Health-related quality of life in children with osteogenesis imperfecta: a large-sample study

Song, Yuwen; Zhao, Dongsheng; Li, L; Lv, Fang; Wang, O; Jiang, Yan; Xia, Weibo; Xing, Xiaoping; Li, M

doi:10.1007/s00198-018-4801-5

Cited by 29 publications

(22 citation statements)

References 32 publications

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“…Self-perceived health status was investigated, and mobility was reduced in the total sample, and the lowest mobility was detected in children with type III, while a positive trend was that many children reported results similar to the general population in other domains, findings consistent with previous research [ 29 , 30 ]. Confirming results were also presented in a review concerning QoL in children and adults with OI, where the authors concluded that physical QoL appeared to be lower than in the general population, while the mental and psychosocial QoL was equal or better [ 31 ].…”

Section: Discussionsupporting

confidence: 86%

Exploring pain interference and self-perceived health status in children with osteogenesis imperfecta - a cross-sectional study

Provenzano

Albrecht

Löwing

2022

BMC Musculoskelet Disord

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Background Chronic pain may affect and interfere in children’s everyday life and can be present in children with Osteogenesis Imperfecta (OI). However, the knowledge is still sparse to what extent pain is present, how pain interfere in children’s everyday life and affect their self-perceived health status. The purpose of the study was therefore to explore presence of chronic pain, pain interference in daily life, and self-perceived health status in children with OI. Methods Children with OI, aged 6–18 years, were recruited consecutively to this cross-sectional study. Participants answered a standardised interview including five pre-structured questions, and the Numeric Pain Rating Scale (NPRS), the Pain Interference Index, and a questionnaire concerning self-perceived health status the Patient Reported Outcomes Measurement Information System Pediatric-25 Profile v1.1 (PROMIS-25). Results Twenty-eight children (median: 11 years, IQR 6) with OI type I, III, or IV participated. Pain was present in 27 of 28 children and interfered in their everyday life regardless of OI-type, sex, and age. The median NPRS for average pain intensity was 4 (IQR 2), the median for pain frequency was 2–3 times/week, and the median frequency of school absence due to pain was 2–3 times per month. The most common pain locations were back and feet. Pain in the feet was more frequently reported in children with type I (p = 0.032), and pain in the hip was more often reported in children ≥13 years (p = 0.011). The children were asked what they thought to be the cause of pain and the most frequent response was “walking long distances”. Self-perceived health status for mobility was lower than the general population, and lowest for children with type III (p = 0.016). Pain interference was associated with children’s self-perceived health status (rs = 0.84, p < 0.001). Conclusion Almost all children experienced pain, which interfered in children’s everyday lives, affected participation in various activities and was associated with reduced self-perceived health status. If children avoid physical activities because of pain, it might cause a vicious circle of inactivity, which further decreases bone density and increase the risk of fractures. The results emphasize the importance to offer adequate pain reducing interventions.

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Section: Discussionsupporting

confidence: 86%

Exploring pain interference and self-perceived health status in children with osteogenesis imperfecta - a cross-sectional study

Provenzano

Albrecht

Löwing

2022

BMC Musculoskelet Disord

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“…Studies have found that children with more severe types of OI (types III and IV) had significantly lower HRQoL scores than those with milder (type I) OI. They also found that children with OI scored lowest in the physical domains of HRQoL tools (Dahan-Oliel et al 2016;Song et al 2019). Comparing these to our study, we see that whilst children with types III and IV OI (severe) did score lower than those with type I OI (mild), the difference was not significant.…”

Section: Discussionsupporting

confidence: 57%

Oral health-related quality of life in children with osteogenesis imperfecta

Mintoff

Riddington

Parekh

2021

Eur Arch Paediatr Dent

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Purpose Osteogenesis imperfecta (OI) results from mutations in the genes involved in the modification or biosynthesis of collagen. This study aimed to assess the oral health-related quality of life (OHRQoL) in children with OI. Methodology Participants were recruited from a highly specialised OI centre for children. The Child Oral-Health Impact Profile—Short Form (COHIP-SF) was used, adding demographic and qualitative questions. Children aged 8–16 years participated between January and October 2019. Statistical analysis was carried out. A higher COHIP-SF score indicates better OHRQoL (maximum score, 76). Results One hundred and six (106) children participated (44 female, mean age 11.93 years). COHIP-SF median score was 59. Children reporting mild OI (n = 55) had higher median scores (62) compared to severe OI (n = 7) with median scores of 55 (P = 0.087). When comparing mixed (< 12 years, n = 46) and permanent dentition (≥ 12, n = 60), no significant difference in OHRQoL was seen (P = 0.977). There was no significant difference between severities for each COHIP-SF domain. Limited data on the presence of dentinogenesis imperfecta did not impact overall score (P = 0.109), but was significant in the oral-health domain (P = 0.033). Qualitative Common themes were the need for braces, discolouration, pain and function. Conclusion This study confirmed that children with OI have dental concerns in areas including oral health, functional well-being and socio-emotional well-being. This was related to severity of OI.

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“…The study conducted by Y. Song et al (2018) [ 41 ] and Vanz A. et al (2018) [ 42 ] used the PedsQL scale to quantify the HRQoL in children with osteogenesis imperfecta. Their results for the mean punctuation on the PedsQL scale (65.6 ± 23.8 vs. 71.13 ± 12 vs. 76.2 ± 17) were slightly lower than those obtained in our study.…”

Section: Discussionmentioning

confidence: 99%

Quality of Life in Children and Adolescents with Stickler Syndrome in Spain

et al. 2022

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Objective: To describe the quality of life and daily functioning of Spanish children and adolescents living with Stickler syndrome (SS) and to estimate the prevalence of associated disease features in a representative sample. Methods: A cross-sectional study of children and adolescents with SS were recruited via telephone calls through the Spanish SS Association. All participants underwent a structured clinical interview and filled in questionnaires reporting their quality of life (EuroQol-5D, TSK-11, CHAQ and PedsQoL). The prevalence of the main features associated with the syndrome and the mean scores of the questionnaires were estimated with 95% confidence intervals (95% CI). Results: The recruited sample included 26 persons who were mainly children (mean age 10.4 ± 4.5 (SD) range: 5–14) and male (65.4%). The prevalence estimates of SS features were as follows: the presence of moderate pain (52%), hearing loss 67% (95% CI: 54.8 to 91.3) and myopia 96% (95% CI: 87.2 to 104.4). The mean scores of the QoL indices were as follows: 22.4 (95% CI: 19.2 to 25.5) (± 7.5) for TSK-11; 76.2 (95% CI: 68.8 to 83.6) (± 17.1) for PedsQoL, 0.8 (95% CI: 0.7 to 0.9) (± 0.3) for EQ-5D and 0.61 (95% CI: 0.24 to 1.0) (± 0.9) for the cHAQ functional index. Conclusions: Our results confirmed a high variability in syndrome-related manifestations, with a large prevalence of visual and hearing deficits, pain and maxillofacial alterations. These findings may facilitate the detection of the most prevalent problems in this population, which could be a target to be addressed during the treatment of children and adolescents with SS.

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Health-related quality of life in children with osteogenesis imperfecta: a large-sample study

Cited by 29 publications

References 32 publications

Exploring pain interference and self-perceived health status in children with osteogenesis imperfecta - a cross-sectional study

Exploring pain interference and self-perceived health status in children with osteogenesis imperfecta - a cross-sectional study

Oral health-related quality of life in children with osteogenesis imperfecta

Quality of Life in Children and Adolescents with Stickler Syndrome in Spain

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