CTDP1

Chr 18AR

CTD phosphatase subunit 1

Also known as: CCFDN, FCP1

This gene encodes a protein which interacts with the carboxy-terminus of the RAP74 subunit of transcription initiation factor TFIIF, and functions as a phosphatase that processively dephosphorylates the C-terminus of POLR2A (a subunit of RNA polymerase II), making it available for initiation of gene expression. Mutations in this gene are associated with congenital cataracts, facial dysmorphism and neuropathy syndrome (CCFDN). Alternatively spliced transcript variants encoding different isoforms have been described for this gene. [provided by RefSeq, Feb 2011]

Primary Disease Associations & Inheritance

Congenital cataracts, facial dysmorphism, and neuropathyMIM #604168
AR
0
Active trials
4
Pubs (1 yr)
67
P/LP submissions
0%
P/LP missense
0.53
LOEUF
LOF
Mechanism· G2P
Clinical SummaryCTDP1
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.33) despite low pLI — interpret in context.
📋
ClinVar Variants
67 unique Pathogenic / Likely Pathogenic· 270 VUS of 700 total submissions
📖
GeneReview available — CTDP1
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.53LOEUF
pLI 0.001
Z-score 3.86
OE 0.33 (0.220.53)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
1.00Z-score
OE missense 0.88 (0.820.95)
501 obs / 567.8 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.33 (0.220.53)
00.351.4
Missense OE0.88 (0.820.95)
00.61.4
Synonymous OE1.12
01.21.6
LoF obs/exp: 13 / 39.0Missense obs/exp: 501 / 567.8Syn Z: -1.50

ClinVar Variant Classifications

700 submitted variants in ClinVar

Classification Summary

Pathogenic61
Likely Pathogenic6
VUS270
Likely Benign288
Benign58
Conflicting9
61
Pathogenic
6
Likely Pathogenic
270
VUS
288
Likely Benign
58
Benign
9
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
0
61
0
61
Likely Pathogenic
0
0
6
0
6
VUS
4
233
32
1
270
Likely Benign
0
35
109
144
288
Benign
0
4
42
12
58
Conflicting
9
Total4272250157692

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

View in ClinVar →

Protein Context — Lollipop Plot

CTDP1 · 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
Open Research Assistant →
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC