A Pilot Study of Larotrectinib for Newly-Diagnosed High-Grade Glioma With NTRK Fusion

NCT ID: NCT04655404

Last Updated: 2025-12-26

Study Results

Results pending

The study team has not published outcome measurements, participant flow, or safety data for this trial yet. Check back later for updates.

Basic Information

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Recruitment Status

RECRUITING

Clinical Phase

EARLY_PHASE1

Total Enrollment

15 participants

Study Classification

INTERVENTIONAL

Study Start Date

2021-04-08

Study Completion Date

2036-12-01

Brief Summary

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This is a pilot study that will evaluate disease status in children that have been newly diagnosed high-grade glioma with TRK fusion. The evaluation will occur after 2 cycles of the medication (Larotrectinib) have been given.

The study will also evaluate the safety of larotrectinib when given with chemotherapy in your children; as well as the safety larotrectinib when given post-focal radiation therapy.

Detailed Description

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In this pilot study, we will assess the disease control rate (Continued Complete Response-CCR, Complete Response-CR, Partial Response-PR and Stable Disease-SD) as well as survival rate (overall survival- OS and progression free survival- PFS) in children with newly diagnosed HGG with TRK fusion who receive 2 cycles of larotrectinib monotherapy administered orally, twice daily, at 100 mg/m2 continuously on a 28-day cycle schedule. After 2 monotherapy cycles of larotrectinib, patients with CCR or CR will continue to receive larotrectinib maintenance therapy as monotherapy for a total of 12 cycles. Continuation of treatment beyond 12 cycles, and up to 24 cycles, may be considered for patients on Larotrectinib monotherapy if they are receiving clinical benefit from the study, at the discretion of the treating physician.

Patients ≤ 48 months with PR or SD after 2 cycles of larotrectinib will go on to receive combination therapy with standard backbone chemotherapy (BABYPOG or HIT-SKK). Patients \> 48 months of age (or patients ≥ 36 months of age, or patients with DIPG \>18 months of age, at the discretion of the local investigator) will receive focal radiation therapy. A surgical cohort study will be explored whereby patients who have had a tumor biopsy/partial resection at their local institution and are planned to subsequently undergo definitive resection will receive 3-5 days (6-10 doses) of larotrectinib pre-surgery.

The study design of this trial requires 15 patients evaluable for disease control and for safety/ toxicity of larotrectinib as monotherapy. The surgical cohort will enroll up to 4 patients and will count towards the total 15 evaluable patients. A minimum of 6 patients will be evaluable for safety toxicity of larotrectinib in combination with standard-of-care chemotherapy or radiotherapy.

Conditions

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High Grade Glioma Diffuse Intrinsic Pontine Glioma

Keywords

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NTRK gene fusion BABYPOG HIT-SKK Larotrectinib

Study Design

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Allocation Method

NON_RANDOMIZED

Intervention Model

SINGLE_GROUP

Feasibility and surgical cohorts
Primary Study Purpose

TREATMENT

Blinding Strategy

NONE

Study Groups

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Surgical Cohort

Larotrectinib administered PO, BID @100 mg/m2 3-5 days prior to definitive surgery, followed by Larotrectinib administered PO, BID @100 mg/m2 on a 28-day cycle schedule.

Group Type EXPERIMENTAL

Larotrectinib

Intervention Type DRUG

1. Larotrectinib monotherapy x2 cycles followed by disease evaluation
2. Larotrectinib with or without chemotherapy backbone

Larotrectinib surgical

Intervention Type PROCEDURE

1. Surgical cohort: Larotrectinib x 3-5 days prior to definitive surgery followed by Larotrectinib monotherapy x2 cycles followed by disease evaluation
2. Larotrectinib with or without chemotherapy backbone

Feasibility Cohort

Larotrectinib administered PO, BID @100 mg/m2 on a 28-day cycle schedule.

Group Type EXPERIMENTAL

Larotrectinib

Intervention Type DRUG

1. Larotrectinib monotherapy x2 cycles followed by disease evaluation
2. Larotrectinib with or without chemotherapy backbone

Interventions

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Larotrectinib

1. Larotrectinib monotherapy x2 cycles followed by disease evaluation
2. Larotrectinib with or without chemotherapy backbone

Intervention Type DRUG

Larotrectinib surgical

1. Surgical cohort: Larotrectinib x 3-5 days prior to definitive surgery followed by Larotrectinib monotherapy x2 cycles followed by disease evaluation
2. Larotrectinib with or without chemotherapy backbone

Intervention Type PROCEDURE

Eligibility Criteria

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Inclusion Criteria

* Age: Patients ≤ 21 years of age (birth to 21 years of age) at the time of study enrollment will be eligible.
* Diagnosis: Patients with newly-diagnosed high-grade (HGG), including diffuse intrinsic pontine gliomas (DIPG), whose tumors are documented in a CLIA/CAP certified lab (or clinically equivalent method considered standard in non-US sites) to harbor an NTRK fusion alteration by FISH, PCR, or next generation sequencing are eligible. Patients must have had histologically verified high-grade glioma such as anaplastic astrocytoma, glioblastoma, or H3 K27-mutant diffuse midline glioma verified at a CONNECT site.

For sites that do not have CLIA-certified equivalent (certified laboratory) to assess NTRK fusion, testing will be conducted centrally at NCH. NTRK testing will be performed by NGS using targeted RNA-sequencing (Archer Solid Tumor analysis) Please submit 10 unstained sections on charged slides at 10uM thickness, or 10 scrolls cut at 10uM thickness, along with submission of an H\&E slide. Formalin-fixed paraffin embedded (FFPE) tissue block and FFPE tissue scroll specimens must contain minimum of 25% tumor Snap-frozen tissue specimens are also acceptable and they must contain a minimum of 10% tumor. Please note that turn-around time for this test is up to 21 days.

* Disease Status: Patients with disseminated DIPG or HGG are eligible only if the patient is to receive chemotherapy only, i.e. no craniospinal RT is intended to be given. MRI of spine must be performed if disseminated disease is suspected clinically by the treating physicians. Patients with primary spinal tumors are eligible only if the patient is to receive either chemotherapy or focal radiation therapy, i.e. no craniospinal RT is intended to be given. Patients with leptomeningeal disease only, with no definitive identifiable primary tumor, and documented NTRK fusion, must be discussed with the Study Chair on a case-by-case basis.
* Surgical Cohort ONLY: Patients with newly-diagnosed HGG with NTRK fusions who have undergone prior biopsy and for whom further resection is indicated for a more definitive surgery at an enrolling site will be eligible to enroll onto the surgical study. DIPG patients are not eligible for the surgical cohort.
* Performance Level: Karnofsky ≥ 50% for patients \> 16 years of age and Lansky ≥ 50 for patients ≤ 16 years of age (See Appendix I). Patients who are unable to walk because of paralysis, but who are up in a wheelchair, will be considered ambulatory for the purpose of assessing the performance score.

Exclusion Criteria

* Organ Function Requirements: Adequate Bone Marrow Function Defined as:

Peripheral absolute neutrophil count (ANC) ≥ 1000/mm3 Platelet count ≥ 100,000/mm3 (transfusion independent, defined as not receiving platelet transfusions for at least 7 days prior to enrollment) Hemoglobin \>8 g/dL (may receive transfusions)

\- Adequate Renal Function Defined as: Serum creatinine within normal institutional limits, or Creatinine clearance or radioisotope GFR ≥ 70ml/min/1.73 m2

\- Adequate Liver Function Defined as: Total bilirubin ≤ 2.5 × institutional upper limit of normal AST(SGOT)/ALT(SGPT) ≤ 2.5 × institutional upper limit of normal

\- Adequate Pulmonary Function Defined as: Pulse oximetry \> 94% on room air if there is clinical indication for determination (e.g. dyspnea at rest).

\- Adequate Neurologic Function Defined as: Patients with seizure disorder may be enrolled if on anticonvulsants and well controlled. See Section 5.5.2 and Appendix III for EIAED guidelines.

\- Informed Consent: All patients and/or their parents or legally authorized representatives must sign a written informed consent. Assent, when appropriate, will be obtained according to institutional guidelines.


* Pregnancy or Breast-Feeding: Pregnant or breast-feeding women will not be entered on this study due to unknown risks of fetal and teratogenic adverse events as seen in animal/human studies. Pregnancy tests must be obtained in girls who are post-menarchal. Males or females of reproductive potential may not participate unless they have agreed to use an effective contraceptive method.
* Concomitant Medications Investigational Drugs: Patients who have previously received or are currently receiving another investigational drug are not eligible.

Anti-cancer Agents: Patients who have previously received or are currently receiving other anti-cancer agents, including chemotherapy, immunotherapy, monoclonal antibodies, biologic or targeted therapy, are not eligible

* Infection: Patients must not have any active, uncontrolled systemic bacterial, viral or fungal infection.
* Patients who have received prior solid organ transplantation are not eligible.
* Patients must not have malabsorption syndrome or other condition affecting oral absorption.
* Patients must not be receiving any treatment with a strong cytochrome P450 3A4 (CYP3A4) inhibitor or inducer. (See Appendix III.) Strong inducers or inhibitors of CYP3A4 should be avoided from 7 days prior to enrollment to the end of the study.
* Patients who in the opinion of the investigator may not be able to comply with the safety monitoring requirements of the study are not eligible.
Maximum Eligible Age

21 Years

Eligible Sex

ALL

Accepts Healthy Volunteers

No

Sponsors

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Nationwide Children's Hospital

OTHER

Sponsor Role lead

Responsible Party

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Responsibility Role SPONSOR

Principal Investigators

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Susan Chi, MD

Role: STUDY_CHAIR

Dana Farber/ Boston Children's Cancer and Blood Disorders Center

Maryam Fouladi, MD

Role: STUDY_CHAIR

Nationwide Children's Hospital

Locations

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Children's Hospital Colorado

Aurora, Colorado, United States

Site Status RECRUITING

Children's National Medical Center

Washington D.C., District of Columbia, United States

Site Status RECRUITING

Ann & Robert H. Lurie Children's Hospital of Chicago

Chicago, Illinois, United States

Site Status NOT_YET_RECRUITING

Dana-Farber Cancer Institute

Boston, Massachusetts, United States

Site Status RECRUITING

Duke University Health System

Durham, North Carolina, United States

Site Status RECRUITING

Cincinnati Children's Hospital Medical Center

Cincinnati, Ohio, United States

Site Status RECRUITING

Nationwide Children's Hospital

Columbus, Ohio, United States

Site Status RECRUITING

Children's Hospital of Philadelphia

Philadelphia, Pennsylvania, United States

Site Status RECRUITING

Texas Children's Hospital

Houston, Texas, United States

Site Status RECRUITING

Seattle Children's Hospital

Seattle, Washington, United States

Site Status RECRUITING

Sydney Children's Hospital

Randwick, New South Wales, Australia

Site Status RECRUITING

Queensland Children's Hospital

South Brisbane, Queensland, Australia

Site Status RECRUITING

Perth Children's Hospital

Perth, Western Australia, Australia

Site Status RECRUITING

The Hospital for Sick Children (SickKids)

Toronto, Ontario, Canada

Site Status RECRUITING

Montreal Children's Hospital

Montreal, Quebec, Canada

Site Status RECRUITING

Hopp Children's Cancer Center at NCT Heidelberg (KiTZ)

Heidelberg, Baden-Wurttemberg, Germany

Site Status RECRUITING

University Medical Center Augsburg

Augsburg, , Germany

Site Status RECRUITING

University Hospital Berlin

Berlin, , Germany

Site Status RECRUITING

University Hospital Koln

Cologne, , Germany

Site Status RECRUITING

University Medical Center Gottingen

Göttingen, , Germany

Site Status RECRUITING

Starship Children's Hospital

Auckland, Grafton, New Zealand

Site Status RECRUITING

Countries

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Austria Netherlands United Kingdom United States Australia Canada Germany New Zealand

Central Contacts

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Kelsey H Troyer, PhD

Role: CONTACT

Phone: 16147223284

Email: [email protected]

Facility Contacts

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Holly Lindsay, MD

Role: primary

Eugene Hwang, MD

Role: primary

Ashley Plant, MD

Role: primary

Susan Chi, MD

Role: primary

David Ashley, MBBS, PhD

Role: primary

Peter de Blank, MD

Role: primary

Maryam Fouladi, MD

Role: primary

Michael J Fisher, MD

Role: primary

Patricia Baxter, MD

Role: primary

Sarah Leary, MD

Role: primary

David Ziegler, MBBS

Role: primary

Tim Hassall, MBBS

Role: primary

Nick Gottardo, MBChB

Role: primary

Eric Bouffet, MD

Role: primary

Genevieve Legault, MD

Role: primary

Olaf Witt, MD

Role: primary

Michael Fruhwald, MD

Role: primary

Pablo Hernaiz Driever, MD

Role: primary

Matthias Fischer, MD

Role: primary

C. Kramm, MD

Role: primary

Sarah Hunter, MB ChB

Role: primary

Other Identifiers

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CONNECT1903

Identifier Type: -

Identifier Source: org_study_id