Risk factors for mortality in infancy and childhood in children with major congenital anomalies: A European population-based cohort study

Joachim Tan; Svetlana V. Glinianaia; Judith Rankin; Anna Pierini; Michele Santoro; Alessio Coi; Ester Garne; Maria Loane; Joanne E. Given; Joanna Brigden; Elisa Ballardini; Clara Cavero-Carbonell; Hermien E.K. de Walle; Laura García-Villodre; Miriam Gatt; Mika Gissler; Anna Heino; Sue Jordan; Babak Khoshnood; Kari Klungsoyr; Nathalie Lelong; Renée L. Lutke; Amanda J. Neville; David Tucker; Stine K. Urhoj; Diana Wellesley; Joan K. Morris

doi:10.1111/ppe.13010

Risk factors for mortality in infancy and childhood in children with major congenital anomalies: A European population-based cohort study

Joachim Tan^*
, Svetlana V. Glinianaia
, Judith Rankin
, Anna Pierini
, Michele Santoro
, Alessio Coi
, Ester Garne
, Maria Loane
, Joanne E. Given
, Joanna Brigden
, Elisa Ballardini
, Clara Cavero-Carbonell
, Hermien E.K. de Walle
, Laura García-Villodre
, Miriam Gatt
, Mika Gissler
, Anna Heino
, Sue Jordan
, Babak Khoshnood
, Kari Klungsoyr

Nathalie Lelong, Renée L. Lutke, Amanda J. Neville, David Tucker, Stine K. Urhoj, Diana Wellesley, Joan K. Morris

^*Corresponding author for this work

St George's, University of London
Newcastle University
Institute of Clinical Physiology
Ulster University
Foundation for the Promotion of Health and Biomedical Research in the Valencian Region
University Medical Center Groningen
Directorate for Health Information and Research
National Institute for Health and Welfare
Region Stockholm
Karolinska Institute
Swansea University
INSERM UMR 1153
University of Bergen
Norwegian Institute of Public Health
University Hospital of Ferrara
Public Health Wales NHS Trust
University of Southampton
University of Ferrara
University of Copenhagen

Research output: Contribution to journal › Journal article › Research › peer-review

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Abstract

Background: Preterm birth and young maternal age are known risk factors for infant and childhood mortality. There is limited knowledge of the impact of these risk factors in children born with major congenital anomalies (CAs), who have inherently higher risks of death compared with other children. Objectives: To investigate the risk factors for mortality up to age 10 years in children born with specific major CAs. Methods: This population-based cohort study involved 150,198 livebirths from 1995 to 2014 in 13 European CA registries linked to mortality data. Cox proportional hazards models estimated the association of gestational age, maternal age and child's sex with death <1 year and 1–9 years for the whole cohort and by CA subgroup. Hazard ratios (HR) from each registry were pooled using multivariate meta-analysis. Results: Preterm birth had a dose–response association with mortality; compared with infants born at 37+ weeks gestation, those born at <28, 28–31 and 32–36 weeks had 14.88 (95% CI 12.57, 17.62), 8.39 (95% CI 7.16, 9.85) and 3.88 (95% CI 3.40, 4.43) times higher risk of death <1 year, respectively. The corresponding risks at 1–9 years were 4.99 (95% CI 2.94, 8.48), 3.09 (95% CI 2.28, 4.18) and 2.04 (95% CI 1.69, 2.46) times higher, respectively. Maternal age <20 years (versus 20–34 years) was a risk factor for death <1 year (HR 1.30, 95% CI 1.09, 1.54) and 1–9 years (HR 1.58, 95% CI 1.19, 2.10). Females had 1.22 (95% CI 1.07, 1.39) times higher risk of death between 1 and 9 years than males. Conclusion: Preterm birth was associated with considerably higher infant and childhood mortality in children with CAs, comparable to estimates reported elsewhere for the background population. Additional risk factors included young maternal age and female sex. Information on risk factors could benefit clinical care and guide counselling of parents following CA diagnoses.

Original language	English
Journal	Paediatric and Perinatal Epidemiology
Volume	37
Issue number	8
Pages (from-to)	679-690
ISSN	0269-5022
DOIs	https://doi.org/10.1111/ppe.13010
Publication status	Published - Nov 2023

Bibliographical note

Publisher Copyright:
© 2023 The Authors. Paediatric and Perinatal Epidemiology published by John Wiley & Sons Ltd.

Funding

This project has received funding from the European Union's Horizon 2020 research and innovation programme under grant agreement No 733001. Start Date: 1 Jan 2017. Duration: 5 years 5 months. ( https://ec.europa.eu/programmes/horizon2020/en ). We thank EUROlinkCAT contributors to this paper for their work on the project (including data provision, linkage and standardisation; writing and running of syntax scripts). Ms Abigail Reid (St George's, University of London, London, United Kingdom); Ms Nicole Siemensma-Muhlenberg (University Medical Center Groningen, Groningen, Netherlands); Sandra Moreno Marro, Laia Barrachina Bonet (Foundation for the Promotion of Health and Biomedical Research in the Valencian Region, Valencia, Spain); Dr Sonja Kiuru-Kuhlefelt, Tuuli Puroharju (THL Finnish Institute for Health and Welfare, Helsinki, Finland); Drs Gianni Astolfi, Aurora Puccini, Annarita Armaroli (Center for Clinical and Epidemiological Research, University of Ferrara, Ferrara, Italy); Nathalie Bertille and Makan Rahshenas (INSERM, Paris, France); Mr Daniel Thayer (Faculty of Medicine, Health and Life Science, Swansea University, Swansea, Wales, UK); Professor Elizabeth Draper (University of Leicester, Leicester, United Kingdom); Professor Jenny Kurinczuk (University of Oxford, Oxford, United Kingdom). We also thank Mr Hugh Claridge for the project management.

Keywords

congenital anomalies
gestational age
maternal age
mortality
risk factors
Pregnancy, Multiple
Humans
Risk Factors
Male
Infant
Pregnancy
Young Adult
Maternal Age
Premature Birth/epidemiology
Female
Adult
Registries
Child
Infant, Newborn
Cohort Studies

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Tan, J., Glinianaia, S. V., Rankin, J., Pierini, A., Santoro, M., Coi, A., Garne, E., Loane, M., Given, J. E., Brigden, J., Ballardini, E., Cavero-Carbonell, C., de Walle, H. E. K., García-Villodre, L., Gatt, M., Gissler, M., Heino, A., Jordan, S., Khoshnood, B., ... Morris, J. K. (2023). Risk factors for mortality in infancy and childhood in children with major congenital anomalies: A European population-based cohort study. Paediatric and Perinatal Epidemiology, 37(8), 679-690. https://doi.org/10.1111/ppe.13010

@article{85892142679f4165870b4adee490a0ec,

title = "Risk factors for mortality in infancy and childhood in children with major congenital anomalies: A European population-based cohort study",

abstract = "Background: Preterm birth and young maternal age are known risk factors for infant and childhood mortality. There is limited knowledge of the impact of these risk factors in children born with major congenital anomalies (CAs), who have inherently higher risks of death compared with other children. Objectives: To investigate the risk factors for mortality up to age 10 years in children born with specific major CAs. Methods: This population-based cohort study involved 150,198 livebirths from 1995 to 2014 in 13 European CA registries linked to mortality data. Cox proportional hazards models estimated the association of gestational age, maternal age and child's sex with death <1 year and 1{\textendash}9 years for the whole cohort and by CA subgroup. Hazard ratios (HR) from each registry were pooled using multivariate meta-analysis. Results: Preterm birth had a dose{\textendash}response association with mortality; compared with infants born at 37+ weeks gestation, those born at <28, 28{\textendash}31 and 32{\textendash}36 weeks had 14.88 (95\% CI 12.57, 17.62), 8.39 (95\% CI 7.16, 9.85) and 3.88 (95\% CI 3.40, 4.43) times higher risk of death <1 year, respectively. The corresponding risks at 1{\textendash}9 years were 4.99 (95\% CI 2.94, 8.48), 3.09 (95\% CI 2.28, 4.18) and 2.04 (95\% CI 1.69, 2.46) times higher, respectively. Maternal age <20 years (versus 20{\textendash}34 years) was a risk factor for death <1 year (HR 1.30, 95\% CI 1.09, 1.54) and 1{\textendash}9 years (HR 1.58, 95\% CI 1.19, 2.10). Females had 1.22 (95\% CI 1.07, 1.39) times higher risk of death between 1 and 9 years than males. Conclusion: Preterm birth was associated with considerably higher infant and childhood mortality in children with CAs, comparable to estimates reported elsewhere for the background population. Additional risk factors included young maternal age and female sex. Information on risk factors could benefit clinical care and guide counselling of parents following CA diagnoses.",

keywords = "congenital anomalies, gestational age, maternal age, mortality, risk factors, Pregnancy, Multiple, Humans, Risk Factors, Male, Infant, Pregnancy, Young Adult, Maternal Age, Premature Birth/epidemiology, Female, Adult, Registries, Child, Infant, Newborn, Cohort Studies",

author = "Joachim Tan and Glinianaia, \{Svetlana V.\} and Judith Rankin and Anna Pierini and Michele Santoro and Alessio Coi and Ester Garne and Maria Loane and Given, \{Joanne E.\} and Joanna Brigden and Elisa Ballardini and Clara Cavero-Carbonell and \{de Walle\}, \{Hermien E.K.\} and Laura Garc{\'i}a-Villodre and Miriam Gatt and Mika Gissler and Anna Heino and Sue Jordan and Babak Khoshnood and Kari Klungsoyr and Nathalie Lelong and Lutke, \{Ren{\'e}e L.\} and Neville, \{Amanda J.\} and David Tucker and Urhoj, \{Stine K.\} and Diana Wellesley and Morris, \{Joan K.\}",

note = "Publisher Copyright: {\textcopyright} 2023 The Authors. Paediatric and Perinatal Epidemiology published by John Wiley \& Sons Ltd.",

year = "2023",

month = nov,

doi = "10.1111/ppe.13010",

language = "English",

volume = "37",

pages = "679--690",

journal = "Paediatric and Perinatal Epidemiology",

issn = "0269-5022",

publisher = "Wiley",

number = "8",

}

Tan, J, Glinianaia, SV, Rankin, J, Pierini, A, Santoro, M, Coi, A, Garne, E, Loane, M, Given, JE, Brigden, J, Ballardini, E, Cavero-Carbonell, C, de Walle, HEK, García-Villodre, L, Gatt, M, Gissler, M, Heino, A, Jordan, S, Khoshnood, B, Klungsoyr, K, Lelong, N, Lutke, RL, Neville, AJ, Tucker, D, Urhoj, SK, Wellesley, D & Morris, JK 2023, 'Risk factors for mortality in infancy and childhood in children with major congenital anomalies: A European population-based cohort study', Paediatric and Perinatal Epidemiology, vol. 37, no. 8, pp. 679-690. https://doi.org/10.1111/ppe.13010

Risk factors for mortality in infancy and childhood in children with major congenital anomalies: A European population-based cohort study. / Tan, Joachim; Glinianaia, Svetlana V.; Rankin, Judith et al.
In: Paediatric and Perinatal Epidemiology, Vol. 37, No. 8, 11.2023, p. 679-690.

Research output: Contribution to journal › Journal article › Research › peer-review

TY - JOUR

T1 - Risk factors for mortality in infancy and childhood in children with major congenital anomalies

T2 - A European population-based cohort study

AU - Tan, Joachim

AU - Glinianaia, Svetlana V.

AU - Rankin, Judith

AU - Pierini, Anna

AU - Santoro, Michele

AU - Coi, Alessio

AU - Garne, Ester

AU - Loane, Maria

AU - Given, Joanne E.

AU - Brigden, Joanna

AU - Ballardini, Elisa

AU - Cavero-Carbonell, Clara

AU - de Walle, Hermien E.K.

AU - García-Villodre, Laura

AU - Gatt, Miriam

AU - Gissler, Mika

AU - Heino, Anna

AU - Jordan, Sue

AU - Khoshnood, Babak

AU - Klungsoyr, Kari

AU - Lelong, Nathalie

AU - Lutke, Renée L.

AU - Neville, Amanda J.

AU - Tucker, David

AU - Urhoj, Stine K.

AU - Wellesley, Diana

AU - Morris, Joan K.

PY - 2023/11

Y1 - 2023/11

N2 - Background: Preterm birth and young maternal age are known risk factors for infant and childhood mortality. There is limited knowledge of the impact of these risk factors in children born with major congenital anomalies (CAs), who have inherently higher risks of death compared with other children. Objectives: To investigate the risk factors for mortality up to age 10 years in children born with specific major CAs. Methods: This population-based cohort study involved 150,198 livebirths from 1995 to 2014 in 13 European CA registries linked to mortality data. Cox proportional hazards models estimated the association of gestational age, maternal age and child's sex with death <1 year and 1–9 years for the whole cohort and by CA subgroup. Hazard ratios (HR) from each registry were pooled using multivariate meta-analysis. Results: Preterm birth had a dose–response association with mortality; compared with infants born at 37+ weeks gestation, those born at <28, 28–31 and 32–36 weeks had 14.88 (95% CI 12.57, 17.62), 8.39 (95% CI 7.16, 9.85) and 3.88 (95% CI 3.40, 4.43) times higher risk of death <1 year, respectively. The corresponding risks at 1–9 years were 4.99 (95% CI 2.94, 8.48), 3.09 (95% CI 2.28, 4.18) and 2.04 (95% CI 1.69, 2.46) times higher, respectively. Maternal age <20 years (versus 20–34 years) was a risk factor for death <1 year (HR 1.30, 95% CI 1.09, 1.54) and 1–9 years (HR 1.58, 95% CI 1.19, 2.10). Females had 1.22 (95% CI 1.07, 1.39) times higher risk of death between 1 and 9 years than males. Conclusion: Preterm birth was associated with considerably higher infant and childhood mortality in children with CAs, comparable to estimates reported elsewhere for the background population. Additional risk factors included young maternal age and female sex. Information on risk factors could benefit clinical care and guide counselling of parents following CA diagnoses.

AB - Background: Preterm birth and young maternal age are known risk factors for infant and childhood mortality. There is limited knowledge of the impact of these risk factors in children born with major congenital anomalies (CAs), who have inherently higher risks of death compared with other children. Objectives: To investigate the risk factors for mortality up to age 10 years in children born with specific major CAs. Methods: This population-based cohort study involved 150,198 livebirths from 1995 to 2014 in 13 European CA registries linked to mortality data. Cox proportional hazards models estimated the association of gestational age, maternal age and child's sex with death <1 year and 1–9 years for the whole cohort and by CA subgroup. Hazard ratios (HR) from each registry were pooled using multivariate meta-analysis. Results: Preterm birth had a dose–response association with mortality; compared with infants born at 37+ weeks gestation, those born at <28, 28–31 and 32–36 weeks had 14.88 (95% CI 12.57, 17.62), 8.39 (95% CI 7.16, 9.85) and 3.88 (95% CI 3.40, 4.43) times higher risk of death <1 year, respectively. The corresponding risks at 1–9 years were 4.99 (95% CI 2.94, 8.48), 3.09 (95% CI 2.28, 4.18) and 2.04 (95% CI 1.69, 2.46) times higher, respectively. Maternal age <20 years (versus 20–34 years) was a risk factor for death <1 year (HR 1.30, 95% CI 1.09, 1.54) and 1–9 years (HR 1.58, 95% CI 1.19, 2.10). Females had 1.22 (95% CI 1.07, 1.39) times higher risk of death between 1 and 9 years than males. Conclusion: Preterm birth was associated with considerably higher infant and childhood mortality in children with CAs, comparable to estimates reported elsewhere for the background population. Additional risk factors included young maternal age and female sex. Information on risk factors could benefit clinical care and guide counselling of parents following CA diagnoses.

KW - congenital anomalies

KW - gestational age

KW - maternal age

KW - mortality

KW - risk factors

KW - Pregnancy, Multiple

KW - Humans

KW - Risk Factors

KW - Male

KW - Infant

KW - Pregnancy

KW - Young Adult

KW - Maternal Age

KW - Premature Birth/epidemiology

KW - Female

KW - Adult

KW - Registries

KW - Child

KW - Infant, Newborn

KW - Cohort Studies

U2 - 10.1111/ppe.13010

DO - 10.1111/ppe.13010

M3 - Journal article

C2 - 37817457

AN - SCOPUS:85173638085

SN - 0269-5022

VL - 37

SP - 679

EP - 690

JO - Paediatric and Perinatal Epidemiology

JF - Paediatric and Perinatal Epidemiology

IS - 8

ER -

Risk factors for mortality in infancy and childhood in children with major congenital anomalies: A European population-based cohort study

Abstract

Bibliographical note

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Keywords

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