TINF2

Chr 14AD

TERF1 interacting nuclear factor 2

Also known as: DKCA3, DKCA5, TIN2

The protein is a critical component of the shelterin complex that protects telomeres from being recognized as DNA breaks and regulates telomere length. Mutations cause dyskeratosis congenita and Revesz syndrome, inherited bone marrow failure syndromes that affect multiple organ systems including skin, nails, and hematopoietic system. The gene follows autosomal dominant inheritance and is highly constrained against loss-of-function variants.

OMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismADLOEUF 0.672 OMIM phenotypes
Clinical SummaryTINF2
🧬
Gene-Disease Validity (ClinGen)
dyskeratosis congenita, autosomal dominant 3 · ADDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
9 unique Pathogenic / Likely Pathogenic· 124 VUS of 200 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.67LOEUF
pLI 0.001
Z-score 2.76
OE 0.38 (0.230.67)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.73Z-score
OE missense 0.87 (0.770.97)
203 obs / 234.2 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.38 (0.230.67)
00.351.4
Missense OE0.87 (0.770.97)
00.61.4
Synonymous OE1.22
01.21.6
LoF obs/exp: 9 / 23.4Missense obs/exp: 203 / 234.2Syn Z: -1.71
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveTINF2-related dyskeratosis congenitaLOFAD
definitiveTINF2-related exudative retinopathy with bone marrow failureLOFAD
DN
0.7133th %ile
GOF
0.77top 25%
LOF
0.2872th %ile

This gene has evidence for multiple mechanisms of pathogenicity (gain-of-function, dominant-negative and loss-of-function). 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
LOF1 literature citation · 44% of P/LP variants are LoF

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

LOFHeterozygous knock-in of the mutations or deletion of one copy of TINF2 resulted in excessive telomere elongation in clonal lines, indicating that TINF2 is haploinsufficient for telomere length control.PMID:33258446

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

200 submitted variants in ClinVar

Classification Summary

Pathogenic6
Likely Pathogenic3
VUS124
Likely Benign61
Benign2
Conflicting4
6
Pathogenic
3
Likely Pathogenic
124
VUS
61
Likely Benign
2
Benign
4
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
3
1
2
0
6
Likely Pathogenic
1
2
0
0
3
VUS
17
98
8
1
124
Likely Benign
0
0
25
36
61
Benign
0
0
2
0
2
Conflicting
4
Total211013737200

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

View in ClinVar →

Protein Context — Lollipop Plot

TINF2 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold

Clinical Trials

Active and recruiting trials from ClinicalTrials.gov

No active trials found for this gene.

Search ClinicalTrials.gov →
Clinical Literature
Open Research Assistant →
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC