ANO5

Chr 11ADAR

anoctamin 5

Also known as: GDD1, LGMD2L, LGMDR12, TMEM16E

The protein functions in plasma membrane repair through interaction with annexins and localizes to the endoplasmic reticulum membrane. Mutations cause autosomal recessive limb-girdle muscular dystrophy type 12 and Miyoshi muscular dystrophy 3, as well as autosomal dominant gnathodiaphyseal dysplasia through a predicted gain-of-function mechanism. The muscular dystrophies result from impaired membrane repair, while the bone dysplasia represents a distinct dominant phenotype.

OMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
MultiplemechanismAD/ARLOEUF 0.953 OMIM phenotypes
VCEP Guidelines: Limb Girdle Muscular DystrophyReleased
ClinGen Panel
Clinical SummaryANO5
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Gene-Disease Validity (ClinGen)
gnathodiaphyseal dysplasia · ADDefinitive

Definitive — sufficient evidence for diagnostic panels

2 total gene-disease associations curated

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
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Clinical Trials
2 active or recruiting trials — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.95LOEUF
pLI 0.000
Z-score 1.86
OE 0.73 (0.570.95)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.12Z-score
OE missense 0.98 (0.911.06)
467 obs / 474.2 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.73 (0.570.95)
00.351.4
Missense OE0.98 (0.911.06)
00.61.4
Synonymous OE1.18
01.21.6
LoF obs/exp: 42 / 57.2Missense obs/exp: 467 / 474.2Syn Z: -1.80
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
moderateANO5-related gnathodiaphyseal dysplasiaGOFAD
limitedANO5-related limb-girdle muscular dystrophyLOFAR
DN
0.7228th %ile
GOF
0.76top 25%
LOF
0.2385th %ile

This gene has evidence for multiple mechanisms of pathogenicity (gain-of-function and dominant-negative). Both the Badonyi & Marsh prediction and the broader genomic evidence point to gain-of-function as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

GOFprediction above median · 1 literature citation
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.

Literature Evidence

GOFGain of function of TMEM16E/ANO5 scrambling activity caused by a mutation associated with gnathodiaphyseal dysplasia.PMID:29124309

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

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

ANO5 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
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Full-Text Mentions
NLP-detected gene mentions in article bodies · via PubTator3
PubTator3
Top 5 full-text resultsSearch PubTator3 ↗