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Bevacizumab In Hereditary Hemorrhagic Telangiectasia

Phase 2
Recruiting
Conditions
Hereditary Hemorrhagic Telangiectasia
Interventions
Registration Number
NCT04404881
Lead Sponsor
Hanny Al-Samkari, MD
Brief Summary

This research study is studying to see whether bevacizumab may treat chronic bleeding and iron deficiency anemia in Hereditary Hemorrhagic Telangiectasia (HHT).

Hereditary Hemorrhagic Telangiectasia (HHT) is a disorder that causes abnormal blood vessel formation. In HHT, there is a mutation in the TGF-β pathway, which results in an increase of vascular endothelial growth factor (VEGF) levels. An increase in VEGF levels can result in poorly formed blood vessels that have a higher rate of bleeding than normal blood vessels. Bevacizumab is designed to block VEGF activity. It is believed that targeting increased VEGF levels may be able to treat HHT.

This research study involves the following study drug:

- Bevacizumab

Detailed Description

In this research study, the investigators are studying the study drug, bevacizumab. Researchers would like to see whether bevacizumab effectively treats Hereditary Hemorrhagic Telangiectasia (HHT) and what side effects occur.

- The research study procedures include: screening for eligibility, pretreatment period, study treatment, end-of-study visit, and follow-up visit.

* Participants will be observed for 12 weeks and then receive study treatment for 24 weeks and will be followed for 30 days after ending study treatment.

* This research study involves the following study drug: Bevacizumab

* It is expected that about 33 people will take part in this research study.

This research study is a Phase II clinical trial. Phase II clinical trials test the safety and effectiveness of an investigational drug to learn whether the drug works in treating Hereditary Hemorrhagic Telangiectasia (HHT). "Investigational" means that the drug is being studied.

The U.S. Food and Drug Administration (FDA) has not approved bevacizumab for Hereditary Hemorrhagic Telangiectasia (HHT), but it has been approved for other uses.

Recruitment & Eligibility

Status
RECRUITING
Sex
All
Target Recruitment
33
Inclusion Criteria

Not provided

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Exclusion Criteria

Not provided

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Study & Design

Study Type
INTERVENTIONAL
Study Design
SINGLE_GROUP
Arm && Interventions
GroupInterventionDescription
BevacizumabBevacizumab* The research study procedures include: screening for eligibility, pretreatment period, study treatment, end-of-study visit, and follow-up visit. Each period consists of 12 weeks, for a total of 36 weeks. * Pretreatment Period:Hematologic Support: iron transfusions and red cell transfusions as determined by study doctor. * Induction Period (first 3 months of bevacizumab treatment): * Hematologic Support: iron transfusions and red cell transfusions as determined by study doctor. * Bevacizumab: once every 2 weeks via intravenous infusion for up to 12 weeks. * Maintenance Period (second 3 months of bevacizumab treatment): * Hematologic Support: Iron transfusions and red cell transfusions as determined by study doctor. * Bevacizumab: once every 4 weeks via intravenous infusion for up to 12 weeks.
Primary Outcome Measures
NameTimeMethod
Change in hematologic support score from pretreatment to maintenance36 Weeks

The change in Hematologic Support Score (HSS) from pretreatment to maintenance will be evaluated with a paired t-test or a Wilcoxon signed-rank test, whichever is most appropriate for the distribution of the data. Presence of a statistically-significant difference (P\<0.05) will determine if the study achieves its primary outcome measure.

Secondary Outcome Measures
NameTimeMethod
Average Maintenance Epistaxis Severity Score36 Weeks

The epistaxis severity score (ESS, not to be confused with the hematologic support score or HSS) is a well-validated, longitudinal, 6-question, 10- point score used specifically to evaluate epistaxis severity in HHT. Individual patient average pretreatment ESS will be compared to individual patient average maintenance ESS with a paired means comparison test, either a paired t-test or a Wilcoxon signed-rank test, whichever is most appropriate for the distribution of the data.

Difference in the individual patient mean hemoglobin36 Weeks

Hemoglobin concentration (g/dL) is the primary clinical measure of red cell mass and blood oxygen-carrying capacity. Hemoglobin thresholds will dictate transfusion on study according to the HSP. Following study completion, for each participant, hemoglobin measurements drawn on day 1 and weeks 2, 4, 6, 8, 10, and 12 will be averaged together to form an average pretreatment hemoglobin and hemoglobin measurements drawn on weeks 24, 26, 28, 30, 32, 34, and 36 will be averaged together to form an average maintenance hemoglobin. Individual patient average pretreatment hemoglobin will be compared to individual patient average maintenance hemoglobin with a paired means comparison test, either a paired ttest or a Wilcoxon signed-rank test, whichever is most appropriate for the distribution of the data.

Difference in the individual patient pRBC transfusion requirement36 Weeks

Number of red cell units transfused will be analyzed separately from iron infusions as a secondary endpoint. The change in number of units of red cells transfused from pretreatment to maintenance will be evaluated with a paired t-test or a Wilcoxon signed-rank test, whichever is most appropriate for the distribution of the data

Difference in the individual patient intravenous iron infusion requirement36 Weeks

Total milligrams of elemental iron infused will be analyzed separately from red cell transfusions as a secondary endpoint. The change in total milligrams of elemental iron infused from pretreatment to maintenance will be evaluated with a paired t-test or a Wilcoxon signed-rank test, whichever is most appropriate for the distribution of the data

Number of Participants With Treatment-Related Adverse Events as Assessed by CTCAE v5.0.All patients will be evaluable for toxicity from the time of their first treatment with bevacizumab up to 36 weeks

Number of subjects experiencing grade 3 or higher adverse events (as defined by CTCAE v. 50) and adverse events requiring bevacizumab discontinuation

Trial Locations

Locations (1)

Massachusetts General Hospital

🇺🇸

Boston, Massachusetts, United States

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