MTM1

Chr XXLR

myotubularin 1

Also known as: CNM, CNMX, MTMX, XLMTM

This gene encodes a dual-specificity phosphatase that dephosphorylates both phosphotyrosine and phosphoserine residues and is required for muscle cell differentiation. Mutations cause X-linked myotubular myopathy (also called X-linked centronuclear myopathy), inherited in an X-linked recessive pattern. The pathogenic mechanism involves disrupted muscle fiber maturation and organization due to loss of phosphatase function.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismXLRLOEUF 0.181 OMIM phenotype
VCEP Guidelines: Congenital MyopathiesReleased
ClinGen Panel
Clinical SummaryMTM1
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Gene-Disease Validity (ClinGen)
X-linked myotubular myopathy · XLDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 1.00). One damaged copy is likely sufficient to cause disease.
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Clinical Trials
4 active or recruiting trials — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.18LOEUF
pLI 1.000
Z-score 4.63
OE 0.04 (0.010.18)
Highly constrained

Highly LoF-intolerant (top ~10% of genes)

Missense Constraint
2.39Z-score
OE missense 0.57 (0.490.65)
138 obs / 243.1 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.04 (0.010.18)
00.351.4
Missense OE0.57 (0.490.65)
00.61.4
Synonymous OE0.77
01.21.6
LoF obs/exp: 1 / 26.9Missense obs/exp: 138 / 243.1Syn Z: 1.65

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

MTM1 · protein map & ClinVar variants

Showing all ClinVar variants across the protein. Search a specific variant to highlight its position.

3D Protein StructureAlphaFold
Clinical Literature
Open Research Assistant →
Key Publications
Landmark & review papers · by relevance
PubMed
Top 5 results · since 2015Search PubMed ↗