NEFH

Chr 22ADAR

neurofilament heavy chain

Also known as: CMT2CC, NFH

The heavy neurofilament protein forms part of the axoskeleton that maintains neuronal caliber and may facilitate intracellular transport to axons and dendrites. Mutations cause Charcot-Marie-Tooth disease type 2CC and confer susceptibility to amyotrophic lateral sclerosis through both autosomal dominant and autosomal recessive inheritance patterns. The pathogenic mechanism involves dominant-negative effects disrupting normal neurofilament function.

OMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
MultiplemechanismAD/ARLOEUF 1.062 OMIM phenotypes
Clinical SummaryNEFH
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Gene-Disease Validity (ClinGen)
amyotrophic lateral sclerosis · ADLimited

Limited evidence — not for standalone diagnostic reporting

2 total gene-disease associations curated

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.06LOEUF
pLI 0.000
Z-score 1.28
OE 0.75 (0.531.06)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.52Z-score
OE missense 0.93 (0.871.01)
470 obs / 503.0 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.75 (0.531.06)
00.351.4
Missense OE0.93 (0.871.01)
00.61.4
Synonymous OE1.05
01.21.6
LoF obs/exp: 22 / 29.5Missense obs/exp: 470 / 503.0Syn Z: -0.52
DN
0.7130th %ile
GOF
0.6346th %ile
LOF
0.4332th %ile

This gene has evidence for multiple mechanisms of pathogenicity (dominant-negative and gain-of-function). Both the Badonyi & Marsh prediction and the broader genomic evidence point to dominant-negative as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

DNprediction above median
GOF1 literature citation

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.

Literature Evidence

GOFThe transfected cells appeared round and had significantly decreased axon-like projections compared to wildtype, and the aggregates trapped NEFL (162280), kinesin, and other proteins, and thus disrupted the neurofilament network in a toxic gain-of-function manner.PMID:27040688

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

NEFH · 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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