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ONC201

Phase 3

Diffuse Intrinsic Pontine Glioma | Small molecule | Oncology |Jazz Pharmaceuticals plc|Last Updated: Feb 5, 2026

Target and mechanism

Molecular targetClpP
Target classMitochondrial Protease
ModalitySmall molecule

Success Probability

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Market & Valuation

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Trial Design

RandomizedCONTROLLEDDMCBiomarker
Total Trials2
Total Enrollment793

FDA Designations

No designations recorded

Clinical trial landscape

ONC201 · 2 trials · 7 indications

Phase 3 1Phase 2 1
NCT05476939Biological Medicine for Diffuse Intrinsic Pontine Glioma (DIPG) Eradication 2.0Diffuse Intrinsic Pontine Glioma
RECRUITING433 Analytics
PHASE3RECRUITING
Biological Medicine for Diffuse Intrinsic Pontine Glioma (DIPG) Eradication 2.0
Diffuse Intrinsic Pontine GliomaUnlock trial analytics

Study Endpoints

Primary Endpoints

Progression-free survival
Until 2 years after inclusion of the last patient

Defined as the time between date of randomization and unequivocal clinical, cytological or radiological progression confirmed by central review, or death whatever the cause.

Progression-free survival at 6 months (PFS6) - Cohorts 1A, 1B Only
6 months after diagnosis

Percentage of participants alive and free from progression at 6 months after the diagnosis. The primary analysis for PFS6 is based on the intention to treat (ITT) population, according to treatment arm assignment. PFS6 is estimated using the Kaplan-Meier method with exact confidence intervals for each cohort and arm. Participants with unknown progression status at 6 months are considered failures (i.e., progressed) for the PFS6 analysis.

Progression-free survival at 6 months (PFS6) - Cohorts 2A, 2B Only
6 months after diagnosis

Percentage of participants alive and free from progression at 6 months after diagnosis. The primary analysis for PFS6 is based on the intention to treat (ITT) population, according to treatment arm assignment. PFS6 is estimated using the Kaplan-Meier method with exact confidence intervals for each cohort and arm. Participants with unknown progression status at 6 months are considered failures (i.e., progressed) for the PFS6 analysis.

Overall survival at 7 months (OS7) - Cohort 3A & 3B Only
7 months after administration of ONC201 in the maintenance phase

OS7 is defined as the percentage of participants alive at 7 months after the initiation of the combination of the backbone (i.e., ONC201) with a novel agent given in the maintenance phase of therapy. The primary analysis for OS7 is based on the ITT population, according to treatment arm assignment. OS7 is estimated using the Kaplan-Meier method with exact confidence intervals for each cohort and arm. Participants with unknown survival status at 7 months are considered failures (i.e., dead) for the OS7 analysis.

Proportion of participants reporting dose-limiting toxicities (DLTs) (Cohort 4)
Up through the first cycle of maintenance therapy, approximately 8 months

The safety and tolerability of ONC201 at a previously untested dose will be measured by the proportion of participants reporting a DLT within the first cycle of treatment.

Number of participants requiring dose modification through first cycle of maintenance (Cohort 5)
Up through the first cycle of maintenance therapy, approximately 8 months

The safety and tolerability of ONC201 in combination with targeted therapies will be measured by the number of participants reporting dose modification during first cycle.

Maximum tolerated number of intratumor infusions of DNX-2401, with a maximum of 6, in participants with thalamic or pontine DMG who have completed radiotherapy (Cohort 6)
A maximum of 6 infusions will be administered every 30 days - approximately 8 months

The safety and tolerability of DNX-2401 at Dose Level 1 (5 × 10\^10 viral particles) will be measured by the proportion of participants reporting a DLT.

Secondary Endpoints

Overall survival (for all the comparisons to historical controls)
Until 5 years after randomization of the last patient
Overall survival (for the internal comparison between randomized groups)
Until 5 years after randomization of the last patient
Progression-free survival after first progression
Until 5 years after randomization of the last patient
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Study Design & Arms

AllocationRANDOMIZED
MaskingNONE
ModelCROSSOVER
PurposeTREATMENT

Treatment Arms

ArmTypeDescription
everolimusACTIVE_COMPARATORTablets of 2.5 mg or 10 mg. The prescribed dose is 5 mg/m²/day, orally, once daily. Dose will be capped at 10 mg once daily. Treatment will be continued until unacceptable toxicity, centrally confirmed tumor progression (either radiologically or histologically) and/or withdrawal of patient, parents or legal representative consent. At the time of centrally confirmed relapse or progression, patients will stop treatment and will be allowed to switch to the other arm in case no better option is available after considering the results of the molecular profiling. In case of switch (everolimus to ONC201 or vice-versa), the patient will observe a wash-out period of 7 days before starting: * the second treatment, * or a reirradiation (if applicable). No treatment is allowed during reirradiation, except setroids and bevacizumab. Then, if an additional treatment is needed, the second treatment will be started within one week after the end of the reirradiation.
Dordaviprone (ONC201)EXPERIMENTALCapsules of 125 mg. The prescribed dose is 375 mg/m², orally, once daily at Day 1 and Day 2 of each week. Dose will be capped at 625 mg per dose. Treatment will be continued until unacceptable toxicity, centrally confirmed tumor progression (either radiologically or histologically), and/or withdrawal of patient, parents or legal representative consent. At the time of centrally confirmed relapse or progression, patients will stop treatment and will be allowed to switch to the other arm in case no better option is available after considering the results of the molecular profiling. In case of switch (everolimus to ONC201 or vice-versa), the patient will observe a wash-out period of 7 days before starting: * the second treatment, * or a reirradiation (if applicable). No treatment is allowed during reirradiation, except setroids and bevacizumab. Then, if an additional treatment is needed, the second treatment will be started within one week after the end of the reirradiation.
NOT CURRENTLY ENROLLING - ARM 2: ONC201 (Day -1), Radiation+ONC201, Paxalisib+ONC201EXPERIMENTALParticipants may receive a safety lead in of ONC201. During the trial validation phase, participants without prior biopsy receive ONC201 PO on day -1 prior to standard of care biopsy. During the radiation/re-irradiation phase, participants without prior radiation therapy or have disease progression after radiation therapy undergo weekly radiation therapy and receive ONC201 PO weekly during radiation therapy. During the maintenance phase, participants receive ONC201 PO weekly and paxalisib PO daily (QD). Cycles repeat every 28 days (4 weeks) in the absence of adverse events of unacceptable toxicity
NOT CURRENTLY ENROLLING - ARM 4: ONC201 (Day -1,-2), Radiation+ONC201, Paxalisib+ONC201EXPERIMENTALParticipants may receive a safety lead in of ONC201. During the trial validation phase, participants without prior biopsy receive ONC201 PO on days -2 and -1 prior to standard of care biopsy. During the radiation/re-irradiation phase, participants may receive ONC201 PO weekly during radiation therapy. During the maintenance phase, participants receive ONC201 PO weekly and paxalisib PO QD. Cycles repeat every 28 days (4 weeks) in the absence of adverse events or unacceptable toxicity
NOT CURRENTLY ENROLLING - ARM 6: Paxalisib (Day -1), Radiation+Paxalisib , Paxalisib+ONC201EXPERIMENTALParticipants may receive a safety lead in of ONC201. During trial validation phase, participants without prior biopsy receive paxalisib PO on day -1 prior to standard of care biopsy. During the radiation/re-irradiation phase, participants without prior radiation therapy or have disease progression after radiation therapy undergo weekly radiation therapy and receive paxalisib PO daily during radiation therapy. During the maintenance phase, participants receive ONC201 PO weekly and paxalisib PO QD. Cycles repeat every 28 days (4 weeks) in the absence of adverse events of unacceptable toxicity
Cohort 4 - Dose Escalation, Starting Dose 2 (625mg ONC201)EXPERIMENTALParticipants will receive a safety lead in of 625mg (or weight-adjusted adult equivalent RP2D for pediatrics) of ONC201 on Day 1 and 2 of each week. During the target validation phase, participants without prior biopsy receive ONC201 PO on days -2 and -1 prior to standard of care biopsy. For participants receiving non-interventional radiation/re-irradiation per standard of care treatment, participants will receive 625 mg as the starting dose of ONC201 Days 1 and 2 on a weekly basis during radiation. Cycles repeat every 28 days (4 weeks) in the absence of adverse events or unacceptable toxicity
Cohort 5 - ONC201 + Targeted therapiesEXPERIMENTALParticipants will receive a starting dose of 625mg (or weight-adjusted adult equivalent RP2D for pediatrics) of ONC201 on Day 1 and 2 of each week in combination with targeted agents to be selected from approved/available agents based on a rational therapy approach guided by molecular data from the tumor tissue or cerebral spinal fluid (CSF). For participants receiving non-interventional radiation/re-irradiation per standard of care treatment, prior to starting the combination therapy, participants will receive 625 mg as the starting dose of ONC201 Days 1 and 2 on a weekly basis during radiation. Observations and schedule of events will be issued based on the chosen agent determined to best fit the molecular profile (e.g. BRAFV600E, PDGFRA, FGFR1, NF1).
Cohort 6 - DNX-2401 Repeated Intratumoral AdministrationEXPERIMENTALParticipants will receive repeated DNX-2401 intratumoral infusions every 30 days, for a maximum of six injections. During Infusion 1 and Infusion 3, participants will have a biopsy for tumor tissue collection.

Interventions

NameTypeDescription
EverolimusDRUGTablets of 2.5 mg or 10 mg. The prescribed dose is 5 mg/m²/day, orally, once daily. Dose will be capped at 10 mg once daily.
ONC201DRUGCapsules of 125mg. The prescribed dose is 375mg/m², orally, once daily at Day 1 and Day 2 of each week. Dose will be capped at 625 mg per dose.
RadiotherapyRADIATIONAll patients will be treated with 30 conventional single daily fractions of 1.8 Gy to a total of 54 Gy over a planned period of 6 weeks. Dose may be increased up to 60 Gy for adult patients with supratentorial ND-DMG. The clinical target volume will include all the areas of abnormality on T2/FLAIR sequences with a 1-cm margin. Radiotherapy will have to start within a maximum of 4 weeks for DIPG, up to 6 weeks for other DMG H3K28-altered (ND-DMG), after the biopsy or last surgery. The study medication will be started at Day 1 (+3 days max) of radiotherapy. Reirradiation is permitted only at disease progression according to local practice. In case of metastatic disease or intramedullary tumors, patients can be included in the study. In this situation, radiotherapy will have to start within a maximum of 4 weeks for DIPG, up to 6 weeks for other DMG H3K28-altered (ND-DMG), after the biopsy or last surgery while targeted treatment will start at the end of the irradiation.
Radiation TherapyRADIATIONUndergo radiation therapy
PaxalisibDRUGGiven PO
DNX-2401DRUGDNX-2401 is an oncolytic adenovirus that will be administered through direct intratumoral infusion of DNX-2401 via a specialized Neuro Ventricular Cannula.
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Eligibility Criteria

Age Range6 Months to N/A
SexALL
Healthy VolunteersNo
Study Sites50

Eligibility criteria for the inclusion (registration) in BIOMEDE 2.0 study: * Diagnosis Criteria: * Diagnosis of DIPG (clinical and radiological). As biopsy is not standard for these tumors, an informed consent is required for the necessary histological verification. \[Biopsy-part of BIOMEDE 2.0...

Countries:DenmarkFranceSpainSwedenUnited StatesAustraliaIsraelNetherlandsNew ZealandSwitzerland
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Frequently asked questions about ONC201

What is ONC201 used for in Diffuse Intrinsic Pontine Glioma?

ONC201 is an investigational small molecule being studied for the treatment of Diffuse Intrinsic Pontine Glioma (DIPG), a type of brain tumor. It is also being evaluated in related conditions such as Diffuse Midline Glioma with H3 K27M mutations. The drug is currently in Phase 3 clinical development for these indications.

What does ONC201 target?

ONC201 targets ClpP, a mitochondrial protease. By acting on this mitochondrial target, the drug is designed to interfere with cancer cell metabolism and promote cell death. This mechanism is being studied in the context of Diffuse Intrinsic Pontine Glioma and other H3 K27M-mutant gliomas.

Who makes ONC201?

ONC201 is being developed by Jazz Pharmaceuticals plc, a biopharmaceutical company traded on the NASDAQ under the ticker JAZZ. The company is conducting clinical trials to evaluate the drug's safety and efficacy in patients with Diffuse Intrinsic Pontine Glioma and related conditions.

What phase is ONC201 in?

ONC201 is in Phase 3 clinical development for Diffuse Intrinsic Pontine Glioma. It is an investigational drug and has not been approved by regulatory authorities. The ongoing Phase 3 trial, NCT05476939, is actively recruiting patients to assess the treatment's effectiveness.

What clinical trials is ONC201 in?

ONC201 is being evaluated in two active clinical trials. NCT05476939 is a Phase 3 study enrolling 433 patients with Diffuse Intrinsic Pontine Glioma and Diffuse Midline Glioma in Denmark, France, Spain, and Sweden. NCT05009992 is a Phase 2 trial with 360 participants studying combination therapy for diffuse midline gliomas across multiple countries.

Is ONC201 the same as any other drug?

ONC201 is the primary name for this investigational drug. No alternative names have been reported for this compound. It is being studied under this designation in clinical trials for Diffuse Intrinsic Pontine Glioma and related H3 K27M-mutant gliomas.