SPATC1L

Chr 21

spermatogenesis and centriole associated 1 like

Also known as: C21orf56

The SPATC1L protein enables identical protein binding and is predicted to regulate cAMP/PKA signaling pathways and actin polymerization. Mutations cause autosomal recessive intellectual disability with severe speech delay and mild dysmorphism. This gene shows very low constraint against loss-of-function variants (pLI near 0, LOEUF 1.8), suggesting tolerance to protein-truncating changes.

Summary from RefSeq
Research Assistant →
0
Active trials
2
Pubs (1 yr)
99
P/LP submissions
0%
P/LP missense
1.80
LOEUF
Multiple*
Mechanism· predicted
Clinical SummarySPATC1L
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
93 unique Pathogenic / Likely Pathogenic· 104 VUS of 214 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.80LOEUF
pLI 0.000
Z-score -0.49
OE 1.17 (0.731.80)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.29Z-score
OE missense 0.94 (0.841.06)
199 obs / 210.9 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE1.17 (0.731.80)
00.351.4
Missense OE0.94 (0.841.06)
00.61.4
Synonymous OE1.22
01.21.6
LoF obs/exp: 11 / 9.4Missense obs/exp: 199 / 210.9Syn Z: -1.84
DN
0.6550th %ile
GOF
0.6638th %ile
LOF
0.3939th %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

214 submitted variants in ClinVar

Classification Summary

Pathogenic85
Likely Pathogenic8
VUS104
Likely Benign6
Benign4
Conflicting1
85
Pathogenic
8
Likely Pathogenic
104
VUS
6
Likely Benign
4
Benign
1
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
85
0
85
Likely Pathogenic
0
0
8
0
8
VUS
0
81
23
0
104
Likely Benign
0
4
1
1
6
Benign
0
4
0
0
4
Conflicting
1
Total0891171208

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

View in ClinVar →

Protein Context — Lollipop Plot

SPATC1L · protein map & ClinVar variants

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

Clinical Trials

Active and recruiting trials from ClinicalTrials.gov

No active trials found for this gene.

Search ClinicalTrials.gov →
Clinical Literature
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Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC