

Molecular weight

30268Da

We found 1 product in 1 category

Primary Antibodies

Target

Application

Reactive species

Search our catalogue for 'C16orf57' (1)

Products