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Study of Subretinally Injected ATSN-101 Administered in Patients With Leber Congenital Amaurosis Caused by Biallelic Mutations in GUCY2D

Phase 1
Active, not recruiting
Conditions
LCA
LCA1
Leber Congenital Amaurosis
Interventions
Drug: Triamcinalone Acetonide
Drug: Trimethoprim/polymyxin B
Registration Number
NCT03920007
Lead Sponsor
Atsena Therapeutics Inc.
Brief Summary

Primary Objective:

To evaluate the safety and tolerability of ascending doses of ATSN-101 administered as a unilateral subretinal injection in patients with Leber Congenital Amaurosis (LCA) caused by autosomal recessive guanylate cyclase 2D (GUCY2D) mutations (GUCY2D-LCA).

Secondary Objective:

To evaluate the efficacy of ascending doses of ATSN-101 administered as a unilateral subretinal injection in patients with GUCY2D-LCA.

Detailed Description

Study duration per participant is approximately 112 weeks including: an approximately 56-day screening/baseline period, an approximately 52-week study observation period including 1 treatment day, and an approximately 52-week safety follow-up period. The end of study visit will be approximately 260 weeks after the Investigational Medicinal Product (IMP) administration.

After completion of the main study (ATSN-101-1), participants may have the option to enroll in a separate long-term follow-up study, in which case they would no longer continue in ATSN-101-1 and their end of study visit would be conducted at Week 52.

The study is separated into 2 parts including a dose escalation phase (Part A) and a dose expansion phase (Part B). In Part B participants will be treated at the maximum tolerated dose (MTD) or maximum administered dose (MAD) determined from Part A.

Recruitment & Eligibility

Status
ACTIVE_NOT_RECRUITING
Sex
All
Target Recruitment
15
Inclusion Criteria

Not provided

Exclusion Criteria

Not provided

Study & Design

Study Type
INTERVENTIONAL
Study Design
SINGLE_GROUP
Arm && Interventions
GroupInterventionDescription
ATSN-101ATSN-101 Diluent SolutionATSN-101 single dose according to an ascending dose design (dose escalation phase) or ATSN-101 single dose (dose expansion phase)
ATSN-1011% PrednisoloneATSN-101 single dose according to an ascending dose design (dose escalation phase) or ATSN-101 single dose (dose expansion phase)
ATSN-101Triamcinalone AcetonideATSN-101 single dose according to an ascending dose design (dose escalation phase) or ATSN-101 single dose (dose expansion phase)
ATSN-101Trimethoprim/polymyxin BATSN-101 single dose according to an ascending dose design (dose escalation phase) or ATSN-101 single dose (dose expansion phase)
ATSN-101PrednisoneATSN-101 single dose according to an ascending dose design (dose escalation phase) or ATSN-101 single dose (dose expansion phase)
ATSN-101ATSN-101ATSN-101 single dose according to an ascending dose design (dose escalation phase) or ATSN-101 single dose (dose expansion phase)
Primary Outcome Measures
NameTimeMethod
Number of participants with adverse events (AEs) from baseline up to the end of the observation periodFrom baseline to week 52

Number of participants with AEs will be summarized in each cohort and overall

Number of participants with AEs from baseline up to the end of the safety follow-up periodFrom baseline to week 260

Number of participants with AEs will be summarized in each cohort and overall

Secondary Outcome Measures
NameTimeMethod
Change in best -corrected visual acuity (BCVA)Baseline to week 52 and Baseline to week 260

Change in BCVA from baseline in the treated and untreated eye (control)

Change in sensitivityBaseline to week 52 and Baseline to week 260

Change in sensitivity from baseline in the treated eye and untreated eye (control) as measured by the full-field stimulus testing

Trial Locations

Locations (2)

Casey Eye Institute - Oregon Health & Science University

🇺🇸

Portland, Oregon, United States

Scheie Eye Institute, University of Pennsylvania

🇺🇸

Philadelphia, Pennsylvania, United States

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