Longitudinal Prospective Natural History Study of Retinopathy in Zellweger Spectrum Disorder

NCT ID: NCT06190626

Last Updated: 2026-01-21

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

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Recruitment Status

RECRUITING

Total Enrollment

30 participants

Study Classification

OBSERVATIONAL

Study Start Date

2023-12-18

Study Completion Date

2029-01-01

Brief Summary

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The goal of this observational study is to define the course of the retinal degeneration in a ZSD patient cohort.

The objective of this study is to gather information so the investigators can:

1. define the course of the retinal degeneration in a ZSD patient cohort with retinal degeneration
2. define what tests best monitor the progression of the retinal degeneration
3. generate prognostic information about vision loss in ZSD.

At each yearly visit, the participants will answer a functional vision questionnaire, have a physical evaluation, blood test, and participate in a variety of vision tests. The investigators will also collect pertinent medical history.

Participants will travel to study site. The study will provide financial support for board and travel.

Detailed Description

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Participants/guardians will have a 1-hour virtual interview before the first visit to review the tests and determine which tests the participant is able to perform.

Below is a list of the vision tests used for this study:

Prior to each visit, the participant will complete the PedEyeQ or VFQ-25 questionnaire about functional vision.

At each clinic visit :

* Refraction and glasses test: this is a general vision test, as commonly conducted by an optometrist, before and after pupil dilation
* Best corrected visual acuity: the participant's vision will be tested using cards or a chart.
* Contrast sensitivity: the participant's ability to detect contrast will be tested using a chart.
* Visual field: the participant's field of vision will be tested by asking them to indicate when they see a stimulus.
* Reading test: the participant's ability to see written text will be tested using the International Reading Speed Test
* Color vision test: the participant's ability to see color will be tested using cards
* Eye exam: the participant's eyes will be examined with a lamp
* Pupil dilation: to allow the completion of some tests, the participant's pupils will be dilated using medicated eye drops
* Microperimetry: the participant's macula (central retina) will be visualized.
* Fundus photography: the participant's fundus (central retina) will be photographed in light and dark conditions
* Full field stimulus threshold test: the participant's ability to perceive light in different parts of their visual field will be tested
* Optical coherence tomography (OCT): detailed images of the participant's retina will be taken
* Fundus autofluorescence: the participant's retinal health will be assessed by imaging the natural fluorescent properties of their retinal pigment epithelium
* Chromatic pupillometry: the function of different cells in the participant's retina will be determined by measuring their response to light
* Multi-luminance mobility test (MLMT): the participant will walk through an obstacle course at varying light levels to test their functional vision in different light conditions.
* Blood collection: a blood sample will be collected and banked to measure peroxisome functions.

Conditions

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Zellweger Spectrum Disorder

Study Design

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Observational Model Type

COHORT

Study Time Perspective

PROSPECTIVE

Eligibility Criteria

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Inclusion Criteria

* confirmed ZSD with deleterious variants in PEX genes identified
* confirmed or expected retinal involvement

Exclusion Criteria

* unable to perform a minimum of one vision test
* severe ZSD disease
Eligible Sex

ALL

Accepts Healthy Volunteers

No

Sponsors

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Canadian Institutes of Health Research (CIHR)

OTHER_GOV

Sponsor Role collaborator

McGill University Health Centre/Research Institute of the McGill University Health Centre

OTHER

Sponsor Role lead

Responsible Party

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Nancy Braverman

Professor

Responsibility Role PRINCIPAL_INVESTIGATOR

Principal Investigators

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Nancy E Braverman, MD, MS

Role: PRINCIPAL_INVESTIGATOR

McGill University Health Centre/Research Institute of the McGill University Health Centre

Locations

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University of Rochester Medical Center/Flaum Eye Institute

Rochester, New York, United States

Site Status RECRUITING

Eye Institute of Alberta

Edmonton, Alberta, Canada

Site Status RECRUITING

Research Institute-McGill University Health Center

Montreal, Quebec, Canada

Site Status RECRUITING

Countries

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United States Canada

Central Contacts

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Nancy E Braverman, MD, MS

Role: CONTACT

514-934-1934 ext. 23404

Frederique Arnaud, BScN

Role: CONTACT

514-934-1934 ext. 23404

Facility Contacts

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Alex Levin, MD

Role: primary

Constantin Mouzaaber, MSc

Role: primary

587-590-5041

Frederique Arnaud, BScN

Role: primary

References

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Braverman NE, Raymond GV, Rizzo WB, Moser AB, Wilkinson ME, Stone EM, Steinberg SJ, Wangler MF, Rush ET, Hacia JG, Bose M. Peroxisome biogenesis disorders in the Zellweger spectrum: An overview of current diagnosis, clinical manifestations, and treatment guidelines. Mol Genet Metab. 2016 Mar;117(3):313-21. doi: 10.1016/j.ymgme.2015.12.009. Epub 2015 Dec 23.

Reference Type BACKGROUND
PMID: 26750748 (View on PubMed)

Yergeau C, Coussa RG, Antaki F, Argyriou C, Koenekoop RK, Braverman NE. Zellweger Spectrum Disorder: Ophthalmic Findings from a New Natural History Study Cohort and Scoping Literature Review. Ophthalmology. 2023 Dec;130(12):1313-1326. doi: 10.1016/j.ophtha.2023.07.026. Epub 2023 Aug 2.

Reference Type BACKGROUND
PMID: 37541626 (View on PubMed)

Other Identifiers

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2024-9679

Identifier Type: -

Identifier Source: org_study_id

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