UNICORNS: Uveitis in Childhood Prospective National Cohort Study (UNICORNS)

Uveitis in Childhood Prospective National Cohort Study

Childhood uveitis (inflammation inside the eye) is an uncommon disorder that carries the risk of blindness. Inadequate treatment of active inflammation has been shown to be related to a poor outcome. There has been no population-based, prospective longitudinal study of all-cause childhood uveitis, with resultant limitations in the evidence base used to counsel affected families, balance treatment decisions, or plan further research. The aim of the study is to describe the characteristics of childhood-onset uveitis and describe outcomes. The investigators shall also aim to identify the socio-demographic, clinical, biological and treatment-related determinants of outcome. Early (1-2 years following diagnosis) outcomes will be described in the first instance: However, through the creation of a national inception cohort, the investigators shall enable longer-term studies of outcome for affected children and families. There will be no change to routine clinical care.

Study Overview

• Status: Active, not recruiting
• Conditions: Uveitis

Detailed Description

Background:

Childhood uveitis descriptive term for a group of rare inflammatory eye disease, which are typically chronic, relapsing-remitting in nature, and of uncertain aetiology (idiopathic). Visual loss occurs due to structural damage caused by uncontrolled inflammation. Understanding of the determinants of long-term outcome is limited, particularly the predictors of therapeutic response or how to define disease control.

Aims:

To describe disease natural history and outcomes amongst a nationally representative group of children with non-infectious uveitis, describe the impact of disease course on quality of life for both child and family, and identify determinants of adverse visual, structural and developmental outcomes.

Methods:

UNICORNS is a prospective longitudinal multicentre cohort study of children newly diagnosed with uveitis about whom a core minimum clinical dataset will be collected. Participants and their families will also complete patient-reported outcome measures (PROMS) at study entry and annually from recruitment.

PROMS collected at baseline are:

• Strength and Difficulties Questionnaire (SDQ): this is to screen the emotional and behavioural aspects of the participants' lives
• Child Health Utility 9D (CHU9D): generic preference based measure of health related quality of life
• Children's Sleep Habits Questionnaire (CHSQ): this evaluates incidence of behaviours linked with typical paediatric sleep difficulties
• 100-mm General Evaluation (GE): visual analogue score 100mm long used to capture overall perception of the burden of disease

PROMS collected at year 1 and annually (subject to investigations of instrument redundancy) are:

• Child Health Utility 9D (CHU9D): generic preference based measure of health related quality of life
• 100-mm General Evaluation (GE): visual analogue score 100mm long used to capture overall perception of the burden of disease
• Paediatric Quality of life Score (PedsQL): a brief questionnaire used to evaluate health related quality of life in children
• Vision related quality of life metric - Children and Young People (VQoL_CYP): these instruments captures the functional and broader impacts of living with a visual disability The association of patient (child- and treatment- dependent) characteristics with outcome will be investigated using logistic and ordinal regression models which incorporate adjustment for within-child correspondence between eyes for those with bilateral disease and repeated outcomes measurement.

Discussion:

Through this population based, prospective longitudinal study of childhood uveitis, the investigators will describe the characteristics of childhood onset disease. Early (1-2 years following diagnosis) outcomes will be described in the first instance, and through the creation of a national inception cohort, longer term studies of outcome for affected children and families will be enabled.

• Study Type: Observational
• Enrollment (Estimated): 250

Contacts and Locations

• Study Contact: Name: Ameenat L Solebo; Phone Number: 0207 405 9200; Email: a.solebo@ucl.ac.uk
• Study Contact Backup: Name: Salomey F Kellett; Email: s.kellett@ucl.ac.uk

Study Locations

• United Kingdom
  • Belfast, United Kingdom, BT9 7AB
    • Belfast Royal Victoria Hospital NHS Foundation Trust
  • Birmingham, United Kingdom, B4 6NH
    • Birmingham Women's and Children's Hospital NHS Foundation Trust
  • Bournemouth, United Kingdom, BH7 7DW
    • Royal Bournemouth Christchurch Hospitals NHS Foundation Trust
  • Bradford, United Kingdom, BD9 6RJ
    • Bradford Teaching Hospitals NHS Foundation Trust
  • Bristol, United Kingdom, BS1 3NU
    • University Hospitals Bristol NHS Foundation Trust
  • Cambridge, United Kingdom, CB2 0QQ
    • Cambridge University Hospital NHS Foundation Trust
  • Cardiff, United Kingdom, CF14 4XW
    • Cardiff and Vale University Health Board Hospitals
  • Chelmsford, United Kingdom, CM1 7ET
    • Mid Essex Hospital Trust
  • Dundee, United Kingdom, DD1 9SY
    • Ninewells Hospital
  • Edinburgh, United Kingdom, EH1 3EG
    • Princess Alexandra Eye Pavilion
  • Glasgow, United Kingdom, G51 4TF
    • Royal Children's Hospital
  • Great Yarmouth, United Kingdom, NR31 6LA
    • James Paget University Hospitals NHS Foundation Trust
  • Hull, United Kingdom, HU3 2JZ
    • Hull University Teaching Hospitals NHS Trust
  • Leeds, United Kingdom, LS9 7TF
    • Leeds Teaching Hospitals NHS Trust
  • Liverpool, United Kingdom, L12 2AP
    • Alder Hey Hospital NHS Foundation Trust
  • London, United Kingdom, NW3 2QG
    • Royal Free London NHS Foundation Trust
  • London, United Kingdom, EC1V 2PD
    • Moorfields Eye Hospital NHS Foundation Trust
  • London, United Kingdom, SE1 7EH
    • St Thomas' Hospital
  • London, United Kingdom, WC1N 3JH
    • Great Ormond Street Hospital NHS Foundation Trust
  • Maidstone, United Kingdom, ME16 9QQ
    • Maidstone and Tunbridge Wells NHS Trust
  • Manchester, United Kingdom, M13 9WL
    • Manchester University NHS Foundation Trust
  • Newcastle, United Kingdom, NE1 4LP
    • Newcastle upon Tyne Hospitals NHS Foundation Trust
  • Norwich, United Kingdom, NR4 7UY
    • Norfolk and Norwich University Hospitals NHS Foundation Trust
  • Nottingham, United Kingdom, NG7 2UH
    • Nottingham University Hospitals NHS Trust
  • Oxford, United Kingdom, OX3 9DU
    • Oxford University Hospitals NHS Foundation Trust
  • Redhill, United Kingdom, RH1 5RH
    • Sussex Eye Hospital and Royal Alexandra Children's Hospital
  • Sheffield, United Kingdom, S10 2TH
    • Sheffield Children's Hospitals NHS Foundation Trust
  • Southampton, United Kingdom, SO16 6YD
    • University Hospital Southampton Nhs Foundation Trust
  • Westcliff-on-Sea, United Kingdom, SS0 0RY
    • Southend University Hospital NHS Foundation Trust
  • Whitechapel, United Kingdom, E1 1FR
    • Royal London Hospital
  • York, United Kingdom, YO31 8HE
    • York Teaching Hospital NHS Foundation Trust

Participation Criteria

Eligibility Criteria

• Ages Eligible for Study: No older than 18 years (Child, Adult)
• Accepts Healthy Volunteers: No

• Sampling Method: Non-Probability Sample

Study Population

Children those aged under 18 years old (at diagnosis) who are newly diagnosed with uveitis newly diagnosed with uveitis (within previous 6 months)

Description

Inclusion Criteria:

• Children newly diagnosed with uveitis (within previous 6 months)
• Age < 18years

Exclusion Criteria:

• Uveitis due to malignancy
• Uveitis due to ocular trauma (including iatrogenic, ie intraocular surgery)
• Uveitis due to confirmed ocular infection
• Children with developmental disorders or impairments which prevent their self-report of their function level or quality of life

Study Plan

How is the study designed?

Design Details

• Observational Models: Cohort
• Time Perspectives: Prospective

Number of groups / cohorts

1

Cohorts and Interventions

Group / Cohort
Childhood Uveitis; Children <18 years old newly diagnosed with non-infectious uveitis

What is the study measuring?

Primary Outcome Measures

Outcome Measure	Measure Description	Time Frame
Incidence of ocular complications	New incident of sight threatening ocular complications, including glaucoma, cataract and macular oedema	Up to 3 years
Pediatric Quality of Life Inventory Score	Quality of life (patient reported outcomes) using Pediatric Quality of Life Inventory (PedsQL) in which items are reversed scored and linearly transformed to a 0-100 scale, so that higher scores indicate better HRQOL (Health-Related Quality of Life)	Up to 3 years

Secondary Outcome Measures

Outcome Measure	Measure Description	Time Frame
Corticosteroid exposure	Total prescribed topical and systemic corticosteroid burden	Up to 3 years
Attainment of disease control	Number of children achieving absolute control defined as absence of inflammation Relative control defined as the absence of inflammation greater than 0.5 The lowest grade of inflammation, with the use of less than one drop of topical corticosteroid	Up to 3 years
Health Utility	Child Health Utility Metric (CHU9D), with scores ranging from 0.33 (worst health state) to 1.00 (best health state)	Up to 3 years
Change in Pediatric Quality of Life Inventory Score	Change in child reported quality of life using Pediatric Quality of Life Inventory (PedsQL) in which items are reversed scored and linearly transformed to a 0-100 scale, so that higher scores indicate better HRQOL (Health-Related Quality of Life), and a change of greater than 5 is clinically meaningful	Over one year from baseline to year one

Collaborators and Investigators

• Sponsor: Institute of Child Health
• Collaborators: National Institute for Health Research, United Kingdom
• Investigators: Principal Investigator: Ameenat L Solebo, University College London (UCL) Great Ormond Street Institute of Child Health

Publications and helpful links

General Publications

• Kellett S, Rahi JS, Dick AD et al. UNICORNS: Uveitis in childhood prospective national cohort study protocol [version 1; peer review: awaiting peer review]. F1000Research 2020, 9:1196 (https://doi.org/10.12688/f1000research.26689.1)

Helpful Links

• Study Website
• Study Video

Study record dates

Study Major Dates

• Study Start (Actual): October 22, 2020
• Primary Completion (Estimated): March 1, 2024
• Study Completion (Estimated): December 1, 2024

Study Registration Dates

• First Submitted: October 16, 2020
• First Submitted That Met QC Criteria: May 19, 2022
• First Posted (Actual): May 23, 2022

Study Record Updates

• Last Update Posted (Actual): June 29, 2023
• Last Update Submitted That Met QC Criteria: June 28, 2023
• Last Verified: June 1, 2023

More Information

Terms related to this study

• Keywords: Child; Quality of life; Vision; Prospective Cohort
• Additional Relevant MeSH Terms: Eye Diseases; Uveal Diseases; Uveitis
• Other Study ID Numbers: 258638

Plan for Individual participant data (IPD)

• Plan to Share Individual Participant Data (IPD)?: NO
• IPD Plan Description: Authorised collaborators will be granted access to aggregated anonymised data, following review of their research protocol study PI and Co-Is. Data and material transfer agreements will be required to be completed, in order to ensure regulatory compliance and that the interests of the participants are upheld and respected throughout.

Drug and device information, study documents

• Studies a U.S. FDA-regulated drug product: No
• Studies a U.S. FDA-regulated device product: No