MTR

Chr 1AR

5-methyltetrahydrofolate-homocysteine methyltransferase

Catalyzes the transfer of a methyl group from methylcob(III)alamin (MeCbl) to homocysteine, yielding enzyme-bound cob(I)alamin and methionine in the cytosol (PubMed:16769880, PubMed:17288554, PubMed:27771510). MeCbl is an active form of cobalamin (vitamin B12) used as a cofactor for methionine biosynthesis. Cob(I)alamin form is regenerated to MeCbl by a transfer of a methyl group from 5-methyltetrahydrofolate (PubMed:16769880, PubMed:17288554, PubMed:27771510). The processing of cobalamin in the cytosol occurs in a multiprotein complex composed of at least MMACHC, MMADHC, MTRR (methionine synthase reductase) and MTR which may contribute to shuttle safely and efficiently cobalamin towards MTR in order to produce methionine (PubMed:16769880, PubMed:27771510)

Primary Disease Associations & Inheritance

{Neural tube defects, folate-sensitive, susceptibility to}MIM #601634
AR
Homocystinuria-megaloblastic anemia, cblG complementation typeMIM #250940
AR
1541
ClinVar variants
42
Pathogenic / LP
0.00
pLI score
6
Active trials
Clinical SummaryMTR
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
📋
ClinVar Variants
42 Pathogenic / Likely Pathogenic· 77 VUS of 1541 total submissions
💊
Clinical Trials
6 active or recruiting trials — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint?LOEUF (Loss-of-function Observed/Expected Upper bound Fraction) is the upper bound of the 90% CI for LoF OE — the preferred gnomAD v4 metric. Lower = more intolerant to LoF. LOEUF < 0.35 = highly constrained.
0.63LOEUF
pLI 0.000
Z-score 4.05
OE 0.46 (0.350.63)
Tolerant

Typical tolerance to LoF variation

Missense Constraint?Missense Z-score: standard deviations fewer missense variants observed vs. expected. Z > 3.09 (p < 0.001) = gene does not tolerate missense variation. OE missense < 0.6 is also considered constrained.
2.04Z-score
OE missense 0.78 (0.720.84)
527 obs / 676.6 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios?Shaded band = 90% confidence interval. Vertical tick = point estimate. Grey threshold line = gnomAD constraint cutoff for that variant class.
LoF OE?Ratio of observed to expected LoF variants. Upper CI bound (LOEUF) ≤ 0.35 = strong LoF constraint signal.0.46 (0.350.63)
00.351.4
Missense OE?Ratio of observed to expected missense variants. OE ≤ 0.6 = fewer missense variants than expected by chance.0.78 (0.720.84)
00.61.4
Synonymous OE?Control metric — synonymous variants are largely neutral and expected near OE = 1.0. Significant deviation may indicate annotation issues.1.05
01.21.6
LoF obs/exp: 31 / 66.7Missense obs/exp: 527 / 676.6Syn Z: -0.58

ClinVar Variant Classifications

1541 submitted variants in ClinVar

Classification Summary

Pathogenic28
Likely Pathogenic14
VUS77
Likely Benign332
Benign13
Conflicting1
28
Pathogenic
14
Likely Pathogenic
77
VUS
332
Likely Benign
13
Benign
1
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
13
2
13
0
28
Likely Pathogenic
11
0
3
0
14
VUS
1
68
8
0
77
Likely Benign
0
2
167
163
332
Benign
0
0
12
1
13
Conflicting
1
Total2572203164465

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

View in ClinVar →

Protein Context — Lollipop Plot

MTR · protein map & ClinVar variants

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

Gene2Phenotype Curations

MTR-related methylcobalamin deficiency

definitive
ARLoss Of FunctionAbsent Gene Product
Dev. DisordersEye
G2P ↗

Gene2Phenotype curations · DECIPHER consortium patient cohort (public variants) · deciphergenomics.org

OMIM — Genotype-Phenotype Relationships

1 OMIM entry

{Neural tube defects, folate-sensitive, susceptibility to}

MIM #601634

Molecular basis of disorder known

Autosomal recessive

Homocystinuria-megaloblastic anemia, cblG complementation type

MIM #250940

Molecular basis of disorder known

Autosomal recessive
Clinical Literature
Landmark / reviewRecent case evidence
Key Publications
Landmark & review papers · by relevance
PubMed
Commensal Neisseria and Antimicrobial-Resistant Gonorrhea.
Wadsworth CB et al.·Annu Rev Microbiol
2025Review
Top 10 resultsSearch PubMed ↗

Clinical Trials

Active and recruiting trials from ClinicalTrials.gov

Sickle Cell Disease (SCD)

Study of Two Doses of Crizanlizumab Versus Placebo in Adolescent and Adult Sickle Cell Disease Patients

ACTIVE NOT RECRUITING
NCT03814746Phase PHASE3Novartis PharmaceuticalsStarted 2019-07-26
Crizanlizumab (SEG101)Placebo
Sickle Cell Disease

A Study to Investigate the Efficacy and Safety of Crizanlizumab (5 mg/kg) Compared With Placebo in Adolescent and Adult Sickle Cell Disease Patients Who Experience Frequent Vaso-Occlusive Crises (SPARKLE)

RECRUITING
NCT06439082Phase PHASE3Novartis PharmaceuticalsStarted 2024-10-24
CrizanlizumabPlacebo
Carpal Tunnel SyndromeNeurodynamic TreatmentNerve Mobilisation

Mechanisms of Neurodynamic Treatments

RECRUITING
NCT05859412Phase NAUniversity of OxfordStarted 2023-05-17
Neurodynamic exercisesSteroid injection (Depomedrone 40mg)Advice
Breast NeoplasmsTriple Negative Breast NeoplasmsHR Low-Positive/HER2-Negative Breast Neoplasms

A Clinical Study of Sacituzumab Tirumotecan (Sac-TMT, MK-2870) in People With Breast Cancer (MK-2870-032)

RECRUITING
NCT06966700Phase PHASE3Merck Sharp & Dohme LLCStarted 2025-06-30
Sacituzumab tirumotecanPembrolizumabRescue Medication
Colon AdenocarcinomaRectal Adenocarcinoma

A Clinical Study of Calderasib (MK-1084) With Targeted Therapy and Chemotherapy in People With Colorectal Cancer (MK-1084-012/KANDLELIT-012)

RECRUITING
NCT06997497Phase PHASE3Merck Sharp & Dohme LLCStarted 2025-07-16
CalderasibOxaliplatinLeucovorin/levofolinate calcium
Organic AcidemiaMethylmalonic AcidemiaInborn Errors of Metabolism

Clinical and Laboratory Study of Methylmalonic Acidemia

RECRUITING
NCT00078078National Human Genome Research Institute (NHGRI)Started 2004-06-07