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A Study of PTR-01 in Recessive Dystrophic Epidermolysis Bullosa

Phase 2
Completed
Conditions
Recessive Dystrophic Epidermolysis Bullosa
Interventions
Registration Number
NCT04599881
Lead Sponsor
Phoenix Tissue Repair, Inc.
Brief Summary

Protocol PTR-01-002 is a 3-part Phase 2, open-label study of PTR-01. While new patients will be enrolled, priority will be given to patients that satisfactorily completed study PTR-01-001.

Detailed Description

Protocol PTR-01-002 is a 3-part Phase 2, open-label study of PTR-01. While new patients will be enrolled, priority will be given to patients that satisfactorily completed study PTR-01-001.

In Part 1, patients will receive a dose of 3.0 mg/kg every week for a total of 4 doses. This will be followed by Part 2 in which patients will receive a dose of 3.0 mg/kg every other week for a total of 7 doses. In Part 3, patients will be followed for 12 weeks. No investigational therapy will be administered during this time. At the end of each dosing period, an efficacy assessment will be performed. Safety will be assessed continuously throughout the study.

Following the end of Part 3, patients may be eligible for a potential long-term extension to further refine the dosing regimen, depending upon study drug availability.

Recruitment & Eligibility

Status
COMPLETED
Sex
All
Target Recruitment
6
Inclusion Criteria

Patients must meet all of the following criteria to be eligible for study participation in the three month run in period of the study:

  1. Willing to provide informed consent form, or if 12 to <18 years of age, legal guardian has provided informed consent form and the minor has signed an assent form acknowledging that they understand and agree to study procedures.

  2. Has a diagnosis of RDEB based on genetic analysis and consistent with a recessive inheritance pattern.

  3. Has deficient C7 staining at the dermal-epidermal junction (DEJ) by IF.

  4. Agrees to use contraception as follows:

    For women of childbearing potential (WOCBP) agrees to use highly effective contraceptive (including abstinence) methods from Screening, through the study, and for at least 10 weeks after the last dose of study drug. Non-childbearing potential is defined as a female who meets either of the following criteria: age ≥50 years and no menses for at least 1 year or documented hysterectomy, bilateral tubal ligation, or bilateral oophorectomy.

    For males, agrees to use a condom with any WOCBP sexual partner from Day 1 of study treatment, through the study, and at least 10 weeks after the last dose of study drug.

  5. Be willing and able to comply with this protocol.

Exclusion Criteria

Patients with any of the following will be excluded from participation in the study:

  1. Has known systemic hypersensitivity to any of the inactive ingredients in PTR-01.
  2. Has previously had an anaphylactic reaction to PTR-01.
  3. Is pregnant or nursing.
  4. Has received in the last six months any investigational gene therapy product or in the last three months any non-gene therapy investigational products.
  5. Is anticipated to receive new regimens of antibiotics or other anti-infectives during the trial.
  6. Has any other medical or personal condition that, in the opinion of the Investigator, may potentially compromise the safety or compliance of the patient, or may preclude the patient's

Study & Design

Study Type
INTERVENTIONAL
Study Design
SINGLE_GROUP
Arm && Interventions
GroupInterventionDescription
PTR-01 3 mg/kgPTR-01All patients will receive a PTR-01 dose of 3.0 mg/kg once weekly every week for a total of 4 doses, followed by a dose of 3.0 mg/kg every other week for a total of 7 doses.
Primary Outcome Measures
NameTimeMethod
Wound healingUp to 162 days

Change in a majority of target lesions of at least 2 levels using a 7-point (1-7) Global Impression of Change instrument (7 being the worst)

Incidence of treatment-emergent adverse eventsUp to 162 days

Safety and tolerability, as assessed by treatment-emergent adverse events

Incidence of anti-drug antibodies (ADA)Up to 162 days

Safety and tolerability, as assessed by immunogenicity through anti-drug antibody (ADA) testing

Incidence of infusion-associated reactionsUp to 162 days

Safety and tolerability, as assessed by infusion-associated reactions (IAR)

Secondary Outcome Measures
NameTimeMethod
Formation of anchoring fibrilsUp to 162 days

Formation of new anchoring fibrils as measured by electron microscopy

Change in wound surface area, as assessed by Investigator Global Impression of Change (IGIC)Up to 162 days

Wound area of target lesions, as assessed by IGIC

Change in total body wound surface areaUp to 162 days

Change in total body wound surface area, using Rule of Nines

Change in skin integrity, as assessed by suction blister timeUp to 162 days

Change in skin integrity, as assessed by suction blister time

Change in itch severity, as assessed by modified Patient-Reported Outcome Measurement Information System (PROMIS) itch domainsUp to 162 days

Severity of itch, as assessed by modified Patient-Reported Outcome Measurement Information System (PROMIS) itch domains

Change in itch severity, as assessed by the Instrument for Scoring Clinical Outcomes for Research of Epidermolysis Bullosa (iscorEB)Up to 162 days

Severity of itch, as assessed by Instrument for Scoring Clinical Outcomes for Research of Epidermolysis Bullosa (iscorEB), maximum score of 234 (worst)

Change in the impact of itch on quality of lifeUp to 162 days

Change in the impact of itch on quality of life, as assessed by the Pruritus-Specific Quality of Life Instrument (ItchyQoL), maximum score of 110 (worst)

Change in pain severity, as assessed by the Instrument for Scoring Clinical Outcomes for Research of Epidermolysis Bullosa (iscorEB)Up to 162 days

Change in pain severity, as assessed by the Instrument for Scoring Clinical, maximum score of 234 (worst)

Change in the impact of pain on quality of lifeUp to 162 days

Change in the impact of pain on quality of life, as assessed by the Instrument for Scoring Clinical Outcomes for Research of Epidermolysis Bullosa (iscorEB) instrument, maximum score of 234 (worst)

Change in corneal symptomsUp to 162 days

Change of corneal symptoms (eye symptoms), as assessed by the Epidermolysis Bullosa Eye Disease Index (EB-EDI)

Rate of change in nutritional markers (total protein/albumin)Up to 162 days

Change of nutritional markers, as assessed by total protein/albumin

Rate of change in nutritional markers (C-reactive protein)Up to 162 days

Change of nutritional markers, as assessed by C-reactive protein

Change in skin integrity, as assessed by time to re-blisteringUp to 162 days

Change in skin integrity, as assessed by time to re-blistering

Change of dysphagia, as assessed using the Brief Esophageal Dysphagia QuestionnaireUp to 162 days

Change of dysphagia, as assessed using the Brief Esophageal Dysphagia Questionnaire, maximum score is 40 (worst)

Stabilization of corneal symptomsUp to 162 days

Stabilization of corneal symptoms (eye symptoms), as assessed by the Epidermolysis Bullosa Eye Disease Index (EB-EDI)

Rate of stabilization of nutritional markers (hemoglobin/hematocrit)Up to 162 days

Stabilization of nutritional markers, as assessed by hemoglobin/hematocrit

Rate of stabilization of nutritional markers (iron/TIBC)Up to 162 days

Stabilization of nutritional markers, as assessed by iron/TIBC

Change in time for wound careUp to 162 daysUp to 162 days

Change in time for wound care, as assessed by patient interviews

Delivery of PTR-01 to skinUp to 162 days

PTR-01 incorporation by immunofluorescence using NC1 \& NC2 staining, by dose frequency period

Change in wound surface area, as assessed by wound imagingUp to 162 days

Wound area of target lesions, as assessed by wound imaging

Stabilization of dysphagia, as assessed using the Brief Esophageal Dysphagia ScaleUp to 162 days

Stabilization of dysphagia, as assessed using the Brief Esophageal Dysphagia Scale

Rate of change in nutritional markers (iron/TIBC)Up to 162 days

Change of nutritional markers, as assessed by iron/TIBC

Rate of stabilization of nutritional markers (C-reactive protein)Up to 162 days

Stabilization of nutritional markers, as assessed by C-reactive protein

Change in Investigator Global Impressions of Change (IGIC)Up to 162 days

Global impressions of change, as assessed through IGIC (1-7), 7 being worst

Change in Investigator Patient Impressions of Change (PGIC)Up to 162 days

Global impressions of change, as assessed through PGIC (1-7), 7 being worst

Change in mental healthUp to 162 days

Change in mental health and social functioning, as assessed by the Patient-Reported Outcomes Measurement Information System (PROMIS) mental health domains

Change in amount of wound careUp to 162 days

Change in amount of wound care, as assessed by patient interviews

Change in pain severity, as assessed by modified Patient-Reported Outcome Measurement Information System (PROMIS) pain domainsUp to 162 days

Change in pain severity, as assessed by Patient-Reported Outcome Measurement Information System (PROMIS) pain domains

Change in overall quality of life, as assessed by the Quality of Life in Epidermolysis Bullosa (QOLEB) questionnaireUp to 162 days

Change in overall quality of life, as assessed by the Quality of Life in Epidermolysis Bullosa (QOLEB) questionnaire

Change in overall healthUp to 162 days

Change in overall disability, as assessed by the Health Assessment Questionnaire or Children's Health Assessment Questionnaire (HAQ/CHAQ)

Change in social functionUp to 162 days

Change in mental health and social functioning, as assessed by the Patient-Reported Outcomes Measurement Information System (PROMIS) social function domains

Change in cost of wound careUp to 162 days

Change in cost of wound care, as assessed by patient interviews

Change in overall patient impression of quality of lifeUp to 162 days

Change in overall quality of life, as assessed by patient interviews

Change in overall patient impression of disabilityUp to 162 days

Change in overall disability, as assessed by patient interviews

Change in dysphagia, as assessed by volume of oral nutritional intakeUp to 162 days

Change of dysphagia, as assessed by volume of oral nutritional intake, using patient interview and diary, maximum score is 40 (worst)

Stabilization of dysphagia, as assessed by volume oral nutritional intakeUp to 162 days

Stabilization of dysphagia, as assessed by volume oral nutritional intake, using patient interview and diary

Rate of change in nutritional markers (hemoglobin/hematocrit)Up to 162 days

Change of nutritional markers, as assessed by hemoglobin/hematocrit

Rate of stabilization of nutritional markers (total protein/albumin)Up to 162 days

Stabilization of nutritional markers, as assessed by total protein/albumin

Change in disease activity and scarringUp to 162 days

Change in disease activity and scarring, as assessed by the Epidermolysis Bullosa Disease Activity and Scarring Index (EBDASI)

Trial Locations

Locations (2)

Stanford University

🇺🇸

Redwood City, California, United States

Children's Hospital Colorado

🇺🇸

Aurora, Colorado, United States

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