Table 1.
Baseline characteristics of included studies (n = 15)
| Author, year | Type of study | Operated/born/tested | Age tests | Tests | Reference population | No. of patients included* | Mortality (n) | No. at follow-up | Type of EA | Gestational age (weeks) birth weight | Comorbidities |
|---|---|---|---|---|---|---|---|---|---|---|---|
| Bouman27 Netherlands, 1999 |
Prospective cohort study | NR | 8–12 years | WISC-RN | Dutch references | 36 | NR | 36 | Isolated EA n = 5 EA with TEF n = 31 |
NR NR |
NR |
| Faugli36 Norway, 2009 |
Prospective cohort study | Born 1999–2002 | 1 year | BSID-II | US references | 44 | 2 | 39 (36) | 10% delayed repair | 23% born <37 weeks 2830 (595–4570)A |
20% ≥1 associated anomaly (tetralogy of Fallot, biliary atresia, anorectal malformation, tracheomalacia) |
| Gischler36 Netherlands, 2009 |
Prospective longitudinal cohort study | Tested 1999–2003 | 6, 12, 18, and 24 months | BSID-I/II** | Dutch references | 17 | NR | 13 | NR | 38.6 (36.9–40.1)B 3000 (2600–3200)B |
Syndromal/chromosomal n = 1, severe neurologic impairment n = 2, major congenital anomalies n = 1# |
| Van der Cammen-van Zijp25 Netherlands, 2010 |
Prospective cohort study | Born 1999–2003 | 5 years | MABC | Dutch references | 29 | NR | 29 | NR | 38.4 (28.6–42.0)A 2900 (800–4500) |
31% ≥1 associated anomaly |
| Kubota29 Japan, 2011 |
Prospective cohort study | NR | 6–17 years | WISC-3 KSPD |
Japanese references | 23 | NR | 23 | NR | NR NR |
NR |
| Walker35 Australia, 2013 |
Prospective case–control study | Operated Aug 2006–Dec 2008 | 1 year | Bayley-3 | Study control group | 34 | 1 | 31 | NR | 37.6C 2718 ± 717D |
44% ≥1 associated anomaly |
| Francesca34 Italy, 2020 |
Observational prospective cohort study | Born 2009–2017 | 6 and 12 months | Bayley-3 | Age-normed | 90 | NR | 82 59 |
Type C and D | 38 (37–39)B 2700 (2450–3030) |
NR |
| Bakal20 Turkey, 2016 |
Cross-sectional study | Operated Jan 1996–Dec 2011 | 6–16 years | ADSI WISC-R |
Turkish references | 57 | 18 | 24 ADSI 15 WISC-R |
Type A n = 6 Type C n = 50 Type E n = 1 |
40% born <37 weeks 2255.26 ± 600.27D |
35% ≥1 associated anomaly |
| Giúdici23 Argentina, 2016 |
Prospective cohort study | Born Jan 2003–Dec 2014 | 1, 3, and 6 years | CAT/ CLAMS PRUNAPE |
Argentinian references | 23 | 4 | 21 at 1 year 14 at 3 years 10 at 6 years |
Type A n = 3 Type C n = 20 |
38.3 ± 1.6D 2917 ± 440D |
Trisomy 21 n = 1, Edwards syndrome n = 1 |
| Walker26 Australia, 2016 |
Prospective case–control study | Operated Aug 2006–Dec 2008 | 3 years | Bayley-3 | Study control group | 31 | 0 | 24 | NR | 38C 2765C |
NR |
| Harmsen24 Netherlands, 2017 |
Prospective cohort study | Born Jan 1999–May 2006 | 5 and 8 years | MABC I/II WISC-3-NL RAKIT |
Dutch references | 78 | 7 | 54 motor 49 cognitive |
91% type C | 39 (29–42)A 2830 (750–4505)A |
12% cardiac anomaly, 5% VACTERL association |
| König28 Germany, 2018 |
Cross-sectional study | NR | 3–12 years | Deutscher Motorik Test | German references | 17 | NR | 12 | NR | 54% born <37 weeks 23% <1500 grams |
46% congenital heart disease, 38% developmental delay, 28% skeletal deformity, 15% anorectal malformation |
| Mawlana17 Canada, 2018 |
Retrospective chart review | Operated Jan 2000–Dec 2015 | 2–3 years | Bayley-3 | US references | 253 | 21 | 182 | Type A n = 13 Type B n = 2 Type C n = 149 Type D n = 4 Type E n = 14 |
36.8 ± 3.2D 2589 ± 800D |
NR |
| Costerus21 Netherlands, 2019 |
Prospective cohort study | Operated Aug 2011–Aug 2013 | 1 and 2 years | BSID-II | Dutch references | 6 | NR | 5 | NR | 39.0 (34.0–40.0)A 2850 (1941–3338)A |
Tetralogy of Fallot n = 1, kidney dysplasia n = 1, Feingold syndrome n = 1, intestinal malrotation n = 1 |
| Batta37 Australia, 2020 |
Retrospective study | Born 2005–2014 | 1 year | GMDS-II | General population references | 44 | 1 | 27 | NR | 37.6 (36.4–39.1)B 3000 (2590–3405)B |
NR |
EA, esophageal atresia; NR, not reported; TEF, tracheoesophageal fistula. Type of EA according to gross classification.54 VACTERL, vertebral, anorectal, tracheoesophageal, renal, or limb defects55
AMedian (range).
BMedian (IQR).
CMean.
DMean ± SD.
These four patients were excluded from neurodevelopmental assessment.
*Included in neurodevelopmental assessment.
**Dutch version of the BISD I/II: BOS 2-30.