Pilot Study on Trametinib for Surgical Unruptured AVMs

NCT ID: NCT06098872

Last Updated: 2024-05-30

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

Get a concise snapshot of the trial, including recruitment status, study phase, enrollment targets, and key timeline milestones.

Recruitment Status

RECRUITING

Clinical Phase

PHASE2

Total Enrollment

10 participants

Study Classification

INTERVENTIONAL

Study Start Date

2024-01-30

Study Completion Date

2025-11-30

Brief Summary

Review the sponsor-provided synopsis that highlights what the study is about and why it is being conducted.

Arteriovenous malformation (AVM) is a tangle of abnormal vessels that can progress through life and cause significant bleeding, deformity, pain, and deficits in day-to-day activities. Surgery is a common treatment option for patients with AVMs where the goal is to safely remove the entire AVM without causing complications. While any surgery has its potential risks, most of the potential modifiable risk factors relate to the AVM's structure, such as the AVM size or presence of high risk structural features seen on scans. The purpose of this pilot study is to see whether taking an oral medication called Trametinib can improve upon the AVM structure in adult patients before their scheduled surgery.

Detailed Description

Dive into the extended narrative that explains the scientific background, objectives, and procedures in greater depth.

The goal of this pilot clinical trial is to see whether an oral medication called Trametinib can be given to patients with arteriovenous malformations (AVMs) of the brain and body before surgery in order to make the AVM structure less risky for surgery.

The main questions it aims to answer are:

1. does taking Trametinib make the structure of the AVM less risky for surgery? This will determined by comparing the size and structure of the AVM on repeat scans before and after taking the drug.
2. does taking Trametinib reduce the blood flow to the AVM? This will be determined by quantifying the blood flow to the AVM with quantitative magnetic resonance imaging software.
3. is the drug well tolerated in this patient population? This will be determined by following for any side effects of the medication
4. how does the drug do what it is supposed to do clinically by looking at its effect at the cell level? This will be determined by taking a piece of the AVM that is removed at the time of surgery and running experiments in the lab to compare its structure and behaviour to other AVMs that were not treated with this medication.

Participants will first undergo screening tests to ensure they are candidates for the medication. They will take oral Trametinib once daily for a total of 60 days prior to their planned surgery. They will be monitored for side effects at days 15, 30 and 60. They will undergo routine scans prior to starting the drug and then again within 5 days of their last dose to see any changes made to the AVM structure after taking the drug. Lastly, at the time of surgery, a part of the AVM removed will be sent to our research lab to see what the drug is doing at the cell level to result in the changes we can see on the scans.

Conditions

See the medical conditions and disease areas that this research is targeting or investigating.

Arteriovenous Malformations

Study Design

Understand how the trial is structured, including allocation methods, masking strategies, primary purpose, and other design elements.

Allocation Method

NA

Intervention Model

SINGLE_GROUP

This is an open-label pilot study assessing the potential efficacy of Trametinib in improving AVM angioarchitecture for patients who are already planned to undergo surgical resection as part of their standard of care.
Primary Study Purpose

TREATMENT

Blinding Strategy

NONE

Study Groups

Review each arm or cohort in the study, along with the interventions and objectives associated with them.

Experimental: Oral Trametinib

Participants will receive oral Trametinib once daily for up to 60 days prior to their elective surgery

Group Type EXPERIMENTAL

Trametinib tablet

Intervention Type DRUG

Drug is supplied in 2mg and 0.5 mg tablets

Interventions

Learn about the drugs, procedures, or behavioral strategies being tested and how they are applied within this trial.

Trametinib tablet

Drug is supplied in 2mg and 0.5 mg tablets

Intervention Type DRUG

Eligibility Criteria

Check the participation requirements, including inclusion and exclusion rules, age limits, and whether healthy volunteers are accepted.

Inclusion Criteria

1. Age ≥18 years.
2. Confirmed diagnosis of an unruptured AVM Spetzler-Martin Lawton Young Grade equal to or less than 6 on magnetic resonance imaging (MRI), CT-angiogram (CTA) or angiogram, and clinical exam by a physician who is familiar with this condition at any time in patient's medical history.
3. Planned surgical resection of AVM at University Health Network within the acceptable window defined by the study calendar (i.e. after the indicated study drug dosing period and approximate week-long follow up).
4. Patients must not have received an investigational drug within the 4 weeks prior to study enrolment.
5. Patients who have previously received biologic therapy treatment must have completed therapy at least 14 days prior to study enrolment.
6. Patients who have previously received myelosuppressive chemotherapy must have completed therapy at least 28 days prior to study enrolment.
7. Patients on anticoagulants must have stopped treatment within 7 days of starting Trametinib.
8. Patient is able to swallow oral medication and/or retain oral medication via G tube.
9. Patients of childbearing potential (as assessed by their local Investigator) and fertile men who are sexually active must agree to the use of 2 forms of contraception (as discussed with the overseeing physician) throughout the period of study treatment and for 16 weeks after last dose of study drug. They are not allowed to donate ova or sperm for up to 16 weeks after the last dose of study drug.

Exclusion Criteria

1. AVM due to known germline mutation such as phosphatase and tensin homolog (PTEN) or known history of familial AVM syndromes.
2. Received prior map kinase (MEK) inhibitor therapy.
3. Known allergy or contraindication to MEK inhibitor treatment.
4. Patients who have undergone major surgery, as defined by the overseeing Investigator, within 28 days prior to study enrolment or who have not recovered from side effects of such a procedure.
5. Patients that are currently pregnant or breastfeeding.
6. A known history of coagulopathy and/or current use of anticoagulant therapy.
7. International normalized ratio (INR) \> 1.5 within 7 days of enrolment.
8. Left ventricular ejection fraction (LVEF) \<50%, or any ECG abnormalities within 7 days of enrolment.
9. Retinal vein occlusion, serous retinopathy or glaucoma diagnosed within 1 month of enrolment.
10. Diagnosis of significant liver failure (Child-Pugh score 2+) within 7 days of enrolment.
11. Rhabdomyolysis (creatinine kinase (CK) \>5x ULN) within 7 days of enrolment.
12. Patients with known risk factors for gastrointestinal perforation (prior perforation, diverticulitis, metastases to the gastrointestinal tract and concomitant use of medications with a recognized risk of gastrointestinal perforation
13. Positive covid-19 polymerase chain reaction (PCR) test within 7 days of enrolment.
14. Patient is unwilling or unable to comply with study requirements.
15. Unstable health status that may interfere with completing the study, as assessed by the overseeing Investigator.
Minimum Eligible Age

18 Years

Eligible Sex

ALL

Accepts Healthy Volunteers

No

Sponsors

Meet the organizations funding or collaborating on the study and learn about their roles.

University Health Network, Toronto

OTHER

Sponsor Role lead

Responsible Party

Identify the individual or organization who holds primary responsibility for the study information submitted to regulators.

Responsibility Role SPONSOR

Principal Investigators

Learn about the lead researchers overseeing the trial and their institutional affiliations.

Ivan Radovanovic, MD PhD

Role: PRINCIPAL_INVESTIGATOR

University Health Network, Toronto

Locations

Explore where the study is taking place and check the recruitment status at each participating site.

University Health Network

Toronto, Ontario, Canada

Site Status RECRUITING

Countries

Review the countries where the study has at least one active or historical site.

Canada

Central Contacts

Reach out to these primary contacts for questions about participation or study logistics.

Ivan Radovanovic, MD PhD

Role: CONTACT

416-603-6207

Ann Mansur, MD

Role: CONTACT

416-603-5800 ext. 7371

Facility Contacts

Find local site contact details for specific facilities participating in the trial.

Ivan Radovanovic, MD PhD

Role: primary

Ann Mansur, MD

Role: backup

References

Explore related publications, articles, or registry entries linked to this study.

Mansur A, Radovanovic I. Vascular malformations: An overview of their molecular pathways, detection of mutational profiles and subsequent targets for drug therapy. Front Neurol. 2023 Feb 10;14:1099328. doi: 10.3389/fneur.2023.1099328. eCollection 2023.

Reference Type BACKGROUND
PMID: 36846125 (View on PubMed)

Nikolaev SI, Fish JE, Radovanovic I. Somatic Activating KRAS Mutations in Arteriovenous Malformations of the Brain. N Engl J Med. 2018 Apr 19;378(16):1561-1562. doi: 10.1056/NEJMc1802190. No abstract available.

Reference Type BACKGROUND
PMID: 29669234 (View on PubMed)

Fish JE, Flores Suarez CP, Boudreau E, Herman AM, Gutierrez MC, Gustafson D, DiStefano PV, Cui M, Chen Z, De Ruiz KB, Schexnayder TS, Ward CS, Radovanovic I, Wythe JD. Somatic Gain of KRAS Function in the Endothelium Is Sufficient to Cause Vascular Malformations That Require MEK but Not PI3K Signaling. Circ Res. 2020 Aug 28;127(6):727-743. doi: 10.1161/CIRCRESAHA.119.316500. Epub 2020 Jun 17.

Reference Type BACKGROUND
PMID: 32552404 (View on PubMed)

Hong T, Yan Y, Li J, Radovanovic I, Ma X, Shao YW, Yu J, Ma Y, Zhang P, Ling F, Huang S, Zhang H, Wang Y. High prevalence of KRAS/BRAF somatic mutations in brain and spinal cord arteriovenous malformations. Brain. 2019 Jan 1;142(1):23-34. doi: 10.1093/brain/awy307.

Reference Type BACKGROUND
PMID: 30544177 (View on PubMed)

Nicholson CL, Flanagan S, Murati M, Boull C, McGough E, Ameduri R, Weigel B, Maguiness S. Successful management of an arteriovenous malformation with trametinib in a patient with capillary-malformation arteriovenous malformation syndrome and cardiac compromise. Pediatr Dermatol. 2022 Mar;39(2):316-319. doi: 10.1111/pde.14912. Epub 2022 Jan 10.

Reference Type BACKGROUND
PMID: 35014097 (View on PubMed)

Lekwuttikarn R, Lim YH, Admani S, Choate KA, Teng JMC. Genotype-Guided Medical Treatment of an Arteriovenous Malformation in a Child. JAMA Dermatol. 2019 Feb 1;155(2):256-257. doi: 10.1001/jamadermatol.2018.4653.

Reference Type BACKGROUND
PMID: 30566190 (View on PubMed)

Edwards EA, Phelps AS, Cooke D, Frieden IJ, Zapala MA, Fullerton HJ, Shimano KA. Monitoring Arteriovenous Malformation Response to Genotype-Targeted Therapy. Pediatrics. 2020 Sep;146(3):e20193206. doi: 10.1542/peds.2019-3206.

Reference Type BACKGROUND
PMID: 32859736 (View on PubMed)

Wright CJ, McCormack PL. Trametinib: first global approval. Drugs. 2013 Jul;73(11):1245-54. doi: 10.1007/s40265-013-0096-1.

Reference Type BACKGROUND
PMID: 23846731 (View on PubMed)

Couto JA, Huang AY, Konczyk DJ, Goss JA, Fishman SJ, Mulliken JB, Warman ML, Greene AK. Somatic MAP2K1 Mutations Are Associated with Extracranial Arteriovenous Malformation. Am J Hum Genet. 2017 Mar 2;100(3):546-554. doi: 10.1016/j.ajhg.2017.01.018. Epub 2017 Feb 9.

Reference Type BACKGROUND
PMID: 28190454 (View on PubMed)

Other Identifiers

Review additional registry numbers or institutional identifiers associated with this trial.

OZUHN-017

Identifier Type: OTHER

Identifier Source: secondary_id

23-5463

Identifier Type: -

Identifier Source: org_study_id

More Related Trials

Additional clinical trials that may be relevant based on similarity analysis.