DNAH9

Chr 17AR

dynein axonemal heavy chain 9

Also known as: CILD40, DNAH17L, DNEL1, DYH9, Dnahc9, HL-20, HL20

This gene encodes a heavy chain subunit of axonemal dynein, a molecular motor that hydrolyzes ATP to generate force for ciliary beating in respiratory epithelia and other tissues. Mutations cause primary ciliary dyskinesia with autosomal recessive inheritance, resulting in chronic respiratory infections, bronchiectasis, and often situs inversus due to defective ciliary function. The gene shows moderate constraint against loss-of-function variants (LOEUF 0.722), and symptoms typically begin in early childhood with recurrent respiratory tract infections.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismARLOEUF 0.721 OMIM phenotype
Clinical SummaryDNAH9
🧬
Gene-Disease Validity (ClinGen)
ciliary dyskinesia, primary, 40 · 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.
📋
ClinVar Variants
35 unique Pathogenic / Likely Pathogenic· 307 VUS of 600 total submissions
📖
GeneReview available — DNAH9
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 5.03
OE 0.62 (0.540.72)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
-0.04Z-score
OE missense 1.00 (0.971.04)
2436 obs / 2430.0 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.62 (0.540.72)
00.351.4
Missense OE1.00 (0.971.04)
00.61.4
Synonymous OE0.97
01.21.6
LoF obs/exp: 130 / 208.4Missense obs/exp: 2436 / 2430.0Syn Z: 0.64
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveDNAH9-related motile cilia defects and situs inversusLOFAR

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

DN
0.6550th %ile
GOF
0.74top 25%
LOF
0.1994th %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

600 submitted variants in ClinVar

Classification Summary

Pathogenic24
Likely Pathogenic11
VUS307
Likely Benign227
Benign3
Conflicting7
24
Pathogenic
11
Likely Pathogenic
307
VUS
227
Likely Benign
3
Benign
7
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
24
0
0
0
24
Likely Pathogenic
11
0
0
0
11
VUS
0
298
8
1
307
Likely Benign
1
7
77
142
227
Benign
0
0
3
0
3
Conflicting
7
Total3630588143579

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

View in ClinVar →

Protein Context — Lollipop Plot

DNAH9 · 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 →