RTEL1

Chr 20ADAR

regulator of telomere elongation helicase 1

Also known as: C20orf41, DKCA4, DKCB5, NHL, PFBMFT3, RTEL

This gene encodes an ATP-dependent DNA helicase that regulates telomere length, prevents toxic DNA recombination, and maintains genomic stability by disassembling problematic DNA structures including telomeric loops and G-quadruplexes. Mutations cause dyskeratosis congenita and pulmonary fibrosis and/or bone marrow failure syndrome with telomere-related pathology, affecting multiple systems including skin, nails, mucous membranes, lungs, and hematopoietic system. The gene shows both autosomal dominant and autosomal recessive inheritance patterns and is highly intolerant to loss-of-function variants.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismAD/ARLOEUF 0.543 OMIM phenotypes
Clinical SummaryRTEL1
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Gene-Disease Validity (ClinGen)
pulmonary fibrosis and/or bone marrow failure, Telomere-related, 3 · SDDefinitive

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
39 unique Pathogenic / Likely Pathogenic· 278 VUS of 500 total submissions
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Clinical Trials
2 active or recruiting trials — potential therapeutic options may be available
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GeneReview available — RTEL1
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.54LOEUF
pLI 0.000
Z-score 4.82
OE 0.39 (0.290.54)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
-1.15Z-score
OE missense 1.12 (1.051.18)
875 obs / 784.3 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.39 (0.290.54)
00.351.4
Missense OE1.12 (1.051.18)
00.61.4
Synonymous OE1.37
01.21.6
LoF obs/exp: 29 / 73.7Missense obs/exp: 875 / 784.3Syn Z: -5.44
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
limitedRTEL1-related dyskeratosis congenitaLOFAD
definitiveRTEL1-related dyskeratosis congenitaLOFAR
DN
0.6744th %ile
GOF
0.5169th %ile
LOF
0.3164th %ile

The highest-scoring mechanism for this gene is dominant-negative.

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

500 submitted variants in ClinVar

Classification Summary

Pathogenic12
Likely Pathogenic27
VUS278
Likely Benign178
Benign1
Conflicting3
12
Pathogenic
27
Likely Pathogenic
278
VUS
178
Likely Benign
1
Benign
3
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
11
0
1
0
12
Likely Pathogenic
24
3
0
0
27
VUS
1
263
12
2
278
Likely Benign
0
7
73
98
178
Benign
0
1
0
0
1
Conflicting
3
Total3627486100499

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

View in ClinVar →

Protein Context — Lollipop Plot

RTEL1 · 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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