CLN6

Chr 15AR

CLN6 transmembrane ER protein

Also known as: CLN4A, CLN6A, HsT18960, nclf

The CLN6 protein is an endoplasmic reticulum membrane protein involved in lysosomal degradation of post-translationally modified proteins. Mutations cause neuronal ceroid lipofuscinosis types 6A and 6B (including Kufs type), autosomal recessive neurodegenerative disorders that comprise part of the Batten disease spectrum affecting children. The pathogenic mechanism involves defective lysosomal storage function leading to accumulation of cellular debris.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
LOFmechanismARLOEUF 1.132 OMIM phenotypes
Clinical SummaryCLN6
🧬
Gene-Disease Validity (ClinGen)
neuronal ceroid lipofuscinosis · 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
68 unique Pathogenic / Likely Pathogenic· 93 VUS of 346 total submissions
💊
Clinical Trials
3 active or recruiting trials — potential therapeutic options may be available
📖
GeneReview available — CLN6
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
1.13LOEUF
pLI 0.000
Z-score 1.23
OE 0.63 (0.371.13)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.01Z-score
OE missense 1.00 (0.891.14)
176 obs / 175.7 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.63 (0.371.13)
00.351.4
Missense OE1.00 (0.891.14)
00.61.4
Synonymous OE1.37
01.21.6
LoF obs/exp: 8 / 12.7Missense obs/exp: 176 / 175.7Syn Z: -2.53
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveCLN6-related neuronal ceroid lipofuscinosis, Kufs Type, adult onsetOTHERAR
definitiveCLN6-related neuronal ceroid lipofuscinosisLOFAR

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

DN
0.6552th %ile
GOF
0.6052th %ile
LOF
0.3455th %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

346 submitted variants in ClinVar

Classification Summary

Pathogenic41
Likely Pathogenic27
VUS93
Likely Benign138
Benign10
Conflicting35
41
Pathogenic
27
Likely Pathogenic
93
VUS
138
Likely Benign
10
Benign
35
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
23
7
11
0
41
Likely Pathogenic
17
9
1
0
27
VUS
1
73
19
0
93
Likely Benign
0
3
61
74
138
Benign
0
0
10
0
10
Conflicting
35
Total419210274344

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

View in ClinVar →

Protein Context — Lollipop Plot

CLN6 · 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 →
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC