Clinical Trials in the UK / NCT05878860
Recruiting
Phase 3
ATSN-201 Gene Therapy in RS1-Associated X-linked Retinoschisis
NCT05878860 · tracked via the Priya Life Science UK tracker
Condition(s) studied
X-linked Retinoschisis
Investigational drug(s) / intervention(s)
ATSN-201
ATSN-201: AAV.SPR-hGRK1-hRS1syn
Study summary
This study will evaluate the safety and efficacy of ATSN-201 in subjects ≥ 5 years of age with RS1-associated X-linked retinoschisis (XLRS).
Eligibility
Part A and B:
Inclusion Criteria:
1. Age ≥ 18 for Cohorts 1 through 4, and age ≥ 6 years and \< 18 years for Cohort 5.
2. Male patients with clinical diagnosis of XLRS caused by mutations in RS1.
3. Best corrected visual acuity (BCVA) in study eye of 34 to 73 Early Treatment Diabetic Retinopathy Study (ETDRS) letters (corresponding to a Snellen acuity of 20/200 to 20/40).
4. Presence of foveal schisis and /or parafoveal/perifoveal schisis in the study eye on SD-OCT per the Principal Investigator.
Exclusion Criteria:
1. Pre-existing eye conditions in the study eye that would contribute significantly to an increased risk of visual loss from a subretinal injection (eg, advanced glaucoma, optic neuropathy, uveitis, corneal transplants).
2. Any intraocular surgery (including laser treatment) in the study eye within 6 months prior to Screening or any intraocular surgery anticipated in the study eye during the first 12 months of the study.
3. Treatment in a prior ocular gene or cell therapy study.
Part C:
Inclusion Criteria:
Note: For patients ineligible for bilateral dosing based on the ocular exclusion criteria, the same eye must meet all ocular inclusion criteria, but none of the ocular exclusion criteria, to be eligible for unilateral dosing.
General All of the following criteria must be met for unilateral or bilateral dosing.
1. Age ≥ 5 years.
2. Genetically male patients with clinical diagnosis of XLRS caused by pathogenic or likely pathogenic mutations in RS1 OR Genetically female patients with clinical diagnosis of XLRS caused by biallelic pathogenic or likely pathogenic mutations in RS1.
Ocular At least 1 eye must meet all of the following criteria for both unilateral and bilateral dosing.
3. BCVA of 34 to 73 ETDRS letters (corresponding to a Snellen acuity of 20/200 to 20/40).
4. Presence of foveal schisis on SD-OCT.
Exclusion Criteria:
General None of the following criteria can be met for unilateral or bilateral dosing.
1. Treatment with any carbonic anhydrase inhibitor (oral or topical) within 1 month prior to Screening.
2. Treatment in a prior ocular gene or cell therapy study.
3. Absence of macular schisis.
4. BCVA better than 75 ETDRS letters (corresponding to a Snellen acuity of 20/32).
5. Pre-existing eye conditions that would contribute significantly to an increased risk of visual loss from a subretinal injection (eg, advanced glaucoma, optic neuropathy, uveitis, corneal transplants).
6. Any intraocular surgery (including laser treatment) within 6 months prior to Screening or any intraocular surgery anticipated during the first 12 months of the study (laser treatment is allowed as long as it occurs at least 2 weeks before surgery).
Primary outcome measure(s)
- Part A (Dose Escalation) and Part B (Dose Expansion): Safety and tolerability as assessed by dose-limiting toxicities and treatment-emergent adverse events — From baseline to week 52
Incidence of dose-limiting toxicities (DLTs) and treatment-emergent adverse events (TEAEs). - Part C (Phase 3, Randomized, Controlled) Effect of ATSN-201 on visual function. — From baseline to week 52
Proportion of subjects with improvement ≥ 7dB from baseline in microperimetry across prespecified loci in the study eye.
Trial sites (21)
| Facility | City | Region | Status |
|---|---|---|---|
| Associated Retina Consultants | Phoenix | Arizona | Recruiting |
| Shiley Eye Institute | La Jolla | California | Not Yet Recruiting |
| Children's Hospital of Los Angeles | Los Angeles | California | Recruiting |
| Stanford University | Palo Alto | California | Not Yet Recruiting |
| UC San Francisco | San Francisco | California | Recruiting |
| Bascom Palmer Eye Institute | Miami | Florida | Recruiting |
| Massachusetts Eye and Ear | Boston | Massachusetts | Recruiting |
| Boston Children's Hospital | Boston | Massachusetts | Not Yet Recruiting |
| University of Michigan | Ann Arbor | Michigan | Recruiting |
| Mayo Clinic | Rochester | Minnesota | Recruiting |
| Columbia University | New York | New York | Recruiting |
| Flaum Eye Institute, University of Rochester | Rochester | New York | Not Yet Recruiting |
| Duke University | Durham | North Carolina | Recruiting |
| Oregon Health Sciences University | Portland | Oregon | Recruiting |
| Children's Hospital of Philadelphia | Philadelphia | Pennsylvania | Recruiting |
| Retina Foundation of the Southwest | Dallas | Texas | Recruiting |
| Moran Eye Center | Salt Lake City | Utah | Recruiting |
| University of Wisconsin | Madison | Wisconsin | Recruiting |
| The Hospital for Sick Children | Toronto | Ontario | Not Yet Recruiting |
| McGill University (MUHC) | Montreal | Quebec | Not Yet Recruiting |
| Oxford Eye Hospital | Oxford | England | Recruiting |
Official registry record
This page summarises publicly available registry data for informational purposes — not medical advice. Eligibility is determined by each study team; patients should discuss participation with their clinician.
View NCT05878860 on ClinicalTrials.gov ↗ ← All trials in the UK