Children with facial morphoea managing everyday life: a qualitative study

Stasiulis, Elaine; Gladstone, Brenda; Boydell, Katherine M.; O’Brien, Claire; Pope, Elena; Laxer, Ronald M.

doi:10.1111/bjd.16449

Cited by 11 publications

(39 citation statements)

References 37 publications

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“…Complementarily to their work, we found that individuals with all LS subtypes were bothered by ‘feeling different’, and that this was influenced by the visibility of their skin involvement, social support (or lack thereof) and their developmental age. Furthermore, the treatment burden reported by our participants was remarkably high compared with studies reporting from the physicians’ perspective, but was consistent with other qualitative and quantitative work . Expanding on the results of Stasiulis et al ., we found that youth with limb involvement also reported serious and impactful physical functioning limitations and related muscle and joint pain and stiffness (Table ), which are important to consider clinically.…”

Section: Discussionsupporting

confidence: 91%

“…Additionally, our findings replicated and expanded on some of the major themes identified in the 2018 qualitative paper of Stasiulis et al . Complementarily to their work, we found that individuals with all LS subtypes were bothered by ‘feeling different’, and that this was influenced by the visibility of their skin involvement, social support (or lack thereof) and their developmental age.…”

Section: Discussionsupporting

confidence: 84%

“…4 Furthermore, a psychometric evaluation of an existing HRQoL tool used in LS suggested that psychosocial effects like feeling embarrassed, experiencing teasing or bullying, or covering skin with clothing were important to this population, but incompletely assessed. 17 Additionally, our findings replicated and expanded on some of the major themes identified in the 2018 qualitative paper of Stasiulis et al 5 Complementarily to their work, we found that individuals with all LS subtypes were bothered by 'feeling different', and that this was influenced by the visibility of their skin involvement, social support (or lack thereof) and their developmental age. Furthermore, the treatment burden reported by our participants was remarkably high compared Table 5 Descriptions of common and distressing side-effects from treatment with systemic medications as per youth focus groups with children and adolescents diagnosed with localized scleroderma and their caregivers…”

Section: Discussionsupporting

confidence: 83%

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Exploring the impact of paediatric localized scleroderma on health‐related quality of life: focus groups with youth and caregivers

et al. 2020

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Background Paediatric localized scleroderma (LS) can negatively impact healthrelated quality of life (HRQoL) by causing skin fibrosis, abnormal limb development, disfigurement, and side-effects from immunosuppressive treatment. Studies to date have rarely included qualitative data gathered directly from paediatric patients with LS. Objectives To assess the impact of LS on HRQoL among affected youth and their caregivers using qualitative description. Methods Youth with all subtypes of LS and their caregivers were purposively sampled to participate in age-appropriate focus groups (younger children, early adolescents, adolescents). Each group started with a drawing exercise followed by in-depth discussion of topics including skin symptoms (e.g. itch, pain, tightness), functional impairment, physical appearance, family and peer relationships, and treatment burden. Focus groups were transcribed verbatim and co-coded, with adjudication of differentially applied codes. The study findings were triangulated via comparison with adult reports and published literature. Results Eleven youth aged 9-16 years and 16 caregivers participated in three focus groups each. Major identified areas of impact included uncomfortable skin symptoms, physical functioning limitations, extracutaneous manifestations, body image, bullying and teasing, unwanted questioning from others, and treatment side-effects and burden. Conclusions This is the first qualitative study of HRQoL in LS to include all major LS subtypes. We identified domains of HRQoL impacted by LS, some of which replicate earlier findings and some of which were novel. As impact also changed with developmental stage, our findings support the need for ongoing, formal evaluation of HRQoL in children and adolescents with LS. What is already known about this topic? • Paediatric localized scleroderma (LS) negatively impacts health-related quality of life (HRQoL) via skin fibrosis, musculoskeletal and other extracutaneous manifestations from the disease process, and side-effects of systemic immunosuppression. • The full impact of LS and its treatment on HRQoL is incompletely understood, with only one published qualitative study of youth with LS, which was limited to facial involvement. • There are no qualitative studies of HRQoL in other LS subtypes to date.

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Section: Discussionsupporting

confidence: 91%

Section: Discussionsupporting

confidence: 84%

Section: Discussionsupporting

confidence: 83%

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Exploring the impact of paediatric localized scleroderma on health‐related quality of life: focus groups with youth and caregivers

et al. 2020

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“…These areas of impact make the experience of having paediatric LS a unique one that has only recently been qualitatively described in the literature in Zigler et al . (submitted) and Stasiulis et al …”

Section: Health‐related Quality Of Life Patient‐reported Outcome Measmentioning

confidence: 87%

“…Three items were subsequently added based on participant feedback. The conceptual framework was also supported by independent qualitative work from a separate institution . Although only individuals with facial involvement participated (compared with a mix of subtypes in the focus groups that were used to inform this PROM development), themes were nearly identical.…”

Section: Discussionmentioning

confidence: 99%

A novel patient‐reported outcome for paediatric localized scleroderma: a qualitative assessment of content validity

et al. 2019

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Summary Background According to current standards, no existing patient‐reported outcome (PRO) measures have high‐quality validity evidence for use with individuals diagnosed with paediatric localized scleroderma (LS). This severely hinders patient‐centred LS‐focused research, including much needed clinical trials. Objectives To develop a valid health‐related quality of life measure for individuals with paediatric LS and to qualitatively evaluate its content validity using a patient‐centred approach. Methods Previously collected qualitative data from youth with LS and their caregivers was used to develop items. The resulting item set was administered in a clinical setting to participants aged 8–18 years old. Cognitive interviews were used to evaluate time to survey completion, readability/understanding of the items, appropriateness of the recall period and construct representation. Results Seventeen children and adolescents with LS participated in the study. Interviews supported readability, understanding of the items and appropriateness of the recall period in individuals > 10 years old. Revisions were made to simplify the instructions and to be more inclusive of different subtypes of LS. Three items were added to improve content representation. Conclusions Content validity was supported by the patient‐centred development process of the outcome measure and via direct feedback from individuals with LS and their families. Although an important first step, the resulting PRO, termed the Localized Scleroderma Quality of Life Instrument, should be further evaluated in a larger sample before being implemented. What's already known about this topic? No current health‐related quality of life (HRQoL) measures have been created using direct input from children and adolescents with localized scleroderma (LS). When compared with qualitative reports of HRQoL impact in youth with all LS subtypes, no existing patient‐reported outcome (PRO) measures have appropriate content validity for individuals with paediatric LS. What does this study add? This study proposes a novel LS‐specific PRO and is the first qualitative assessment of content validity for any PRO measure in this population. Results from cognitive interviews with children and adolescents support the content validity of the newly developed item set and its ability to capture HRQoL impact in a clinical context. What are the clinical implications of this work? Incorporating a content‐valid PRO of HRQoL impact into clinical practice would allow for the valid, ongoing capture of patient experience in LS. Although content validity is an important and necessary step in the process of evaluating validity, items within this novel measure will undergo additional psychometric evaluation before implementation in research and clinical settings.

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Systematic Review of Health‐Related Quality of Life Impact in Juvenile Localized Scleroderma

Sanchez‐Espino,

Luca,

Pope

et al. 2024

Arthritis Care & Research

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IntroductionThe prevalence and types of psychosocial complications of juvenile localized scleroderma (JLS), also known as morphea, an inflammatory and sclerosing disease involving the skin, fascia, muscle, and bone are poorly understood.MethodsWe performed a systematic review of PUBMED, EMBASE, Cochrane Skin Group Specialized Register, Cochrane Central Register of Controlled Trials (CENTRAL), and the Cumulative Index to Nursing and Allied Health Literature (CINAHL) between 2000 to 2020 using search terms: “scleroderma, localized” or “Morphea”, “anxiety” or “depression” or “resilience” or “social stigma” or “quality of life” or “mood” or “stress” and limited to pediatric patients and English language. Patient demographics, characteristics of JLS, and comorbidities were extracted. Outcomes included: measures of health‐related quality of life (HRQoL), psychosocial functioning, evaluation of self‐perception and treatment burden of the study population. The protocol was registered with PROSPERO (CRD42021257124). Thematic synthesis generated descriptive analysis.ResultsThirteen studies fulfilled the inclusion criteria: three retrospective cohort studies, two prospective cohort studies and eight cross‐sectional studies. A total of 690 pediatric patients with JLS were included (n=484 with linear scleroderma). Six studies used the Children's Dermatology Life Quality Index, reporting small to no‐effect in HRQoL. One study used the health‐Related Quality of Life in Children and Adolescents Questionnaire, and did not find differences between children with JLS, atopic dermatitis and healthy controls. One study used a self‐perception questionnaire that showed normal self‐worth of patients with JLS. Two studies used focus groups, both reporting elevated levels of stress, decreased self‐worth, “feeling different”, and bullying/teasing in JLS patients. These emotions were associated with skin symptoms (pain, itch, and tightness), physical limitations, and treatment burden.ConclusionsOverall, quantitative studies did not report a statistically significant impairment in HRQoL in JLS. However, qualitative studies (focus groups) reported significant psychosocial impacts related to JLS. There is a need to develop a JLS‐specific tool for HRQoL evaluation of this population.This article is protected by copyright. All rights reserved.

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Children with facial morphoea managing everyday life: a qualitative study

Cited by 11 publications

References 37 publications

Exploring the impact of paediatric localized scleroderma on health‐related quality of life: focus groups with youth and caregivers

Exploring the impact of paediatric localized scleroderma on health‐related quality of life: focus groups with youth and caregivers

A novel patient‐reported outcome for paediatric localized scleroderma: a qualitative assessment of content validity

Systematic Review of Health‐Related Quality of Life Impact in Juvenile Localized Scleroderma

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