ATAD3B

Chr 1

ATPase family AAA domain containing 3B

Also known as: AAA-TOB3, TOB3

ATAD3B encodes a mitochondrial inner membrane protein that negatively regulates ATAD3A's interaction with matrix nucleoid complexes and may organize mitochondrial networks in stem cells by reducing mitochondrial metabolism and fragmenting the mitochondrial network. Mutations cause autosomal recessive mitochondrial disorders affecting multiple organ systems. The gene shows tolerance to loss-of-function variants in the general population (LOEUF 1.477), suggesting pathogenic variants may require specific functional consequences rather than simple protein loss.

Summary from RefSeq, UniProt
Research Assistant →
0
Active trials
3
Pubs (1 yr)
147
P/LP submissions
0%
P/LP missense
1.48
LOEUF
Multiple*
Mechanism· predicted
Clinical SummaryATAD3B
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
140 unique Pathogenic / Likely Pathogenic· 220 VUS of 427 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.48LOEUF
pLI 0.000
Z-score -0.50
OE 1.10 (0.831.48)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-2.79Z-score
OE missense 1.39 (1.301.49)
557 obs / 399.9 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE1.10 (0.831.48)
00.351.4
Missense OE1.39 (1.301.49)
00.61.4
Synonymous OE1.33
01.21.6
LoF obs/exp: 32 / 29.1Missense obs/exp: 557 / 399.9Syn Z: -3.46
DN
0.74top 25%
GOF
0.76top 25%
LOF
0.3357th %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
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

427 submitted variants in ClinVar

Classification Summary

Pathogenic131
Likely Pathogenic9
VUS220
Likely Benign20
Benign10
Conflicting3
131
Pathogenic
9
Likely Pathogenic
220
VUS
20
Likely Benign
10
Benign
3
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
131
0
131
Likely Pathogenic
0
0
9
0
9
VUS
1
185
34
0
220
Likely Benign
0
14
3
3
20
Benign
0
3
6
1
10
Conflicting
3
Total12021834393

LoF = frameshift, stop gained/lost, canonical splice · Counts from ClinVar esearch · Updated hourly

View in ClinVar →

Protein Context — Lollipop Plot

ATAD3B · 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.

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Clinical Literature
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