DHX37

Chr 12ADAR

DEAH-box helicase 37

Also known as: DDX37, Dhr1, NEDBAVC, SRXY11

This gene encodes an ATP-binding RNA helicase that is required for maturation of the small ribosomal subunit and release of U3 snoRNP from pre-ribosomal particles during ribosome biogenesis. Mutations cause 46XY sex reversal and neurodevelopmental disorder with brain anomalies and with or without vertebral or cardiac anomalies, following both autosomal dominant and autosomal recessive inheritance patterns. The gene is highly constrained against loss-of-function variants (pLI 0.99), consistent with its essential role in ribosome biogenesis and development of multiple organ systems including brain and gonads.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt
AD/ARLOEUF 0.292 OMIM phenotypes
Clinical SummaryDHX37
Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 0.99). One damaged copy is likely sufficient to cause disease.
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ClinVar Variants
28 unique Pathogenic / Likely Pathogenic· 13 VUS of 74 total submissions
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GeneReview available — DHX37
Authoritative clinical overview · Recommended first read
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Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.29LOEUF
pLI 0.993
Z-score 5.89
OE 0.17 (0.100.29)
Highly constrained

Highly LoF-intolerant (top ~10% of genes)

Missense Constraint
1.83Z-score
OE missense 0.81 (0.760.87)
594 obs / 733.3 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.17 (0.100.29)
00.351.4
Missense OE0.81 (0.760.87)
00.61.4
Synonymous OE0.96
01.21.6
LoF obs/exp: 10 / 58.7Missense obs/exp: 594 / 733.3Syn Z: 0.54

ClinVar Variant Classifications

74 submitted variants in ClinVar

Classification Summary

Pathogenic20
Likely Pathogenic8
VUS13
Likely Benign4
Benign26
Conflicting3
20
Pathogenic
8
Likely Pathogenic
13
VUS
4
Likely Benign
26
Benign
3
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
3
16
1
20
Likely Pathogenic
0
7
1
0
8
VUS
2
10
1
0
13
Likely Benign
0
2
0
2
4
Benign
0
6
9
11
26
Conflicting
3
Total228271474

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

View in ClinVar →

Protein Context — Lollipop Plot

DHX37 · 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.

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Clinical Literature
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Full-Text Mentions
NLP-detected gene mentions in article bodies · via PubTator3
PubTator3
Top 5 full-text resultsSearch PubTator3 ↗