MSH2

Chr 2ADAR

mutS homolog 2

Also known as: COCA1, FCC1, HNPCC, HNPCC1, LCFS2, LYNCH1, MMRCS2, MSH-2

MSH2 encodes a component of the post-replicative DNA mismatch repair system that forms heterodimers (MutS alpha and beta) to recognize and bind DNA mismatches, initiating repair processes. Mutations cause Lynch syndrome 1, mismatch repair cancer syndrome 2, and Muir-Torre syndrome with both autosomal dominant and autosomal recessive inheritance patterns. The gene is highly constrained against loss-of-function variants (pLI 0.90, LOEUF 0.33), reflecting its critical role in maintaining genomic stability.

Summary from RefSeq, OMIM, UniProt
Research Assistant →

Primary Disease Associations & Inheritance

Lynch syndrome 1MIM #120435
AD
Mismatch repair cancer syndrome 2MIM #619096
AR
Muir-Torre syndromeMIM #158320
AD
UniProtEndometrial cancer
UniProtColorectal cancer
12
Active trials
338
Pubs (1 yr)
99
P/LP submissions
1%
P/LP missense
0.33
LOEUF· LoF intol.
LOF
Mechanism· G2P
Clinical SummaryMSH2
🧬
Gene-Disease Validity (ClinGen)
Lynch syndrome · ADDefinitive

Definitive — sufficient evidence for diagnostic panels

3 total gene-disease associations curated

Population Constraint (gnomAD)
Moderately constrained gene (pLI 0.90) — some intolerance to loss-of-function variants.
📋
ClinVar Variants
99 unique Pathogenic / Likely Pathogenic· 186 VUS of 600 total submissions
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Clinical Trials
12 active or recruiting trials — potential therapeutic options may be available
📖
GeneReview available — MSH2
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.33LOEUF
pLI 0.895
Z-score 5.13
OE 0.19 (0.110.33)
Highly constrained

Highly LoF-intolerant (top ~10% of genes)

Missense Constraint
-2.45Z-score
OE missense 1.31 (1.231.40)
634 obs / 482.6 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.19 (0.110.33)
00.351.4
Missense OE1.31 (1.231.40)
00.61.4
Synonymous OE1.18
01.21.6
LoF obs/exp: 9 / 47.0Missense obs/exp: 634 / 482.6Syn Z: -1.83
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveMSH2-related Lynch syndromeLOFAD
definitiveMSH2-related constitutional mismatch repair deficiency syndrome (CMMRD)LOFAR
DN
0.4686th %ile
GOF
0.3292th %ile
LOF
0.60top 25%

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

LOF1 literature citation · 76% of P/LP variants are LoF · LOEUF 0.33
DN1 literature citation

Literature Evidence

DNThe hMSH2(M688R) Lynch syndrome mutation may function as a dominant negative.PMID:22739024
LOFDetection of exon deletions and duplications of the mismatch repair genes in hereditary nonpolyposis colorectal cancer families using multiplex polymerase chain reaction of short fluorescent fragmentsPMID:10850409

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

600 submitted variants in ClinVar

Classification Summary

Pathogenic79
Likely Pathogenic20
VUS186
Likely Benign213
Benign85
Conflicting5
79
Pathogenic
20
Likely Pathogenic
186
VUS
213
Likely Benign
85
Benign
5
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
64
0
15
0
79
Likely Pathogenic
11
1
8
0
20
VUS
7
153
23
3
186
Likely Benign
1
40
94
78
213
Benign
0
7
12
66
85
Conflicting
5
Total83201152147588

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

View in ClinVar →

Protein Context — Lollipop Plot

MSH2 · 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

Prostate CancerBRCA MutationMismatch Repair Gene Mutation

The IMPACT Study - Identification of Men With a Genetic Predisposition to ProstAte Cancer

ACTIVE NOT RECRUITING
NCT00261456Institute of Cancer Research, United KingdomStarted 2005-03
PSA testProstate Biopsy
BRCA-Mutated Ovarian CarcinomaBRIP1 Gene MutationMSH2 A636P

Cascade Testing in Families With Newly Diagnosed Hereditary Breast and Ovarian Cancer Syndrome

ACTIVE NOT RECRUITING
NCT04009148NYU Langone HealthStarted 2019-03-01
CASCADE genetic screening
Pancreatic CancerPancreatic Ductal AdenocarcinomaPDAC

A Prospective Registry for Patients at High-Risk for Pancreatic Cancer

RECRUITING
NCT06151223Mayo ClinicStarted 2021-07-13
Bio-specimen Collection: BloodBio-specimen Collection: Pancreatic JuiceMRI
MMR MutationSmall Bowel AdenomaSmall-bowel Adenocarcinoma

Small Bowel Capsule Endoscopy in Lynch Syndrome

RECRUITING
NCT07472686Assistance Publique - Hôpitaux de ParisStarted 2025-06-16
Pancreas CancerExosomesExtracellular Vesicles

ExoLuminate Study for Early Detection of Pancreatic Cancer

RECRUITING
NCT05625529Biological DynamicsStarted 2022-12-19
Prostate Cancer Metastatic Castration-ResistantAbnormal DNA RepairMetastatic Prostate Carcinoma

Abiraterone/Prednisone, Olaparib, or Abiraterone/Prednisone + Olaparib in Patients With Metastatic Castration-Resistant Prostate Cancer With DNA Repair Defects

ACTIVE NOT RECRUITING
NCT03012321Phase PHASE2Northwestern UniversityStarted 2017-01-12
OlaparibAbiraterone AcetatePrednisone
Colorectal CancerAdenoma ColonAdenoma Colon Polyp

Liquid Biopsy and Machine Learning for Early Colorectal Cancer, Adenomas, Lynch Cancers, and Residual Disease Detection

RECRUITING
NCT07450612San Raffaele UniversityStarted 2024-01-01
BEACON
Pancreas CancerPancreas CystPancreatic Ductal Adenocarcinoma

Pancreatic Cancer Early Detection Consortium

RECRUITING
NCT04970056Arbor Research Collaborative for HealthStarted 2020-09-18
Pancreatic Cancer

Pancreatic Cancer Genetics

RECRUITING
NCT01102569Columbia UniversityStarted 2008-01
Advanced Solid Tumor

A Modular Multi-Basket Trial to Improve Personalized Medicine in Cancer Patients (Basket of Baskets)

RECRUITING
NCT03767075Phase PHASE2Vall d'Hebron Institute of OncologyStarted 2018-12-10
AtezolizumabFutibatinibAmivantamab
Lynch SyndromeLynch Syndrome ILynch Syndrome II

Lynch Syndrome X-Talk of Enteral Mucosa With Immune System

RECRUITING
NCT06708429San Raffaele UniversityStarted 2023-06-01
LYNX EYE (Lynch syndrome X-Talk of Enteral mucosa with Immune System)
Lymphoma, Non-HodgkinMultiple MyelomaAdvanced Solid Tumors

TAPUR: Testing the Use of Food and Drug Administration (FDA) Approved Drugs That Target a Specific Abnormality in a Tumor Gene in People With Advanced Stage Cancer

RECRUITING
NCT02693535Phase PHASE2American Society of Clinical OncologyStarted 2016-03-14
PalbociclibSunitinibTemsirolimus
Clinical Literature
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Key Publications
Landmark & review papers · by relevance
PubMed
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