Using n-of-1 methodology to inform the development of individualised, evidence-based interventions for patients with xeroderma pigmentosum

Kirby Sainsbury, Jessica Walburn, Rute Vieira, Falko F Sniehotta, John Weinman, Robert Sarkany, Vera Araujo-Soares

Research output: Contribution to journalAbstract

Abstract

Introduction: XP is a very rare inherited disease affecting 1 in 250,000
people (~90 UK patients). It involves extreme sensitivity to UV light; the
only treatment is complete avoidance of UV – not going outside during
daylight, fitting UV screens to windows, or ensuring complete skin coverage.
Most patients die by age 35, usually from skin cancers. No research
on the psychological consequences of XP or factors relating to UV protection
currently exists.
Methods: In phase one, an N-of-1 study was conducted in 25 adult XP
patients, as part of a mixed methods approach to intervention development.
Participants wore a UV wristwatch and completed a brief survey,
including adherence behaviors and psychological predictors (e.g., emotions,
self-regulation), for 50 consecutive days. Constructs were selected
from the theory domains framework and a review of behavioral maintenance
theories.
Results: N-of-1 analysis revealed differences in the patterns of predictors of
adherence across participants, supporting the need for an individualized
approach. Following MRC guidelines, phase two involves the development
of a series of individualized interventions to improve adherence, informed
directly by the specific predictors identified for each participant in phase
one (e.g., including BCTs to target motivation, planning, or emotions).
After PPI feedback and piloting, the interventions will be tested in a
waitlist-controlled RCT in a matched sample of non-adherent adults.
Conclusions: The XP study provides a novel example of systematic intervention
development in a rare and unstudied condition, which could be
used as a model for understanding and improving health outcomes in
other rare diseases.
Original languageEnglish
Pages (from-to)S46
JournalInternational Journal of Behavioral Medicine
Volume23
Issue numberSuppl. 1
Publication statusPublished - 2016
EventInternational Congress of Behavioral Medicine - Melbourne, Australia
Duration: 7 Dec 2016 → …

Fingerprint

Xeroderma Pigmentosum
Rare Diseases
Emotions
Psychology
Skin Neoplasms
Ultraviolet Rays
Motivation
Guidelines
Skin
Health
Therapeutics
Self-Control
Surveys and Questionnaires

Cite this

Using n-of-1 methodology to inform the development of individualised, evidence-based interventions for patients with xeroderma pigmentosum. / Sainsbury, Kirby; Walburn, Jessica; Vieira, Rute; Sniehotta, Falko F; Weinman, John; Sarkany, Robert; Araujo-Soares, Vera.

In: International Journal of Behavioral Medicine, Vol. 23, No. Suppl. 1, 2016, p. S46.

Research output: Contribution to journalAbstract

Sainsbury, Kirby ; Walburn, Jessica ; Vieira, Rute ; Sniehotta, Falko F ; Weinman, John ; Sarkany, Robert ; Araujo-Soares, Vera. / Using n-of-1 methodology to inform the development of individualised, evidence-based interventions for patients with xeroderma pigmentosum. In: International Journal of Behavioral Medicine. 2016 ; Vol. 23, No. Suppl. 1. pp. S46.
@article{6e30305396a14fc8b1ce0c176e174cff,
title = "Using n-of-1 methodology to inform the development of individualised, evidence-based interventions for patients with xeroderma pigmentosum",
abstract = "Introduction: XP is a very rare inherited disease affecting 1 in 250,000people (~90 UK patients). It involves extreme sensitivity to UV light; theonly treatment is complete avoidance of UV – not going outside duringdaylight, fitting UV screens to windows, or ensuring complete skin coverage.Most patients die by age 35, usually from skin cancers. No researchon the psychological consequences of XP or factors relating to UV protectioncurrently exists.Methods: In phase one, an N-of-1 study was conducted in 25 adult XPpatients, as part of a mixed methods approach to intervention development.Participants wore a UV wristwatch and completed a brief survey,including adherence behaviors and psychological predictors (e.g., emotions,self-regulation), for 50 consecutive days. Constructs were selectedfrom the theory domains framework and a review of behavioral maintenancetheories.Results: N-of-1 analysis revealed differences in the patterns of predictors ofadherence across participants, supporting the need for an individualizedapproach. Following MRC guidelines, phase two involves the developmentof a series of individualized interventions to improve adherence, informeddirectly by the specific predictors identified for each participant in phaseone (e.g., including BCTs to target motivation, planning, or emotions).After PPI feedback and piloting, the interventions will be tested in awaitlist-controlled RCT in a matched sample of non-adherent adults.Conclusions: The XP study provides a novel example of systematic interventiondevelopment in a rare and unstudied condition, which could beused as a model for understanding and improving health outcomes inother rare diseases.",
author = "Kirby Sainsbury and Jessica Walburn and Rute Vieira and Sniehotta, {Falko F} and John Weinman and Robert Sarkany and Vera Araujo-Soares",
year = "2016",
language = "English",
volume = "23",
pages = "S46",
journal = "International Journal of Behavioral Medicine",
issn = "1070-5503",
publisher = "Routledge",
number = "Suppl. 1",

}

TY - JOUR

T1 - Using n-of-1 methodology to inform the development of individualised, evidence-based interventions for patients with xeroderma pigmentosum

AU - Sainsbury, Kirby

AU - Walburn, Jessica

AU - Vieira, Rute

AU - Sniehotta, Falko F

AU - Weinman, John

AU - Sarkany, Robert

AU - Araujo-Soares, Vera

PY - 2016

Y1 - 2016

N2 - Introduction: XP is a very rare inherited disease affecting 1 in 250,000people (~90 UK patients). It involves extreme sensitivity to UV light; theonly treatment is complete avoidance of UV – not going outside duringdaylight, fitting UV screens to windows, or ensuring complete skin coverage.Most patients die by age 35, usually from skin cancers. No researchon the psychological consequences of XP or factors relating to UV protectioncurrently exists.Methods: In phase one, an N-of-1 study was conducted in 25 adult XPpatients, as part of a mixed methods approach to intervention development.Participants wore a UV wristwatch and completed a brief survey,including adherence behaviors and psychological predictors (e.g., emotions,self-regulation), for 50 consecutive days. Constructs were selectedfrom the theory domains framework and a review of behavioral maintenancetheories.Results: N-of-1 analysis revealed differences in the patterns of predictors ofadherence across participants, supporting the need for an individualizedapproach. Following MRC guidelines, phase two involves the developmentof a series of individualized interventions to improve adherence, informeddirectly by the specific predictors identified for each participant in phaseone (e.g., including BCTs to target motivation, planning, or emotions).After PPI feedback and piloting, the interventions will be tested in awaitlist-controlled RCT in a matched sample of non-adherent adults.Conclusions: The XP study provides a novel example of systematic interventiondevelopment in a rare and unstudied condition, which could beused as a model for understanding and improving health outcomes inother rare diseases.

AB - Introduction: XP is a very rare inherited disease affecting 1 in 250,000people (~90 UK patients). It involves extreme sensitivity to UV light; theonly treatment is complete avoidance of UV – not going outside duringdaylight, fitting UV screens to windows, or ensuring complete skin coverage.Most patients die by age 35, usually from skin cancers. No researchon the psychological consequences of XP or factors relating to UV protectioncurrently exists.Methods: In phase one, an N-of-1 study was conducted in 25 adult XPpatients, as part of a mixed methods approach to intervention development.Participants wore a UV wristwatch and completed a brief survey,including adherence behaviors and psychological predictors (e.g., emotions,self-regulation), for 50 consecutive days. Constructs were selectedfrom the theory domains framework and a review of behavioral maintenancetheories.Results: N-of-1 analysis revealed differences in the patterns of predictors ofadherence across participants, supporting the need for an individualizedapproach. Following MRC guidelines, phase two involves the developmentof a series of individualized interventions to improve adherence, informeddirectly by the specific predictors identified for each participant in phaseone (e.g., including BCTs to target motivation, planning, or emotions).After PPI feedback and piloting, the interventions will be tested in awaitlist-controlled RCT in a matched sample of non-adherent adults.Conclusions: The XP study provides a novel example of systematic interventiondevelopment in a rare and unstudied condition, which could beused as a model for understanding and improving health outcomes inother rare diseases.

M3 - Abstract

VL - 23

SP - S46

JO - International Journal of Behavioral Medicine

JF - International Journal of Behavioral Medicine

SN - 1070-5503

IS - Suppl. 1

ER -