Abstract
ObjectivesA systematic review was conducted in high-income country settings to analyse: (i) spina bifida neonatal and IMRs over time, and (ii) clinical and socio-demographic factors associated with mortality in the first year after birth in infants affected by spina bifida.Data sourcesPubMed, Embase, Ovid, Web of Science, CINAHL, Scopus and the Cochrane Library were searched from 1st January, 1990 to 31st August, 2020 to review evidence.Study selectionPopulation-based studies that provided data for spina bifida infant mortality and case fatality according to clinical and socio-demographical characteristics were included. Studies were excluded if they were conducted solely in tertiary centres. Spina bifida occulta or syndromal spina bifida were excluded where possible.Data extraction and synthesisIndependent reviewers extracted data and assessed their quality using MOOSE guideline. Pooled mortality estimates were calculated using random-effects (+/- fixed effects) models meta-analyses. Heterogeneity between studies was assessed using the Cochrane Q test and I2 statistics. Meta-regression was performed to examine the impact of year of birth cohort on spina bifida infant mortality.ResultsTwenty studies met the full inclusion criteria with a total study population of over 30 million liveborn infants and approximately 12,000 spina bifida-affected infants. Significant declines in spina bifida associated infant and neonatal mortality rates (e.g. 4.76% decrease in IMR per 100, 000 live births per year) and case fatality (e.g. 2.70% decrease in infant case fatality per year) were consistently observed over time. Preterm birth (RR 4.45; 2.30–8.60) and low birthweight (RR 4.77; 2.67–8.55) are the strongest risk factors associated with increased spina bifida infant case fatality.SignificanceSignificant declines in spina bifida associated infant/neonatal mortality and case fatality were consistently observed, advances in treatment and mandatory folic acid food fortification both likely play an important role. Particular attention is warranted from clinicians caring for preterm and low birthweight babies affected by spina bifida.
Highlights
Neural tube defects (NTDs) constitute the largest group of congenital anomalies of the central nervous system [1]; the aetiology of spina bifida is multifactorial [2,3]
Significant declines in spina bifida associated infant and neonatal mortality rates (e.g. 4.76% decrease in IMR per 100, 000 live births per year) and case fatality
Significant declines in spina bifida associated infant/neonatal mortality and case fatality were consistently observed, advances in treatment and mandatory folic acid food fortification both likely play an important role
Summary
Neural tube defects (NTDs) constitute the largest group of congenital anomalies of the central nervous system [1]; the aetiology of spina bifida is multifactorial [2,3]. Mortality among infants with spina bifida has been previously investigated in several studies, mainly restricted to selected geographical regions especially in high-income countries where data are more available [4]. Infant mortality associated with spina bifida has been changing over time depending on various factors including folic acid supplementation and food fortification programme [5], prenatal screening [6], treatment and termination of pregnancy [7], and the health care system to tract and link all cases with death registers [4]. We conducted a systematic review and metaanalysis of population-based studies focusing on liveborn infants with spina bifida in highincome countries. The aims of this study were to assess: (1) spina bifida- specific neonatal and infant mortality rates over time; and (2) the socio-demographic and clinical factors associated with mortality in the first year for infants affected by spina bifida
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