FHL3

Chr 1AR

four and a half LIM domains 3

Also known as: SLIM2

The protein is a transcriptional coactivator that contains four-and-a-half LIM domains and interacts with developmental regulators including SMAD proteins and MyoD. Mutations cause familial hemophagocytic lymphohistiocytosis type 3, a severe immune dysregulation disorder affecting the hematopoietic system. The condition follows autosomal recessive inheritance.

OMIMResearchSummary from RefSeq, OMIM, UniProt
MultiplemechanismARLOEUF 0.841 OMIM phenotype
Clinical SummaryFHL3
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
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Clinical Trials
1 active or recruiting trial — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.84LOEUF
pLI 0.002
Z-score 2.01
OE 0.45 (0.260.84)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.79Z-score
OE missense 0.84 (0.730.95)
153 obs / 183.2 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.45 (0.260.84)
00.351.4
Missense OE0.84 (0.730.95)
00.61.4
Synonymous OE0.95
01.21.6
LoF obs/exp: 7 / 15.6Missense obs/exp: 153 / 183.2Syn Z: 0.35
DN
0.6454th %ile
GOF
0.6737th %ile
LOF
0.3647th %ile

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

GOFprediction above median
DNprediction above median

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.

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

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

FHL3 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
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