MedPath

Global Patient Registry of Inherited Retinal Diseases

Recruiting
Conditions
Inherited Retinal Diseases
Registration Number
NCT05957276
Lead Sponsor
Janssen Research & Development, LLC
Brief Summary

The purpose of this study is to better understand the natural history of Inherited Retinal Disease (IRD) and help inform patient management.

Detailed Description

Not available

Recruitment & Eligibility

Status
RECRUITING
Sex
All
Target Recruitment
7000
Inclusion Criteria

For Participant Selection:

  • Participant has any clinically documented sign(s) and/or symptom(s) consistent with an Inherited Retinal Disease (IRD), or asymptomatic with documented retinal changes detected by imaging or electrophysiology
  • Participant has documented genetic variant(s) (known pathogenic, likely pathogenic, or variants of uncertain significance) in relevant genes for any of the following IRDs: X-Linked Retinitis Pigmentosa (XLRP) and/or Achromatopsia (ACHM)
  • Participant or legally acceptable representative has provided informed consent (and participant assent, when applicable) in accordance with local requirements
  • Participant is able to have relevant visual and/or retinal assessments performed

For Caregiver Selection:

  • Caregiver has consent from the associated participant to participate in the study, or participant assent and consent from their legally acceptable representative
  • Male or female aged greater than or equal to (>=)18 years
  • Identified by an enrolled participant (or their legally acceptable representative*) as a primary caregiver
  • Caregiver has provided informed consent in accordance with local requirements
Exclusion Criteria

For Participant Selection:

  • Participant has received a treatment in an IRD-related interventional trial, or is being screened for an IRD-related interventional trial

For Caregiver Selection:

  • Caregiver has an IRD diagnosis and presents with symptoms (visual impairment)

Study & Design

Study Type
OBSERVATIONAL
Study Design
Not specified
Primary Outcome Measures
NameTimeMethod
Visual Acuity (VA)Baseline up to 8 years

VA is a measure of the sharpness of vision. The test uses a chart with letters or symbols of different sizes, at a specific distance, and is reported using various scales, such as fraction, decimal, minimum angle of resolution (MAR), logMAR. When a participant is unable to read a chart, visual acuity can be measured by counting fingers, hand motion, or light perception.

Visual Field (VF)Baseline up to 8 years

VF is used to determine scope of vision, including central and peripheral vision. It can determine place, size, and shape of scotoma in vision.

Secondary Outcome Measures
NameTimeMethod
Association Between IRD Genotype and Visual FieldBaseline up to 8 years

Association between IRD genotype and visual field will be reported as incidence of visual field for given IRD genotype.

Hospital Anxiety and Depression Scale (HADS)Baseline up to 8 years

The HADS is a 14-item questionnaire to assess the presence of anxiety and depression in individuals aged 16-65 years, using 4-point Likert scales. Summary scores are reported for the 2 domains. Each domain has a score range of 0-21, with higher scores reflecting increased anxiety or depression.

Association Between IRD Genotype and Change in Visual AcuityBaseline up to 8 years

Association between IRD genotype and change in visual acuity will be reported as change in visual acuity for given IRD genotype.

Association Between IRD Genotype and Change in Visual FieldBaseline up to 8 years

Association between IRD genotype and change in visual field will be reported as change in visual field for given IRD genotype.

IRD Variants and SubtypesBaseline up to 8 years

Number and distribution of IRD variants and subtypes will be described.

Number of Participants With Various Signs and SymptomsBaseline

Number of participants with various signs and symptoms (for example: amblyopia, blindness, corneal disease/dystrophy) will be reported.

Clinical Global Impression of Change (CGIC)First post-baseline visit up to 8 years

CGIC score will be reported for XLRP participants. CGIC is a global, generic, 7-point clinician-administered (observer-rated) scale that assesses change in illness severity. The score ranging from 1 (very much improved) through 7 (very much worse). A higher score indicates worsening of disease.

Achromatopsia (ACHM) Vision Impact Questionnaire (AVIQ)Baseline up to 8 years

The AVIQ was developed to assess the impact of ACHM on functional vision in children and adults. The AVIQ uses 5-point or 6-point Likert scales. There are 3 age versions: the adult/adolescent (\>= 12 years) version includes 15 items, the child (8-11 years) version includes 8 items, and the caregiver (5-7 years) version includes 5 items. Scores will be described per age class separately.

Association Between Inherited Retinal Disease (IRD) Genotype and Visual AcuityBaseline up to 8 years

Association between IRD genotype and visual acuity will be reported as incidence of visual acuity for given IRD genotype.

Family History and Inheritance PatternBaseline up to 8 years

Number and relationship with family members diagnosed with IRD will be described.

Demographic Characteristics of Participants: AgeBaseline

Demographic characteristics of participants (age) will be reported.

Demographic Characteristics of Participants: SexBaseline

Demographic characteristics of participants (sex) will be reported.

Demographic Characteristics of Participants: RaceBaseline

Demographic characteristics of participants (race) will be reported.

Number of Participants With ComorbiditiesBaseline

Number of participants with comorbidities will be reported.

Number of Participants With Other Ocular EventsBaseline up to 8 years

Number of participants with other ocular events will be reported. Other ocular events of interest including cystoid macular edema, macular hole, epiretinal membrane formation, intraocular inflammation, cataracts, glaucoma, chorioretinal atrophy will be described.

Number and Type of Hospital/Clinic Visit After IRD DiagnosisBaseline up to 8 years

Participant management after diagnosis including number and type of hospital/clinic visit will be described.

Clinician Global Impression of Severity (CGIS)Baseline up to 8 years

CGIS score will be reported for participants with X-linked retinitis pigmentosa (XLRP) and achromatopsia (ACHM) separately. CGIS is a generic, global, 5-point clinician-administered (observer-rated) scale that assesses illness severity. The score ranging from 1 (no symptoms) through 5 (very severe) to assess disease severity. A higher score indicates more severe disease.

Number and Type of Healthcare Professional Visits Prior to Confirmed IRD DiagnosisBaseline up to 8 years

Participant diagnostic pathway prior to diagnosis including number and type of healthcare professional visits will be described.

Medical Resource UtilizationBaseline up to 8 years

Number and type of hospital/clinic visit, use of assistive device, supportive care, adaptation, and service will be described.

Participant Global Impression of Severity (PGIS)Baseline up to 8 years

The PGIS is a 5-point scale to assess disease severity, for participants with XLRP and ACHM separately. The XLRP PGIS measures participant reported disease severity and impact of XLRP on daily activities, items include: daily activities, mobility, mobility under low luminance/at night, and global rating of severity. A higher score indicates more severe disease. The ACHM PGIS measures participant reported disease severity and impact of ACHM on daily activities, items include: photo aversion (indoors and outdoors), impact on daily activities, and global rating of severity. A higher score indicates more severe disease.

Participant Global Impression of Change (PGIC)First post-baseline visit up to 8 years

PGIC is a 5-point scale to assess the patient-reported change in disease severity. The XLRP PGIC assesses participant reported perceived change in disease severity and impact of XLRP on daily activities, items include: daily activities, mobility, mobility under low luminance/at night, and global rating of change in severity. A higher score indicates worsening of disease.

Modified Low Luminance Questionnaire (mLLQ)Baseline up to 8 years

The mLLQ is a modified version of the original low luminance questionnaire developed for use in eye diseases to assess self-reported task difficulty under low luminance and at night. The mLLQ uses 5-point or 6-point Likert scales, consists of 6 domains: driving, extreme lighting, mobility, emotional distress, general dim lighting, and peripheral vision. There are 3 age versions: the adult (greater than or equal to \[\>=\] 18 years) version includes 30 items, the adolescent (12-17 years) version includes 22 items, and the caregiver (3-11 years) version includes 19 items. Each domain has a score range of 0-100, with higher scores reflecting a higher level of functioning. Scores will be described per age class separately only for participants with XLRP.

Achromatopsia (ACHM) Symptom and Impact DiaryBaseline up to 8 years

The ACHM symptom and impact diary assesses the severity of key symptoms of photosensitivity and impaired visual acuity, contrast sensitivity, and color vision. There are 3 age versions: the adult/adolescent (\>=12 years) version includes 9 items, the child (8-11 years) version includes 9 items, and the caregiver (5-7 years) version includes 16 items. Scores will be described per age class separately.

Caregiver Burden ScoreBaseline up to 8 years

The caregiver burden score is a 14-item questionnaire developed to assess the impact of IRDs on caregivers of children (ages 3-17 years) with an IRD diagnosis, using 4-point or 6-point Likert scales. It measures caregiver burden in terms of perception of their physical and emotional health, relationships, social life, work, and finances. This questionnaire applies to caregivers of minors only.

Work Productivity and Activity Impairment (WPAI)Baseline up to 8 years

The WPAI is a 6-item questionnaire that measures the effects of IRD symptoms on work productivity and absenteeism and activity impairment outside of work, using dichotomous (Yes/No) and 0-10 numerical rating scale. The productivity loss would be the total work impairment; the sum of absenteeism and presenteeism. Scores are expressed as impairment percentages, with higher scores reflecting more impairment. This questionnaire will be answered by both study participants and caregiver participants.

Trial Locations

Locations (74)

University of Alabama Birmingham

🇺🇸

Birmingham, Alabama, United States

University of Arkansas for Medical Sciences

🇺🇸

Little Rock, Arkansas, United States

University of Southern California

🇺🇸

Los Angeles, California, United States

UCSF

🇺🇸

San Francisco, California, United States

Bascom Palmer Eye Institute

🇺🇸

Miami, Florida, United States

Emory University

🇺🇸

Atlanta, Georgia, United States

University of Iowa

🇺🇸

Iowa City, Iowa, United States

Ochsner Medical Center

🇺🇸

New Orleans, Louisiana, United States

John Hopkins Hospital

🇺🇸

Baltimore, Maryland, United States

Univ of Michigan Medical Center

🇺🇸

Ann Arbor, Michigan, United States

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University of Alabama Birmingham
🇺🇸Birmingham, Alabama, United States

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