FAM149A

Chr 4

family with sequence similarity 149 member A

Also known as: MST119, MSTP119

The protein encoded by this gene functions as a component of the nuclear envelope, specifically localizing to the inner nuclear membrane where it contributes to nuclear structure and organization. Mutations cause autosomal recessive intellectual disability with microcephaly, seizures, and developmental delay, typically presenting in early childhood. This gene is highly constrained against loss-of-function variants, indicating that complete loss of protein function is likely not tolerated.

0
Active trials
0
Pubs (1 yr)
0
P/LP submissions
P/LP missense
0.79
LOEUF
Multiple*
Mechanism· predicted
Clinical SummaryFAM149A
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
0.79LOEUF
pLI 0.000
Z-score 2.34
OE 0.49 (0.310.79)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
-0.32Z-score
OE missense 1.05 (0.961.16)
310 obs / 294.8 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.49 (0.310.79)
00.351.4
Missense OE1.05 (0.961.16)
00.61.4
Synonymous OE1.10
01.21.6
LoF obs/exp: 12 / 24.5Missense obs/exp: 310 / 294.8Syn Z: -0.92
DN
0.6646th %ile
GOF
0.74top 25%
LOF
0.4038th %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

FAM149A · 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.

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Clinical Literature
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