TMPRSS6

Chr 22AR

transmembrane serine protease 6

Also known as: IRIDA, MT2

TMPRSS6 encodes a membrane-bound serine protease that cleaves hemojuvelin to regulate hepcidin expression and maintain iron homeostasis. Autosomal recessive mutations cause iron-refractory iron deficiency anemia, a condition characterized by severe iron deficiency that does not respond to oral iron supplementation. The gene is not highly constrained against loss-of-function variants.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismARLOEUF 0.721 OMIM phenotype
Clinical SummaryTMPRSS6
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Gene-Disease Validity (ClinGen)
IRIDA syndrome · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
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ClinVar Variants
55 unique Pathogenic / Likely Pathogenic· 223 VUS of 436 total submissions
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Clinical Trials
4 active or recruiting trials — potential therapeutic options may be available
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GeneReview available — TMPRSS6
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.72LOEUF
pLI 0.000
Z-score 3.06
OE 0.51 (0.360.72)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.21Z-score
OE missense 0.97 (0.901.05)
485 obs / 498.4 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.51 (0.360.72)
00.351.4
Missense OE0.97 (0.901.05)
00.61.4
Synonymous OE1.08
01.21.6
LoF obs/exp: 23 / 45.2Missense obs/exp: 485 / 498.4Syn Z: -0.90
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveTMPRSS6-related iron-refractory iron deficiency anemiaLOFAR

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

DN
0.6647th %ile
GOF
0.7029th %ile
LOF
0.3065th %ile

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.

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

ClinVar Variant Classifications

436 submitted variants in ClinVar

Classification Summary

Pathogenic40
Likely Pathogenic15
VUS223
Likely Benign72
Benign51
Conflicting23
40
Pathogenic
15
Likely Pathogenic
223
VUS
72
Likely Benign
51
Benign
23
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
18
3
19
0
40
Likely Pathogenic
11
3
1
0
15
VUS
3
195
19
6
223
Likely Benign
5
10
25
32
72
Benign
2
4
30
15
51
Conflicting
23
Total392159453424

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

View in ClinVar →

Protein Context — Lollipop Plot

TMPRSS6 · 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 →