HSPB1

Chr 7AD

heat shock protein family B (small) member 1

Also known as: CMT2F, HEL-S-102, HMN2B, HMND3, HS.76067, HSP27, HSP28, Hsp25

The protein functions as a molecular chaperone that maintains denatured proteins in a folding-competent state and regulates neurofilament protein phosphorylation and axonal transport. Mutations cause autosomal dominant Charcot-Marie-Tooth disease type 2F and distal hereditary motor neuropathy, affecting peripheral nerves. The gene is highly tolerant to loss-of-function variants (pLI near zero, LOEUF 1.87), suggesting the pathogenic variants likely affect protein function through other mechanisms.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt
DNmechanismADLOEUF 1.872 OMIM phenotypes
Clinical SummaryHSPB1
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Gene-Disease Validity (ClinGen)
Charcot-Marie-Tooth disease axonal type 2F · ADDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📖
GeneReview available — HSPB1
Authoritative clinical overview · Recommended first read
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Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.87LOEUF
pLI 0.000
Z-score -0.63
OE 1.26 (0.741.87)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.51Z-score
OE missense 1.13 (0.981.30)
136 obs / 120.1 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE1.26 (0.741.87)
00.351.4
Missense OE1.13 (0.981.30)
00.61.4
Synonymous OE1.46
01.21.6
LoF obs/exp: 9 / 7.2Missense obs/exp: 136 / 120.1Syn Z: -2.68
DN
0.7034th %ile
GOF
0.5562th %ile
LOF
0.4529th %ile

The highest-scoring mechanism for this gene is dominant-negative.

DNprediction above median · 1 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

DNWe demonstrate that the stable truncated HSPB1 binds wild type HSPB1, suggesting a dominant-negative effect, and strongly impairs the ability of the cells to cope with unfolded protein stress.PMID:26675522

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

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

HSPB1 · 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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Key Publications
Landmark & review papers · by relevance
PubMed
Top 5 results · since 2015Search PubMed ↗