9  Indolent Systemic Mastocytois

9.1 Avapritinib

9.1.1 PIONEER trial

  • S/E: Photosensitivity, edema, cognitive effects

  • 25% of patients on POINEER was on 50mg dose, which is associated with higher rate of AEs

9.2 Bezuclastinib

9.2.1 SUMMIT trial

  • Part A

  • Part B

A global, randomized, double-blind, placebo-controlled, 3-part phase 2 clinical trial of bezuclastinib in adult patients with nonadvanced systemic mastocytosis

9.2.1.1 SUMMIT Trial: Bezuclastinib in NonAdvSM Patients - Summary Report

9.2.1.1.1 Executive Summary

The SUMMIT Part 2 trial evaluated bezuclastinib versus placebo in patients with non-advanced systemic mastocytosis (NonAdvSM). The study met its primary endpoint and all secondary endpoints with statistically significant results, demonstrating meaningful symptom reduction and objective measures of disease burden improvement.

9.2.1.1.2 Study Design

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SUMMIT Part 2 Study Design Overview
Parameter Details
Study Type Double-blind, placebo-controlled, randomized
Primary Endpoint Mean change in Total Symptom Score (TSS) at Week 24
Treatment Arms Bezuclastinib 100mg QD + BSC vs Placebo + BSC
Randomization 2:1 (Bezuclastinib:Placebo)
Study Duration 24 weeks with open-label extension
Sample Size 179 patients (119 bezuclastinib, 60 placebo)
Data Cutoff May 22, 2025
9.2.1.1.3 Patient Demographics and Baseline Characteristics
Patient Demographics and Baseline Characteristics
Characteristic Bezuclastinib (N=119) Placebo (N=60) Overall (N=179)
Total Patients 119 60 179
Female (%) 74 (62.2%) 44 (73.3%) 118 (65.9%)
Median Age (range) 51 (24-73) 52 (23-78) 51 (23-78)
ECOG PS 0 (%) 58 (48.7%) 30 (50.0%) 88 (49.2%)
ECOG PS 1 (%) 50 (42.0%) 26 (43.3%) 76 (42.5%)
ECOG PS 2 (%) 11 (9.2%) 4 (6.7%) 15 (8.4%)
Disease Characteristics at Baseline
Characteristic Bezuclastinib (N=119) Placebo (N=60) Overall (N=179)
Indolent SM (ISM) 97 (81.5%) 50 (83.3%) 147 (82.1%)
Smoldering SM (SSM) 8 (6.7%) 4 (6.7%) 12 (6.7%)
Bone Marrow Mastocytosis (BMM) 14 (11.8%) 6 (10.0%) 20 (11.2%)
Median KIT D816V (%) 0.22 (0-32) 0.30 (0-34) 0.25 (0-34)
Below Detection Limit 28 (23.5%) 12 (20.0%) 40 (22.3%)
Median BM MC Burden (%) 10 (1-75) 10 (1-75) 10 (1-75)
Median Serum Tryptase (ng/mL) 40 (6-448) 41 (7-692) 40 (6-692)
Serum Tryptase < 20 ng/mL 22 (18.5%) 10 (16.7%) 32 (17.9%)
9.2.1.1.4 Primary and Secondary Endpoints Results
Primary and Secondary Endpoint Results
Endpoint Bezuclastinib Placebo P-Value Endpoint Type
Mean Change in TSS at Week 24 -24.32 (-27.56, -21.08) -15.41 (-19.58, -11.24) 0.0002 Primary
≥50% Reduction in Serum Tryptase 87.4% 0% <0.0001 Secondary
≥50% Reduction in KIT D816V VAF Data not shown Data not shown <0.0001 Secondary
≥50% Reduction in TSS Data not shown Data not shown 0.0142 Secondary
≥50% Reduction in Bone Marrow MC Data not shown Data not shown <0.0001 Secondary
≥30% Reduction in TSS Data not shown Data not shown 0.0004 Secondary
Mean Change in Most Severe Symptom Score Data not shown Data not shown 0.0001 Secondary
Treatment Effect Size Analysis
Measure Value Clinical Interpretation
Placebo-Adjusted Effect Size -8.91 (-13.56, -4.26) Clinically meaningful reduction
Percent Improvement vs Placebo 57% improvement over placebo Substantial benefit vs control
9.2.1.1.5 Safety Profile
Safety Overview
Safety Parameter Bezuclastinib (N=118) Placebo (N=60)
Any TEAE 116 (98.3%) 53 (88.3%)
Serious AEs 5 (4.2%) 3 (5.0%)
Dose Reductions due to TRAEs 13 (11.0%) 0
Discontinuations due to TRAEs 7 (5.9%) 0
Grade 1 TEAEs 70% of all TEAEs Not specified
≥Grade 3 ALT/AST Elevations 5.9% Not specified
Treatment-Emergent Adverse Events (>10% in Bezuclastinib Arm)
Adverse Event Bezuclastinib (N=118) Placebo (N=60)
Hair color changes 82 (69.5%) 3 (5.0%)
Altered taste 28 (23.7%) 0
Nausea 26 (22.0%) 8 (13.3%)
ALT/AST increased 26 (22.0%) 4 (6.6%)
Headache 21 (17.8%) 7 (11.7%)
Alopecia 14 (11.9%) 2 (3.3%)
ALP increased 12 (10.2%) 2 (3.3%)
9.2.1.1.6 Case Studies Summary
Representative Case Study Outcomes
Patient Baseline TSS Week 24 TSS % Reduction Tryptase Reduction Key Outcomes
Case 1: 44yo Woman with ISM 67.9 27.8 68% 91% (39.4 → 3.4 ng/mL) Discontinued all BSC meds in OLE, continued benefit >1 year
Case 2: 63yo Man with SM 75.9 19.5 74% 92% (95.5 → 7.9 ng/mL) Discontinued 3 BSC meds in OLE, continued benefit 32 weeks

9.3 Market Opportunity and Pipeline

Bezuclastinib Development Pipeline and Market Opportunity
Study Indication Market Size Status Primary Endpoint
SUMMIT (NonAdvSM) Non-Advanced Systemic Mastocytosis $2+ billion US annual TLR complete (Today) Mean change in TSS at Week 24
PEAK (2nd-line GIST) Gastrointestinal Stromal Tumors $1+ billion US annual TLR expected EOY 2025 Progression Free Survival
APEX (AdvSM) Advanced Systemic Mastocytosis $300 million US annual TLR expected 2H 2025 Overall Response Rate
9.3.0.0.1 Key Clinical Takeaways
Key Clinical Takeaways from SUMMIT Trial
Category Key Finding Clinical Significance
Efficacy 24.3 point TSS reduction vs 15.4 point placebo reduction Clinically meaningful symptom improvement
Efficacy 8.91 point placebo-adjusted effect (57% improvement) Superior to existing approved KIT inhibitors
Safety 70% of TEAEs were Grade 1; only 5.9% discontinued Favorable tolerability for chronic use
Biomarkers 87.4% achieved ≥50% serum tryptase reduction Objective evidence of disease modification
Clinical Utility Patients able to discontinue background medications Potential to simplify treatment regimens
9.3.0.0.2 Regulatory and Commercial Timeline
Regulatory and Commercial Timeline
Milestone Status/Timing Significance
SUMMIT TLR Complete (July 7, 2025) Primary trial success for NonAdvSM
NDA Submission Planned by end of 2025 Registration filing
PEAK TLR Expected EOY 2025 2nd indication data
APEX TLR Expected 2H 2025 3rd indication data
Expanded Access Program Currently active in US Patient access while pending approval
9.3.0.0.3 Statistical Summary
Statistical Analysis Summary
Analysis Type Result Interpretation
Primary Endpoint Power p = 0.0002 (highly significant) Strong evidence of efficacy
All Secondary Endpoints All met statistical significance Consistent benefit across measures
Effect Size Cohen’s d estimate: Large effect Substantial treatment benefit
Clinical Significance Exceeds minimal clinically important difference Meaningful patient benefit

Data Source: Cogent Biosciences SUMMIT Trial Investor Webcast, July 7, 2025
Analysis Date: `r Sys.Date()# SUMMIT Trial: Bezuclastinib in NonAdvSM Patients - Summary Report

9.4 Avapritinib vs Bezuclastinib

This analysis compares the SUMMIT trial (bezuclastinib) with the PIONEER trial (avapritinib), both randomized, double-blind, placebo-controlled studies in patients with non-advanced systemic mastocytosis (NonAdvSM). Both trials met their primary endpoints but show distinct efficacy and safety profiles.

9.4.1 Study Design Comparison

Study Design Comparison: SUMMIT vs PIONEER
Parameter SUMMIT (Bezuclastinib) PIONEER (Avapritinib)
Study Name SUMMIT Part 2 PIONEER Part 2
Drug/Dose Bezuclastinib 100mg QD Avapritinib 25mg QD
Primary Population NonAdvSM (ISM, SSM, BMM) Indolent SM (ISM only)
Randomization 2:1 2:1
Sample Size 179 (119 vs 60) 212 (141 vs 71)
Primary Endpoint Mean change MS2D2 TSS at Week 24 Mean change ISM-SAF TSS at Week 24
Study Duration 24 weeks + OLE 24 weeks + OLE
Status Complete (July 2025) Complete, FDA approved

9.4.2 Patient Population Comparison

Patient Population Characteristics
Characteristic SUMMIT (Bezuclastinib) PIONEER (Avapritinib)
Total Randomized 179 212
Median Age 51 years ~50 years
Female (%) 65.9% ~65%
ECOG PS 0-1 (%) 91.7% ~95%
Prior KIT Inhibitor (%) 12.3% Not specified
NonAdvSM Subtypes ISM 82.1%, SSM 6.7%, BMM 11.2% ISM 100%
Median Baseline TSS 55.6 (MS2D2) ~53 (ISM-SAF)
Median Serum Tryptase (ng/mL) 40 Not specified
Median KIT D816V VAF (%) 0.25 Not specified

9.4.3 Primary Efficacy Results Comparison

Primary Endpoint Results Comparison
Metric SUMMIT (Bezuclastinib) PIONEER (Avapritinib)
Mean TSS Reduction at Week 24 -24.32 points (95% CI: -27.56, -21.08) -15.6 points (95% CI: -18.6, -12.6)
Placebo TSS Reduction -15.41 points (95% CI: -19.58, -11.24) -9.2 points (95% CI: -13.1, -5.2)
Placebo-Adjusted Effect -8.91 points (95% CI: -13.56, -4.26) -6.4 points
P-Value 0.0002 0.003
Effect Size Magnitude 57% improvement over placebo 41% improvement over placebo

9.4.4 Secondary Efficacy Endpoints

Secondary Endpoints Comparison
Endpoint SUMMIT (Bezuclastinib) PIONEER (Avapritinib)
≥50% TSS Reduction Not specified 24.8% vs 9.9% placebo (p=0.005)
≥30% TSS Reduction Not specified 45.4% vs 29.6% placebo (p=0.009)
≥50% Serum Tryptase Reduction 87.4% vs 0% placebo (p<0.0001) 53.9% vs 0% placebo (p<0.0001)
≥50% KIT D816V VAF Reduction Met endpoint (p<0.0001) Met endpoint
≥50% Bone Marrow MC Reduction Met endpoint (p<0.0001) Met endpoint
Mean TSS at 48 weeks (OLE) Not yet reported -20.2 points improvement

9.4.5 Safety Profile Comparison

Safety Profile Comparison
Safety Parameter SUMMIT (Bezuclastinib) PIONEER (Avapritinib)
Any TEAE (%) 98.3% (vs 88.3% placebo) 90.8% (vs 93% placebo)
Serious AEs (%) 4.2% (vs 5.0% placebo) 5.0% (vs 11.3% placebo)
Discontinuation due to TRAEs (%) 5.9% (vs 0% placebo) 0.7% (vs 0% placebo)
Dose Reductions due to TRAEs (%) 11.0% (vs 0% placebo) Not specified
Treatment Completion Rate (%) Not specified 96.5% (vs 93% placebo)
Grade ≥3 AEs (%) Not specified 21% (vs 21% placebo)

9.4.6 Common Adverse Events Comparison

Common Adverse Events Comparison (>5% incidence)
Adverse Event SUMMIT (Bezuclastinib) PIONEER (Avapritinib)
Hair color changes 69.5% (vs 5% placebo) Not reported
Edema (peripheral/periorbital) Not specified as major AE 12.8% combined (vs 4.2% placebo)
Nausea 22.0% (vs 13.3% placebo) 6.4% (vs 8.5% placebo)
Headache 17.8% (vs 11.7% placebo) 7.8% (vs 9.9% placebo)
Cognitive effects Not specified 7.8% (vs 7.0% placebo)
Altered taste/Flushing 23.7% altered taste (vs 0% placebo) ≥5% flushing
Hepatic (ALT/AST elevation) 22.0% (vs 6.6% placebo) Not major concern
Alkaline phosphatase elevation 10.2% (vs 3.3% placebo) Increased (≥5%)

9.4.7 Efficacy Superiority Analysis

Bezuclastinib Efficacy Advantages vs Avapritinib
Comparison Metric Bezuclastinib Advantage Clinical Interpretation
Absolute TSS Reduction 24.3 vs 15.6 points (56% greater) Clinically meaningful difference
Placebo-Adjusted Effect 8.9 vs 6.4 points (39% greater) Substantial additional benefit
Magnitude of Benefit Larger absolute and relative benefit Bezuclastinib more effective
Statistical Significance Stronger (p=0.0002 vs p=0.003) More robust statistical evidence
Tryptase Response Rate 87.4% vs 53.9% (62% relative advantage) More patients achieve biomarker response
Clinical Advantage Superior symptom control Better patient outcomes expected

9.4.8 Safety Profile Analysis

Safety Profile Analysis and Clinical Implications
Safety Domain Bezuclastinib Avapritinib Clinical Implication
Discontinuation Rate Higher (5.9% vs 0.7%) Lower (0.7%) Bezuclastinib: more monitoring needed
Edema/Fluid Retention Not prominent Notable (12.8% edema) Avapritinib: edema management required
Cognitive Effects Not reported as major issue Similar to placebo (7.8%) Both drugs: acceptable cognitive safety
Hepatic Safety Manageable, reversible ALT/AST elevations Not major concern Bezuclastinib: requires LFT monitoring
Hair/Cosmetic Changes Common (69.5%) but reversible Not reported Bezuclastinib: cosmetic but reversible
Overall Tolerability Generally well-tolerated despite higher TEAE rate Well-tolerated with lower completion rate Both suitable for chronic treatment

9.4.9 Biomarker Response Comparison

Biomarker Response Comparison
Biomarker SUMMIT (Bezuclastinib) PIONEER (Avapritinib) Advantage
≥50% Serum Tryptase Reduction 87.4% vs 0% placebo 53.9% vs 0% placebo Bezuclastinib: 62% relative advantage
KIT D816V VAF Response Significant reduction achieved Achieved in majority Both effective
Bone Marrow Mast Cell Reduction Significant reduction achieved Achieved in majority Both effective
Objective Disease Modification Strong evidence across all measures Good evidence of disease modification Bezuclastinib: more patients respond

9.4.10 Clinical Implications and Market Position

Clinical Implications and Strategic Considerations
Factor Bezuclastinib Avapritinib Strategic Consideration
Regulatory Status NDA submission planned Q4 2025 FDA approved May 2023 Bezuclastinib following approved comparator
Target Population Broader NonAdvSM (ISM, SSM, BMM) ISM only Bezuclastinib targets broader patient population
Efficacy Positioning Superior TSS reduction and tryptase response Clinically meaningful benefit Bezuclastinib shows superior efficacy metrics
Safety Considerations Manageable with monitoring Well-characterized, manageable Both have manageable safety profiles
Clinical Utility May enable BSC medication discontinuation Established clinical experience Both enable improved patient management
Market Differentiation Potentially best-in-class efficacy First-to-market advantage Competition based on efficacy vs first-mover advantage

9.4.11 Summary

Summary of Key Differences and Clinical Bottom Line
Domain Bezuclastinib Advantage Avapritinib Advantage Clinical Bottom Line
Primary Efficacy 56% greater TSS reduction Established efficacy Bezuclastinib shows superior symptom control
Biomarker Response 62% higher tryptase response rate Proven disease modification Bezuclastinib achieves higher response rates
Patient Population Broader NonAdvSM subtypes ISM focus with deep experience Bezuclastinib addresses unmet need in SSM/BMM
Safety Profile Predictable, manageable AEs Lower discontinuation rate Both suitable for chronic use
Market Position Potential best-in-class First approved, established Market competition likely
Clinical Experience Newer data, recent results 2+ years clinical experience Both represent major advances

Conclusions Efficacy Advantage: Bezuclastinib

Superior symptom reduction: 24.3 vs 15.6 point TSS decrease (56% greater) Better biomarker response: 87.4% vs 53.9% tryptase reduction (62% advantage) Stronger statistical significance: p=0.0002 vs p=0.003

Population Advantage: Bezuclastinib

Broader indication: Includes SSM and BMM subtypes beyond ISM Addresses unmet need: No approved therapies for SSM

Safety Considerations

Avapritinib: Lower discontinuation rate, established safety profile Bezuclastinib: Higher efficacy with manageable safety, requires monitoring

Market Dynamics

Avapritinib: First-mover advantage, established market presence Bezuclastinib: Potential best-in-class positioning based on superior efficacy data

Clinical Bottom Line: Bezuclastinib demonstrates superior efficacy across multiple endpoints compared to avapritinib, with the potential to set a new standard of care for NonAdvSM patients, while avapritinib maintains advantages in established clinical experience and current market access.

9.5 Elenestanib

  • a novel oral next generation TKI with limited CNS penetration.

9.5.1 HARBOR trial

9.6 TL-895

  • BTKi

9.6.1 TELIO study

  • This study evaluates TL-895, a potent, orally-available and highly selective irreversible tyrosine kinase inhibitor for the treatment of Myelofibrosis (Cohorts 1-3) or Indolent Systemic Mastocytosis (Cohort 5).