MFSD8

Chr 4AR

major facilitator superfamily domain containing 8

Also known as: CCMD, CLN7, SLC74A1

The encoded protein functions as an outward-rectifying chloride channel that maintains endolysosomal chloride homeostasis, regulates lysosomal calcium content by activating TRPML1 channels, and contributes to progressive acidification from endosome to lysosome. Mutations cause neuronal ceroid lipofuscinosis type 7 (variant late infantile-onset) and macular dystrophy with central cone involvement through an autosomal recessive inheritance pattern. Disease results from loss-of-function mutations that disrupt endolysosomal membrane function and ion homeostasis.

Summary from RefSeq, OMIM, UniProt, Mechanism
Research Assistant →

Primary Disease Associations & Inheritance

Ceroid lipofuscinosis, neuronal, 7MIM #610951
AR
Macular dystrophy with central cone involvementMIM #616170
AR
2
Active trials
11
Pubs (1 yr)
109
P/LP submissions
11%
P/LP missense
1.07
LOEUF
LOF
Mechanism· annotated
Clinical SummaryMFSD8
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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
91 unique Pathogenic / Likely Pathogenic· 184 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 — MFSD8
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
1.07LOEUF
pLI 0.000
Z-score 1.27
OE 0.75 (0.531.07)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.11Z-score
OE missense 0.98 (0.891.08)
275 obs / 280.4 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.75 (0.531.07)
00.351.4
Missense OE0.98 (0.891.08)
00.61.4
Synonymous OE1.00
01.21.6
LoF obs/exp: 22 / 29.5Missense obs/exp: 275 / 280.4Syn Z: -0.02
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveMFSD8-related neuronal ceroid-lipofuscinosisLOFAR
Mechanism Note (expert annotation)
LOF

Lysosomal membrane transporter. Biallelic LOF causes CLN7 (variant late-infantile neuronal ceroid lipofuscinosis). This is an autosomal recessive LOF gene.

References:PMID:17564974

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

DN
0.7229th %ile
GOF
0.6149th %ile
LOF
0.2874th %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

500 submitted variants in ClinVar

Classification Summary

Pathogenic49
Likely Pathogenic42
VUS184
Likely Benign203
Benign3
Conflicting8
49
Pathogenic
42
Likely Pathogenic
184
VUS
203
Likely Benign
3
Benign
8
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
24
1
24
0
49
Likely Pathogenic
22
9
11
0
42
VUS
1
144
29
10
184
Likely Benign
0
3
81
119
203
Benign
0
0
3
0
3
Conflicting
8
Total47157148129489

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

View in ClinVar →

Protein Context — Lollipop Plot

MFSD8 · protein map & ClinVar variants

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

Clinical Literature
Open Research Assistant →
Key Publications
Landmark & review papers · by relevance
PubMed
Top 5 results · since 2015Search PubMed ↗