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Gene | TSC2 |
Variant | E114* |
Impact List | nonsense |
Protein Effect | loss of function - predicted |
Gene Variant Descriptions | TSC2 E114* results in a premature truncation of the Tsc2 protein at amino acid 114 of 1807 (UniProt.org). E114* has not been characterized however, due to the effects of other truncation mutations downstream of E114 (PMID: 22903760, PMID: 31454656), is predicted to lead to a loss of Tsc2 protein function. |
Associated Drug Resistance | |
Category Variants Paths |
TSC2 mutant TSC2 inact mut TSC2 E114* |
Transcript | NM_000548.5 |
gDNA | chr16:g.2054299G>T |
cDNA | c.340G>T |
Protein | p.E114* |
Source Database | RefSeq |
Genome Build | GRCh38/hg38 |
Transcript | gDNA | cDNA | Protein | Source Database | Genome Build |
---|---|---|---|---|---|
XM_017023615.1 | chr16:g.2054299G>T | c.340G>T | p.E114* | RefSeq | GRCh38/hg38 |
NM_000548.5 | chr16:g.2054299G>T | c.340G>T | p.E114* | RefSeq | GRCh38/hg38 |
NM_001406664.1 | chr16:g.2054299G>T | c.340G>T | p.E114* | RefSeq | GRCh38/hg38 |
NM_001363528.2 | chr16:g.2054299G>T | c.340G>T | p.E114* | RefSeq | GRCh38/hg38 |
XM_011522636.3 | chr16:g.2054299G>T | c.340G>T | p.E114* | RefSeq | GRCh38/hg38 |
NM_001406663.1 | chr16:g.2054299G>T | c.340G>T | p.E114* | RefSeq | GRCh38/hg38 |
XM_011522637.3 | chr16:g.2054299G>T | c.340G>T | p.E114* | RefSeq | GRCh38/hg38 |
NM_001406671.1 | chr16:g.2054299G>T | c.340G>T | p.E114* | RefSeq | GRCh38/hg38 |
XM_005255529.4 | chr16:g.2054299G>T | c.340G>T | p.E114* | RefSeq | GRCh38/hg38 |
NM_001077183.3 | chr16:g.2054299G>T | c.340G>T | p.E114* | RefSeq | GRCh38/hg38 |
NM_021055.3 | chr16:g.2054299G>T | c.340G>T | p.E114* | RefSeq | GRCh38/hg38 |
NM_001114382.3 | chr16:g.2054299G>T | c.340G>T | p.E114* | RefSeq | GRCh38/hg38 |
NM_000548.4 | chr16:g.2054299G>T | c.340G>T | p.E114* | RefSeq | GRCh38/hg38 |
XM_011522636.2 | chr16:g.2054299G>T | c.340G>T | p.E114* | RefSeq | GRCh38/hg38 |
NM_001370405.1 | chr16:g.2054299G>T | c.340G>T | p.E114* | RefSeq | GRCh38/hg38 |
NM_001406665.1 | chr16:g.2054299G>T | c.340G>T | p.E114* | RefSeq | GRCh38/hg38 |
XM_017023616.1 | chr16:g.2054299G>T | c.340G>T | p.E114* | RefSeq | GRCh38/hg38 |
XM_011522637.2 | chr16:g.2054299G>T | c.340G>T | p.E114* | RefSeq | GRCh38/hg38 |
NM_001370404.1 | chr16:g.2054299G>T | c.340G>T | p.E114* | RefSeq | GRCh38/hg38 |
NM_001077183.2 | chr16:g.2054299G>T | c.340G>T | p.E114* | RefSeq | GRCh38/hg38 |
XM_024450413.1 | chr16:g.2054299G>T | c.340G>T | p.E114* | RefSeq | GRCh38/hg38 |
XM_011522639.3 | chr16:g.2054299G>T | c.340G>T | p.E114* | RefSeq | GRCh38/hg38 |
XM_011522639.2 | chr16:g.2054299G>T | c.340G>T | p.E114* | RefSeq | GRCh38/hg38 |
NM_001406673.1 | chr16:g.2054299G>T | c.340G>T | p.E114* | RefSeq | GRCh38/hg38 |
XM_011522640.2 | chr16:g.2054299G>T | c.340G>T | p.E114* | RefSeq | GRCh38/hg38 |
XM_005255531.4 | chr16:g.2054299G>T | c.340G>T | p.E114* | RefSeq | GRCh38/hg38 |
NM_001114382.2 | chr16:g.2054299G>T | c.340G>T | p.E114* | RefSeq | GRCh38/hg38 |
Molecular Profile | Indication/Tumor Type | Response Type | Therapy Name | Approval Status | Evidence Type | Efficacy Evidence | References |
---|---|---|---|---|---|---|---|
TSC2 inact mut | perivascular epithelioid cell tumor | predicted - sensitive | Nab-rapamycin | Phase II | Actionable | In a Phase II trial (AMPECT), Fyarro (nab-rapamycin) treatment in patients with perivascular epithelioid cell tumors (PEComas) resulted in partial response in 20% (1/5) of patients with TSC1 mutations, 89% (8/9) of patients with TSC2 mutations, and 9% (1/11) of patients without a TSC1 or TSC2 mutation (PMID: 34637337; NCT02494570). | 34637337 |
TSC2 inact mut | clear cell renal cell carcinoma | not applicable | N/A | Guideline | Risk Factor | Germline inactivating mutations in TSC2 result in tuberous sclerosis complex (TSC), which is associated with increased risk of developing clear cell renal cell carcinoma (NCCN.org). | detail... |
TSC2 inact mut | Advanced Solid Tumor | no benefit | LY3023414 | Case Reports/Case Series | Actionable | In a Phase II trial (NCI-COG Pediatric MATCH), Samotolisib (LY3023414) treatment resulted in no objective responses in pediatric patients with advanced solid tumors harboring PI3K/mTOR pathway mutations, including PTEN (n=6), PIK3CA (n=5), TSC1 (n=2), TSC2 (n=3), and PIK3R1 (n=1), and resulted in a 3-month progression-free survival of 12%, a 6-month overall survival of 44%, and a 12-month overall survival of 15% (PMID: 39298693; NCT03155620). | 39298693 |
TSC2 inact mut | castration-resistant prostate carcinoma | predicted - sensitive | CC-115 + Enzalutamide | Phase I | Actionable | In a Phase Ib trial, Xtandi (enzalutamide) plus CC-115 was safe and led to a PSA reduction >= 50% (PSA50) in 80% (32/40) and >=90% (PSA90) in 58% (23/40) of metastatic castration-resistant prostate cancer patients at 12 weeks, and patients with PIK3CA activating mutations or PTEN or TSC1/2 loss of function (n=16) achieved a PSA50, PSA90, and median radiographic progression-free survival of 94%, 63%, and 19.6 mo vs 67%, 47%, and 22.1 mo in PI3K pathway wild-type patients (n=15) (PMID: 37980367; NCT02833883). | 37980367 |
TSC2 inact mut | renal cell carcinoma | predicted - sensitive | Everolimus | Clinical Study - Cohort | Actionable | In a retrospective analysis, 28% (12/43) of metastatic renal cell carcinoma patients who responded to rapalogs, Afinitor (everolimus) or Torisel (temsirolimus), harbored inactivating TSC1, TSC2 mutations and/or activating MTOR mutations, compared to 11% (4/36) in patients who did not respond to therapy (PMID: 26831717). | 26831717 |
TSC2 inact mut | transitional cell carcinoma | no benefit | Sapanisertib | Phase II | Actionable | In a Phase II trial, treatment with Sapanisertib (MLN0128) in metastatic urothelial carcinoma patients with either a TSC1 or TSC2 activating mutation (n=13) did not result in an objective response and led to a median overall survival of 3.4 months, and the trial was terminated early due to limited clinical activity and poor drug tolerability (Journal of Clinical Oncology 39, no. 6_suppl (February 20, 2021) 431-431; NCT03047213). | detail... |
TSC2 inact mut | islet cell tumor | not applicable | N/A | Guideline | Risk Factor | Germline inactivating mutations in TSC2 result in tuberous sclerosis complex (TSC), which is associated with increased risk of developing pancreatic neuroendocrine tumors (NCCN.org). | detail... |
TSC2 inact mut | renal cell carcinoma | predicted - sensitive | Temsirolimus | Clinical Study - Cohort | Actionable | In a retrospective analysis, 28% (12/43) of metastatic renal cell carcinoma patients who responded to rapalogs, Afinitor (everolimus) or Torisel (temsirolimus), harbored inactivating TSC1, TSC2 mutations and/or activating MTOR mutations, compared to 11% (4/36) in patients who did not respond to therapy (PMID: 26831717). | 26831717 |
TSC2 inact mut | Advanced Solid Tumor | no benefit | Everolimus | Phase II | Actionable | In a Phase II trial, Afinitor (everolimus) treatment resulted in limited activity with an objective response rate of 7% (2/30, 2 partial responses), stable disease in an additional 40% (12/30), a clinical benefit rate of 13% (4/30), a median progression-free survival of 2.3 months, and a median overall survival of 7.3 months in patients with advanced solid tumors harboring inactivating TSC1 or TSC2 mutations or activating MTOR mutations (PMID: 33727259; NCT02201212). | 33727259 |
TSC2 inact mut | Advanced Solid Tumor | predicted - sensitive | Nab-rapamycin | Clinical Study | Actionable | In a clinical study, Fyarro (nab-rapamycin) treatment in patients with advanced solid tumors harboring mutations in TSC1 or TSC2 led to a partial response in 4 patients, stable disease in 2 patients and progressive disease in 1 patient of 7 enrolled patients (J Clin Oncol 39, no. 15_suppl (May 20, 2021) 3111-3111; NCT03817515). | detail... |
TSC2 mutant | osteosarcoma | no benefit | Temsirolimus | Preclinical - Pdx | Actionable | In a preclinical study, Torisel (temsirolimus) treatment did not improve event-free survival in an osteosarcoma patient-derived xenograft (PDX) model harboring a TSC2 mutation (PMID: 37523146). | 37523146 |
TSC2 mutant | renal cell carcinoma | predicted - sensitive | Temsirolimus | Case Reports/Case Series | Actionable | In a clinical study, treatment with Afinitor (everolimus) or Torisel (temsirolimus) resulted in more partial responses (odds ratio = 0.08, p=0.030) in patients with renal cell carcinoma harboring mTOR pathway mutations, including MTOR (n=8), TSC1 (n=1), and TSC2 (n=2), than those without mutations (n=76) (PMID: 31335987). | 31335987 |
TSC2 mutant | subependymal giant cell astrocytoma | predicted - sensitive | Everolimus | Phase III | Actionable | In a Phase III trial (EXIST-1), Afinitor (everolimus) treatment resulted in a 50% or more tumor reduction in 35% (27/78) of adult and pediatric patients diagnosed with tuberous sclerosis complex and had subependymal giant cell astrocytoma, compared to 0% (0/39) in the placebo group, 85% (99/117) of the patients harbored mutations in TSC1 and/or TSC2 (PMID: 23158522; NCT00789828). | 23158522 |
TSC2 mutant | hepatocellular carcinoma | decreased response | Sorafenib | Clinical Study - Cohort | Actionable | In a clinical case study, Nexavar (sorafenib) treatment of patients with hepatocellular carcinoma harboring Mtor pathway mutations in PIK3CA, PTEN, TSC2, or TSC1 (n=12), resulted in a lower disease control rate (8.3% vs. 40.2%), shorter progression-free survival (1.9 months vs. 5.3 months) and shorter overall survival (10.4 months vs. 17.9 months) compared to patients without mutations in this pathway (n=67) (PMID: 30373752; NCT01775072). | 30373752 |
TSC2 mutant | lung non-small cell carcinoma | no benefit | Vistusertib | Case Reports/Case Series | Actionable | In a Phase II trial (NLMT), Vistusertib (AZD2014) treatment did not result in a confirmed response (0/5) or durable clinical benefit (0/5) in patients with non-small cell lung cancer harboring TSC1 or TSC2 mutations, thus the cohort was closed due to futility (PMID: 32669708, NCT02664935). | 32669708 |
TSC2 mutant | renal cell carcinoma | conflicting | Everolimus | Case Reports/Case Series | Actionable | In a clinical study, treatment with Afinitor (everolimus) or Torisel (temsirolimus) resulted in more partial responses (odds ratio = 0.08, p=0.030) in patients with renal cell carcinoma harboring mTOR pathway mutations, including MTOR (n=8), TSC1 (n=1), and TSC2 (n=2), than those without mutations (n=76) (PMID: 31335987). | 31335987 |
TSC2 mutant | renal cell carcinoma | conflicting | Everolimus | Clinical Study - Cohort | Actionable | In a retrospective analysis, TSC1, TSC2, or MTOR mutation status was not associated with progression-free survival in renal cell carcinoma patients treated with Afinitor (everolimus) (PMID: 30327302). | 30327302 |
TSC2 mutant | osteosarcoma | no benefit | Irinotecan + Temozolomide + Temsirolimus | Case Reports/Case Series | Actionable | In a clinical case study, Torisel (temsirolimus), Temodar (temozolomide), and Camptosar (irinotecan) combination treatment resulted in progressive disease in a pediatric patient with osteosarcoma harboring a TSC2 mutation (PMID: 37523146; NCT03336931). | 37523146 |