MECP2

Chr XX-linkedXLRXLD

methyl-CpG binding protein 2

Also known as: AUTSX3, MRX16, MRX79, MRXS13, MRXSL, PPMX, RS, RTS

MECP2 encodes a methyl-CpG binding protein that binds specifically to methylated DNA and represses transcription from methylated gene promoters, playing an essential role in embryonic development. Mutations cause Rett syndrome and its variants, X-linked intellectual disability syndromes, neonatal severe encephalopathy, and autism susceptibility, with inheritance patterns including X-linked dominant (typical in Rett syndrome) and X-linked recessive forms. The gene shows high constraint against loss-of-function variants, and mutations predominantly cause disease through loss-of-function mechanisms, though the clinical phenotype varies significantly based on the specific variant and X-inactivation patterns.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
LOFmechanismX-linked/XLR/XLDLOEUF 0.417 OMIM phenotypes
VCEP Guidelines: Rett/Angelman-like DisordersReleased
View SpecificationsClinGen Panel
Clinical SummaryMECP2
🧬
Gene-Disease Validity (ClinGen)
Rett syndrome · XLDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Moderately constrained gene (pLI 0.89) — some intolerance to loss-of-function variants.
📋
ClinVar Variants
192 unique Pathogenic / Likely Pathogenic· 171 VUS of 498 total submissions
💊
Clinical Trials
12 active or recruiting trials — potential therapeutic options may be available
📖
GeneReview available — MECP2
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.41LOEUF
pLI 0.894
Z-score 2.89
OE 0.09 (0.030.41)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
-1.21Z-score
OE missense 1.23 (1.111.35)
281 obs / 229.3 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.09 (0.030.41)
00.351.4
Missense OE1.23 (1.111.35)
00.61.4
Synonymous OE2.03
01.21.6
LoF obs/exp: 1 / 11.7Missense obs/exp: 281 / 229.3Syn Z: -7.95
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveMECP2-related duplication syndromeOTHERXLR
definitiveMECP2-related Rett syndromeLOFmonoallelic_X_heterozygous
definitiveMECP2-related encephalopathy neonatal severeLOFXLR
DN
0.2299th %ile
GOF
0.2398th %ile
LOF
0.89top 5%

The highest-scoring mechanism for this gene is loss-of-function (haploinsufficiency).

LOFprediction above median · 60% of P/LP variants are LoF · LOEUF 0.41

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

498 submitted variants in ClinVar

Classification Summary

Pathogenic142
Likely Pathogenic50
VUS171
Likely Benign94
Benign12
Conflicting3
142
Pathogenic
50
Likely Pathogenic
171
VUS
94
Likely Benign
12
Benign
3
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
81
4
57
0
142
Likely Pathogenic
35
14
1
0
50
VUS
9
131
27
4
171
Likely Benign
0
8
31
55
94
Benign
0
2
8
2
12
Conflicting
3
Total12515912461472

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

View in ClinVar →

Protein Context — Lollipop Plot

MECP2 · 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

CDKL5CDKL5 Deficiency DisorderCDD

International CDKL5 Clinical Research Network

RECRUITING
NCT05558371University of Colorado, DenverStarted 2021-02-15
No intervention.
Rett Syndrome

Gene Editing as a Therapeutic Approach for Rett Syndrome

RECRUITING
NCT05740761University of SienaStarted 2021-03-01
Gene editing in vitro
Rett Syndrome

Safety and Efficacy of TSHA-102 in Pediatric Females With Rett Syndrome (REVEAL Pediatric Study)

ACTIVE NOT RECRUITING
NCT06152237Phase PHASE1, PHASE2Taysha Gene Therapies, Inc.Started 2023-12-12
TSHA-102
Rett Syndrome

A Novel, Regulated Gene Therapy (NGN-401) Study for Females With Rett Syndrome

RECRUITING
NCT05898620Phase PHASE3Neurogene Inc.Started 2023-06-13
NGN-401
RETT Syndrome With Proven MECP2 Mutation

Repurposing Mirtazapine in Rett Syndrome

RECRUITING
NCT07430046Phase PHASE2University of TriesteStarted 2025-07-09
mirtazapine
RETT Syndrome With Proven MECP2 MutationRett Syndrome

Efficacy and Safety of NTI164 in Children and Young Adults With Rett Syndrome

NOT YET RECRUITING
NCT07257978Phase PHASE2, PHASE3Fenix Innovation GroupStarted 2026-07-01
NTI164Placebo
Epilepsy

Precision Medicine in the Treatment of Epilepsy

RECRUITING
NCT05450822Gitte Moos KnudsenStarted 2022-02-18
LevetiracetamLevetiracetam TabletsLamotrigine tablet
Rett Syndrome

A Phase 1/2/3 Study of TSHA-102 Gene Therapy in Females With Rett Syndrome (REVEAL Pivotal Study)

RECRUITING
NCT05606614Phase PHASE3Taysha Gene Therapies, Inc.Started 2023-03-06
TSHA-102
Pitt Hopkins Syndrome

An Exploratory Evaluation of the Safety and Efficacy of Vorinostat in Pitt Hopkins Syndrome

RECRUITING
NCT07150026Phase PHASE1Unravel Biosciences, Inc.Started 2026-03-15
Vorinostat (SAHA)Placebo
Rett Syndrome

Rett REVOLUTION Trial: An Exploratory Evaluation of the Safety and Efficacy of Vorinostat in Rett Syndrome

RECRUITING
NCT07150013Phase PHASE1Unravel Biosciences, Inc.Started 2026-03-15
Vorinostat (SAHA)Placebo
Rett Syndrome

Safety and Preliminary Efficacy of TSHA-102 Gene Therapy in Pediatric Females Aged >2 to <4 Years With Rett Syndrome

NOT YET RECRUITING
NCT07480564Phase PHASE3Taysha Gene Therapies, Inc.Started 2026-03
TSHA-102
Prader-Willi SyndromePWS-like SyndromeSilver Russel Syndrome

GROWing Up With Rare GENEtic Syndromes

RECRUITING
NCT04463316dr. Laura C. G. de Graaff-HerderStarted 2018-10-01
Retrospective file studies
Clinical Literature
Open Research Assistant →
Full-Text Mentions
NLP-detected gene mentions in article bodies · via PubTator3
PubTator3
Top 5 full-text resultsSearch PubTator3 ↗
Key Publications
Landmark & review papers · by relevance
PubMed
Rett syndrome.
Gold WA et al.·Nat Rev Dis Primers
2024Review
X-Linked Epilepsies: A Narrative Review.
Bernardo P et al.·Int J Mol Sci
2024Review
Top 5 results · since 2015Search PubMed ↗