ISCU

Chr 12

iron-sulfur cluster assembly enzyme

Also known as: 2310020H20Rik, HML, ISU2, NIFU, NIFUN, hnifU

This gene encodes a component of the iron-sulfur (Fe-S) cluster scaffold. Fe-S clusters are cofactors that play a role in the function of a diverse set of enzymes, including those that regulate metabolism, iron homeostasis, and oxidative stress response. Alternative splicing results in transcript variants encoding different protein isoforms that localize either to the cytosol or to the mitochondrion. Mutations in this gene have been found in patients with hereditary myopathy with lactic acidosis. A disease-associated mutation in an intron may activate a cryptic splice site, resulting in the production of a splice variant encoding a putatively non-functional protein. A pseudogene of this gene is present on chromosome 1. [provided by RefSeq, Feb 2016]

ResearchGenerating clinical summary…

Primary Disease Associations & Inheritance

UniProtMyopathy with exercise intolerance Swedish type

Clinical highlights

Gene-disease validity (ClinGen)
mitochondrial disease · ARDefinitivesufficient evidence for diagnostic panels
0
Active trials
35
Pubs (1 yr)
P/LP submissions
P/LP missense
1.16
LOEUF
DN
Mechanism· predicted
Some data sources returned errors (1)

omim: Error: OMIM fetch failed: 429

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint?
1.16LOEUF
pLI 0.041
Z-score 1.32
OE 0.45 (0.201.16)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint?
0.21Z-score
OE missense 0.94 (0.781.13)
77 obs / 82.3 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios?
LoF OE?0.45 (0.201.16)
00.351.4
Missense OE?0.94 (0.781.13)
00.61.4
Synonymous OE?1.20
01.21.6
LoF obs/exp: 3 / 6.7Missense obs/exp: 77 / 82.3Syn Z: -0.93

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

ISCU · 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 →