Long-term survival of children born with congenital anomalies: A systematic review and meta-analysis of population-based studies

Abstract

Background Following a reduction in global child mortality due to communicable diseases, the relative contribution of congenital anomalies to child mortality is increasing. Although infant survival of children born with congenital anomalies has improved for many anomaly types in recent decades, there is less evidence on survival beyond infancy. We aimed to systematically review, summarise, and quantify the existing population-based data on long-term survival of individuals born with specific major congenital anomalies and examine the factors associated with survival. Methods and findings Seven electronic databases (Medline, Embase, Scopus, PsycINFO, CINAHL, ProQuest Natural, and Biological Science Collections), reference lists, and citations of the included articles for studies published 1 January 1995 to 30 April 2020 were searched. Screening for eligibility, data extraction, and quality appraisal were performed in duplicate. We included original population-based studies that reported long-term survival (beyond 1 year of life) of children born with a major congenital anomaly with the follow-up starting from birth that were published in the English language as peer-reviewed papers. Studies on congenital heart defects (CHDs) were excluded because of a recent systematic review of population-based studies of CHD survival. Meta-analysis was performed to pool survival estimates, accounting for trends over time. Of 10,888 identified articles, 55 (n = 367,801 live births) met the inclusion criteria and were summarised narratively, 41 studies (n = 54,676) investigating eight congenital anomaly types (

European trends in mortality in children with congenital anomalies: 2000–2015

“…Research has shown that the survival of infants with congenital anomalies has improved in recent years (Glinianaia et al, 2020; Schneuer et al, 2019; Tennant, Pearce, Bythell, & Rankin, 2010; Wang, Hu, Druschel, & Kirby, 2011). However, other studies have found no significant change in survival (Dastgiri, Gilmour, & Stone, 2003; Roncancio et al, 2018).…”

Timeliness of postnatal surgery in newborns with open neural tube defects: a single center experience

“…14,15 Another poor prognostic factor for newborns with NTDs has been addressed as concurrence of NTDs with other congenital abnormalities, chromosomal aberrations and genetic syndromes. 14,16 The major reason for this observation is that isolated cases of NTD tend to be less complicated. 16 On the other hand, it has been shown that early intervention after birth enhances the survival and well being of the neonates diagnosed with NTDs.…”

“…14,16 The major reason for this observation is that isolated cases of NTD tend to be less complicated. 16 On the other hand, it has been shown that early intervention after birth enhances the survival and well being of the neonates diagnosed with NTDs. 14,15 The timing of surgery for postnatal treatment of NTD remains a controversial topic in the literature.…”

Linking a European cohort of children born with congenital anomalies to vital statistics and mortality records: A EUROlinkCAT study

“…The European surveillance of congenital anomalies (EUROCAT) network of population-based CA registries provides essential epidemiologic information and surveillance on CAs in Europe but information is mainly collected up to a baby's first year of life [2][3][4]. There is little information on survival after one-year of age in Europe [5], with studies either analysing all anomalies combined [6] or concentrating on a few specific anomalies, such as spina bifida or Down syndrome [7,8]. One study investigated 20-year survival for a range of CAs in the North of England, but was unable to report survival for many rare CAs due to small numbers [9].…”