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1.
BMJ Open ; 13(1): e064774, 2023 01 06.
Article in English | MEDLINE | ID: mdl-36609326

ABSTRACT

OBJECTIVES: To assess the diagnostic accuracy of non-invasive or minimally invasive autopsy techniques in deaths under 1 year of age. DESIGN: This is a systematic review of diagnostic test accuracy. The protocol is registered on PROSPERO. PARTICIPANTS: Deaths from conception to one adjusted year of age. SEARCH METHODS: MEDLINE (Ovid), EMBASE (Ovid), CINAHL (EBSCO), the Cochrane Library, Scopus and grey literature sources were searched from inception to November 2021. DIAGNOSTIC TESTS: Non-invasive or minimally invasive diagnostic tests as an alternative to traditional autopsy. DATA COLLECTION AND ANALYSIS: Studies were included if participants were under one adjusted year of age, with index tests conducted prior to the reference standard.Data were extracted from eligible studies using piloted forms. Risk of bias was assessed using Quality Assessment of Diagnostic Accuracy Studies-2. A narrative synthesis was conducted following the Synthesis without Meta-Analysis guidelines. Vote counting was used to assess the direction of effect. MAIN OUTCOME MEASURES: Direction of effect was expressed as percentage of patients per study. FINDINGS: We included 54 direct evidence studies (68 articles/trials), encompassing 3268 cases and eight index tests. The direction of effect was positive for postmortem ultrasound and antenatal echography, although with varying levels of success. Conversely, the direction of effect was against virtual autopsy. For the remaining tests, the direction of effect was inconclusive.A further 134 indirect evidence studies (135 articles/trials) were included, encompassing 6242 perinatal cases. The addition of these results had minimal impact on the direct findings yet did reveal other techniques, which may be favourable alternatives to autopsy.Seven trial registrations were included but yielded no results. CONCLUSIONS: Current evidence is insufficient to make firm conclusions about the generalised use of non-invasive or minimally invasive autopsy techniques in relation to all perinatal population groups.PROSPERO registration numberCRD42021223254.


Subject(s)
Diagnostic Tests, Routine , Infant, Newborn , Pregnancy , Humans , Infant , Female , Autopsy , Cause of Death , Ultrasonography , Sensitivity and Specificity
2.
Birth Defects Res ; 113(20): 1431-1462, 2021 12 01.
Article in English | MEDLINE | ID: mdl-34672115

ABSTRACT

Children with congenital anomalies have poorer intellectual and cognitive development compared to their peers, but evidence for academic achievement using objective measures is lacking. We aimed to summarize and synthesize evidence on academic outcomes and special education needs (SEN) of school-aged children born with selected major structural congenital anomalies. Electronic databases (MEDLINE, EMBASE, Scopus, PsycINFO, CINAHL, ProQuest Natural Science and Education Collections), reference lists and citations for 1990-2020 were systematically searched. We included original-research articles on academic achievement in children with non-syndromic congenital anomalies that involved school test results, standardized tests and/or SEN data. Random-effects meta-analyses were performed to estimate pooled mean test scores in mathematics and/or reading where possible and pooled odds ratios (ORs) for SEN in children with severe congenital heart defects (CHDs) and children with orofacial clefts (OFCs). Thirty-nine eligible studies (n = 21,066 children) were synthesized narratively. Sixteen studies were included in meta-analyses. Children with non-syndromic congenital anomalies were at a higher risk of academic underachievement than controls across school levels. Children with severe CHD (pooled OR = 2.32, 95% CI: 1.90, 2.82), and children with OFC (OR = 1.38 (95% CI: 1.20, 1.57), OR = 3.07 (95% CI: 2.65, 3.56), and OR = 3.96 (95% CI: 3.31, 4.72) for children with cleft lip, cleft palate and cleft lip/palate, respectively) had significantly higher ORs for SEN than controls. Children with non-syndromic congenital anomalies underperform academically and have higher SEN rates compared to their peers. Early monitoring and development of differential SEN are important to promote academic progress in these children.


Subject(s)
Academic Success , Cleft Lip , Cleft Palate , Child , Educational Status , Humans , Schools
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