FOXE1

Chr 9ADAR

forkhead box E1

Also known as: BAMLAZ, FKHL15, FOXE2, HFKH4, HFKL5, NMTC4, TITF2, TTF-2

FOXE1 encodes a forkhead transcription factor that binds gene promoters to regulate thyroid morphogenesis and palate formation through direct targets including MSX1 and TGFB3. Loss-of-function mutations cause Bamforth-Lazarus syndrome (autosomal recessive) and increase susceptibility to nonmedullary thyroid cancer (autosomal dominant). The syndrome results from disrupted thyroid development and palatal morphogenesis due to impaired transcriptional regulation of key developmental genes.

OMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
LOFmechanismAD/ARLOEUF 0.582 OMIM phenotypes
Clinical SummaryFOXE1
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Gene-Disease Validity (ClinGen)
Bamforth-Lazarus syndrome · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Moderately constrained gene (pLI 0.79) — some intolerance to loss-of-function variants.
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ClinVar Variants
38 unique Pathogenic / Likely Pathogenic· 75 VUS of 151 total submissions
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Clinical Trials
1 active or recruiting trial — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.58LOEUF
pLI 0.792
Z-score 2.11
OE 0.00 (0.000.58)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
0.31Z-score
OE missense 0.93 (0.811.07)
149 obs / 160.2 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.00 (0.000.58)
00.351.4
Missense OE0.93 (0.811.07)
00.61.4
Synonymous OE0.95
01.21.6
LoF obs/exp: 0 / 5.2Missense obs/exp: 149 / 160.2Syn Z: 0.33
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveFOXE1-related Bamforth-Lazarus syndromeLOFAR

Predictions shown for reference only — model trained on dominant genes, not applicable to AR conditions.

DN
0.5180th %ile
GOF
0.3788th %ile
LOF
0.70top 10%

The Badonyi & Marsh prediction model was trained exclusively on dominant disease genes. Predictions are not reliable for genes with autosomal recessive inheritance and are shown at reduced opacity for reference only.

Literature Evidence

GOFThe gain-of-function effect of the FOXE1-p.R73S mutant gene was confirmed by an increase in endogenous TG production in primary human thyrocytes.CONCLUSION: We identified a new homozygous FOXE1 mutation responsible for enhanced expression of the TG and TPO genes in a boy whose phenotype is similar tPMID:24219130

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

ClinVar Variant Classifications

151 submitted variants in ClinVar

Classification Summary

Pathogenic32
Likely Pathogenic6
VUS75
Likely Benign14
Benign23
Conflicting1
32
Pathogenic
6
Likely Pathogenic
75
VUS
14
Likely Benign
23
Benign
1
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
1
3
28
0
32
Likely Pathogenic
0
2
4
0
6
VUS
1
71
3
0
75
Likely Benign
0
2
2
10
14
Benign
0
4
12
7
23
Conflicting
1
Total2824917151

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

View in ClinVar →

Protein Context — Lollipop Plot

FOXE1 · 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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