MROH1

Chr 8

maestro heat like repeat family member 1

Also known as: HEATR7A

MROH1 encodes a lysosome fission factor that mediates the constriction and scission of lysosomal tubules by homooligomerizing at scission sites, which is essential for maintaining lysosome number, shape, size, and function. The gene is highly constrained against loss-of-function variants (LOEUF 0.486), suggesting that mutations would likely cause severe disease, though specific associated disorders have not yet been established in the literature. Given the critical role of lysosomes in cellular function, mutations would be expected to follow an autosomal recessive inheritance pattern and potentially affect multiple organ systems.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
3
Pubs (1 yr)
0
P/LP submissions
P/LP missense
0.49
LOEUF
DN
Mechanism· predicted
Clinical SummaryMROH1
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.25) despite low pLI — interpret in context.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.49LOEUF
pLI 0.203
Z-score 3.45
OE 0.25 (0.130.49)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
0.60Z-score
OE missense 0.90 (0.811.00)
252 obs / 280.2 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.25 (0.130.49)
00.351.4
Missense OE0.90 (0.811.00)
00.61.4
Synonymous OE1.10
01.21.6
LoF obs/exp: 6 / 24.3Missense obs/exp: 252 / 280.2Syn Z: -0.86
DN
0.6552th %ile
GOF
0.6151th %ile
LOF
0.3261th %ile

The highest-scoring mechanism for this gene is dominant-negative.

DNprediction above median

Note: In-silico variant effect predictors (SIFT, PolyPhen, REVEL, CADD) may underestimate pathogenicity of missense variants in genes with GOF or DN mechanisms. Consider functional evidence and clinical context.

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

MROH1 · 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 →