Quality of life among children with spina bifida in Uganda

Sims‐Williams, Helen; Sims-Williams, Hugh P; Kabachelor, Edith Mbabazi; Warf, Benjamin C.

doi:10.1136/archdischild-2016-312307

Cited by 30 publications

(21 citation statements)

References 24 publications

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“…Neural tube defects (NTDs) are a group of severe congenital disorders associated with substantial mortality, morbidity, long‐term disability, and psychological and economic costs . Many NTDs are preventable with folic acid, and long‐term survival and quality of life among those living with NTDs can be improved through access to appropriate clinical care and rehabilitative services . However, efforts for primary prevention and addressing the needs of those living with NTD have been hampered by a lack of transparent prevalence estimates to quantify the burden, especially in resource‐poor settings.…”

Section: Introductionmentioning

confidence: 99%

“…1 Many NTDs are preventable with folic acid, [2][3][4] and long-term survival and quality of life among those living with NTDs can be improved through access to appropriate clinical care and rehabilitative services. [5][6][7] However, efforts for primary prevention and addressing the needs of those living with NTD have been hampered by a lack of transparent prevalence estimates to quantify the burden, especially in resource-poor settings. The Modell Database of Congenital Disorders (MGDb) was developed recently to estimate the birth prevalence of congenital disorders globally.…”

Section: Introductionmentioning

confidence: 99%

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Estimates of global and regional prevalence of neural tube defects for 2015: a systematic analysis

Blencowe

Kancherla

Moorthie

et al. 2018

Annals of the New York Academy of Sciences

242

233

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Neural tube defects (NTDs) are associated with substantial mortality, morbidity, disability, and psychological and economic costs. Many are preventable with folic acid, and access to appropriate services for those affected can improve survival and quality of life. We used a compartmental model to estimate global and regional birth prevalence of NTDs (live births, stillbirths, and elective terminations of pregnancy) and subsequent under-5 mortality. Data were identified through web-based reviews of birth defect registry databases and systematic literature reviews. Meta-analyses were undertaken where appropriate. For 2015, our model estimated 260,100 (uncertainty interval (UI): 213,800-322,000) NTD-affected birth outcomes worldwide (prevalence 18.6 (15.3-23.0)/10,000 live births). Approximately 50% of cases were elective terminations of pregnancy for fetal anomalies (UI: 59,300 (47,900-74,500)) or stillbirths (57,800 (UI: 35,000-88,600)). Of NTD-affected live births, 117,900 (ß75%) (UI: 105,500-186,600) resulted in under-5 deaths. Our systematic review showed a paucity of high-quality data in the regions of the world with the highest burden. Despite knowledge about prevention, NTDs remain highly prevalent worldwide. Lack of surveillance and incomplete ascertainment of affected pregnancies make NTDs invisible to policy makers. Improved surveillance of all adverse outcomes is needed to improve the robustness of total NTD prevalence estimation, evaluate effectiveness of prevention through folic acid fortification, and improve outcomes through care and rehabilitation.

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Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Estimates of global and regional prevalence of neural tube defects for 2015: a systematic analysis

Blencowe

Kancherla

Moorthie

et al. 2018

Annals of the New York Academy of Sciences

242

233

View full text Add to dashboard Cite

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“…We recently found that male sex, large family size, urinary incontinence and hydrocephalus were all associated with poorer self-reported quality of life (QOL) among 62 children aged 10–14 with spina bifida from Eastern Uganda 1. A limitation of this study was the absence of corresponding QOL scores for unaffected Ugandan children.…”

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confidence: 90%

Children with spina bifida in Eastern Uganda report a reasonable quality of life relative to their healthy school-attending peers

et al. 2018

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“…Two studies reported various levels of correlation between PBMs and other outcome measures in SB [21,54]. Child self-reported HUI3 utility scores were not found to be highly correlated with VAS scores (r = 0.488) [54].…”

Section: Convergent Validity: Comparing Measuresmentioning

confidence: 99%

“…The sample sizes of relevant study groups ranged from 13 [15] to 770 [44]. Fourteen studies included data relating to CP [22, 33, 35, 36, 39, 46, 48-51, 53, 55, 57, 58], six included data relating to SB [21,38,51,52,54,56] and five included data relating to childhood hydrocephalus [40][41][42][43]45]. Data for a range of other relevant conditions were also found in either single studies or from extractable subgroups, these included muscular dystrophy [34,44], spinal muscular atrophy [47], Morquio A syndrome [37], congenital clubfoot [59] and microcephaly [51].…”

Section: Study and Patient Characteristicsmentioning

confidence: 99%

Preference-based measures of health-related quality of life in congenital mobility impairment: a systematic review of validity and responsiveness

2020

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Introduction: Mobility impairment is the leading cause of disability in the UK. Individuals with congenital mobility impairments have unique experiences of health, quality of life and adaptation. Preference-based outcomes measures are often used to help inform decisions about healthcare funding and prioritisation, however the applicability and accuracy of these measures in the context of congenital mobility impairment is unclear. Inaccurate outcome measures could potentially affect the care provided to these patient groups. The aim of this systematic review was to examine the performance of preference-based outcome measures for the measurement of utility values in various forms of congenital mobility impairment. Methods: Ten databases were searched, including Science Direct, CINAHL and PubMed. Screening of reference lists and hand-searching were also undertaken. Descriptive and narrative syntheses were conducted to combine and analyse the various findings. Results were grouped by condition. Outcome measure performance indicators were adapted from COSMIN guidance and were grouped into three broad categories: validity, responsiveness and reliability. Screening, data extraction and quality appraisal were carried out by two independent reviewers. Results: A total of 31 studies were considered eligible for inclusion in the systematic review. The vast majority of studies related to either cerebral palsy, spina bifida or childhood hydrocephalus. Other relevant conditions included muscular dystrophy, spinal muscular atrophy and congenital clubfoot. The most commonly used preference-based outcome measure was the HUI3. Reporting of performance properties predominantly centred around construct validity, through known group analyses and assessment of convergent validity between comparable measures and different types of respondents. A small number of studies assessed responsiveness, but assessment of reliability was not reported. Increased clinical severity appears to be associated with decreased utility outcomes in congenital mobility impairment, particularly in terms of gross motor function in cerebral palsy and lesion level in spina bifida. However, preference-based measures exhibit limited correlation with various other condition-specific and clinically relevant outcome measures.(Continued on next page) Conclusion: Preference-based measures exhibit important issues and discrepancies relating to validity and responsiveness in the context of congenital mobility impairment, thus care must be taken when utilising these measures in conditions associated with congenital mobility impairments.

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Quality of life among children with spina bifida in Uganda

Cited by 30 publications

References 24 publications

Estimates of global and regional prevalence of neural tube defects for 2015: a systematic analysis

Estimates of global and regional prevalence of neural tube defects for 2015: a systematic analysis

Children with spina bifida in Eastern Uganda report a reasonable quality of life relative to their healthy school-attending peers

Preference-based measures of health-related quality of life in congenital mobility impairment: a systematic review of validity and responsiveness

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