TSC2

Chr 16AD

TSC complex subunit 2

Also known as: LAM, PPP1R160, TSC4

The protein tuberin interacts with hamartin to form the TSC protein complex that negatively regulates mTORC1 signaling, thereby controlling anabolic cell growth. Loss-of-function mutations cause tuberous sclerosis-2, an autosomal dominant disorder characterized by benign tumor growth in multiple organs, as well as lymphangioleiomyomatosis and focal cortical dysplasia type II. The gene is highly intolerant to loss-of-function variants, consistent with its role as a critical tumor suppressor.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
LOFmechanismADLOEUF 0.075 OMIM phenotypes
Clinical SummaryTSC2
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Gene-Disease Validity (ClinGen)
tuberous sclerosis · ADDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 1.00). One damaged copy is likely sufficient to cause disease.
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ClinVar Variants
25 unique Pathogenic / Likely Pathogenic· 143 VUS of 300 total submissions
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Clinical Trials
6 active or recruiting trials — potential therapeutic options may be available
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GeneReview available — TSC2
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.07LOEUF
pLI 1.000
Z-score 8.37
OE 0.02 (0.010.07)
Highly constrained

Among the most LoF-intolerant genes (~top 3%)

Missense Constraint
-0.33Z-score
OE missense 1.03 (0.981.08)
1099 obs / 1068.6 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.02 (0.010.07)
00.351.4
Missense OE1.03 (0.981.08)
00.61.4
Synonymous OE1.48
01.21.6
LoF obs/exp: 2 / 85.4Missense obs/exp: 1099 / 1068.6Syn Z: -8.39
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveTSC2-related tuberous sclerosisLOFAD
DN
0.3196th %ile
GOF
0.3689th %ile
LOF
0.74top 10%

This gene has evidence for multiple mechanisms of pathogenicity (loss-of-function and gain-of-function). 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.

LOFprediction above median · 1 literature citation · 80% of P/LP variants are LoF · LOEUF 0.07
GOF1 literature citation

Literature Evidence

GOFRanek et al. (2019) showed that phosphorylation or gain- or loss-of-function mutations at either of 2 adjacent serine residues in TSC2 (S1365 and S1366 in mice; S1364 and S1365 in humans) could bidirectionally control mTORC1 activity stimulated by growth factors or hemodynamic stress, and consequentPMID:30700906
LOFIntriguingly, the LAM-like features of these cells suggest that haploinsufficiency at the TSC2 locus contributes to LAM pathology, and demonstrated that iPSC reprogramming and SMC lineage differentiation of somatic patient cells with germline mutations was a viable approach to generate LAM-like cellPMID:28830860

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

300 submitted variants in ClinVar

Classification Summary

Pathogenic22
Likely Pathogenic3
VUS143
Likely Benign118
Benign5
Conflicting9
22
Pathogenic
3
Likely Pathogenic
143
VUS
118
Likely Benign
5
Benign
9
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
20
0
1
1
22
Likely Pathogenic
0
3
0
0
3
VUS
1
122
20
0
143
Likely Benign
0
3
74
41
118
Benign
0
1
3
1
5
Conflicting
9
Total211299843300

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

View in ClinVar →

Protein Context — Lollipop Plot

TSC2 · 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

Lymphoma, Non-HodgkinMultiple MyelomaAdvanced Solid Tumors

Canadian Profiling and Targeted Agent Utilization Trial (CAPTUR)

RECRUITING
NCT03297606Phase PHASE2Canadian Cancer Trials GroupStarted 2018-03-23
OlaparibDasatinibNivolumab plus Ipilimumab
Polycythemia VeraEssential ThrombocythaemiaMyelofibrosis

Prevalence Of Germline Gene Mutations In Patients With Myeloproliferative Neoplasms With Family History

NOT YET RECRUITING
NCT06923670Phase NAFondazione Policlinico Universitario Agostino Gemelli IRCCSStarted 2025-05-21
NGS testingNGS analysis for mutations in genes involved in familial predisposition to hematological malignancies
Malignant Solid Neoplasms

Adapting Treatment to the Tumor Molecular Alterations for Patients With Advanced Solid Tumors: MyOwnSpecificTreatment

RECRUITING
NCT02029001Phase PHASE2Centre Leon BerardStarted 2014-03
Nilotinib (400 mg BID)Everolimus (10 mg QD)Sorafenib (400 mg BID)
Advanced LymphomaAdvanced Malignant Solid NeoplasmBladder Carcinoma

Targeted Therapy Directed by Genetic Testing in Treating Patients With Advanced Refractory Solid Tumors, Lymphomas, or Multiple Myeloma (The MATCH Screening Trial)

ACTIVE NOT RECRUITING
NCT02465060Phase PHASE2National Cancer Institute (NCI)Started 2015-08-17
AdavosertibAfatinibAfatinib Dimaleate
Advanced Malignant Solid NeoplasmAnn Arbor Stage III Non-Hodgkin LymphomaAnn Arbor Stage IV Non-Hodgkin Lymphoma

Targeted Therapy Directed by Genetic Testing in Treating Pediatric Patients With Relapsed or Refractory Advanced Solid Tumors, Non-Hodgkin Lymphomas, or Histiocytic Disorders (The Pediatric MATCH Screening Trial)

ACTIVE NOT RECRUITING
NCT03155620Phase PHASE2National Cancer Institute (NCI)Started 2017-07-31
Biopsy ProcedureBiospecimen CollectionBone Marrow Aspiration and Biopsy
TumorTumor, SolidMetastasis

Phase 2 Basket Trial of Nab-sirolimus in Patients With Malignant Solid Tumors With Pathogenic Alterations in TSC1/TSC2 Genes (PRECISION 1)

ACTIVE NOT RECRUITING
NCT05103358Phase PHASE2Aadi Bioscience, Inc.Started 2022-02-15
nab-sirolimus
Clinical Literature
Open Research Assistant →
Key Publications
Landmark & review papers · by relevance
PubMed
Top 5 results · since 2015Search PubMed ↗