Publication

Analysis of Mortality among Neonates and Children with Spina Bifida: An International Registry-Based Study, 2001-2012

Bakker, M. K., Kancherla, V., Canfield, M. A., Bermejo-Sanchez, E., Cragan, J. D., Dastgiri, S., De Walle, H. E. K., Feldkamp, M. L., Groisman, B., Gatt, M., Hurtado-Villa, P., Kallen, K., Landau, D., Lelong, N., Lopez Camelo, J. S., Martínez, L., Morgan, M., Mutchinick, O. M., Nembhard, W. N., Pierini, A., Rissmann, A., Sipek, A., Szabova, E., Tagliabue, G., Wertelecki, W., Zarante, I. & Mastroiacovo, P., Nov-2019, In : Paediatric and Perinatal Epidemiology. 33, 6, p. 436-448 13 p.

Research output: Contribution to journalArticleAcademicpeer-review

APA

Bakker, M. K., Kancherla, V., Canfield, M. A., Bermejo-Sanchez, E., Cragan, J. D., Dastgiri, S., ... Mastroiacovo, P. (2019). Analysis of Mortality among Neonates and Children with Spina Bifida: An International Registry-Based Study, 2001-2012. Paediatric and Perinatal Epidemiology, 33(6), 436-448. https://doi.org/10.1111/ppe.12589

Author

Bakker, Marian K ; Kancherla, Vijaya ; Canfield, Mark A ; Bermejo-Sanchez, Eva ; Cragan, Janet D ; Dastgiri, Saeed ; De Walle, Hermien E K ; Feldkamp, Marcia L ; Groisman, Boris ; Gatt, Miriam ; Hurtado-Villa, Paula ; Kallen, Karin ; Landau, Daniella ; Lelong, Nathalie ; Lopez Camelo, Jorge S ; Martínez, Laura ; Morgan, Margery ; Mutchinick, Osvaldo M ; Nembhard, Wendy N ; Pierini, Anna ; Rissmann, Anke ; Sipek, Antonin ; Szabova, Elena ; Tagliabue, Giovanna ; Wertelecki, Wladimir ; Zarante, Ignacio ; Mastroiacovo, Pierpaolo. / Analysis of Mortality among Neonates and Children with Spina Bifida : An International Registry-Based Study, 2001-2012. In: Paediatric and Perinatal Epidemiology. 2019 ; Vol. 33, No. 6. pp. 436-448.

Harvard

Bakker, MK, Kancherla, V, Canfield, MA, Bermejo-Sanchez, E, Cragan, JD, Dastgiri, S, De Walle, HEK, Feldkamp, ML, Groisman, B, Gatt, M, Hurtado-Villa, P, Kallen, K, Landau, D, Lelong, N, Lopez Camelo, JS, Martínez, L, Morgan, M, Mutchinick, OM, Nembhard, WN, Pierini, A, Rissmann, A, Sipek, A, Szabova, E, Tagliabue, G, Wertelecki, W, Zarante, I & Mastroiacovo, P 2019, 'Analysis of Mortality among Neonates and Children with Spina Bifida: An International Registry-Based Study, 2001-2012', Paediatric and Perinatal Epidemiology, vol. 33, no. 6, pp. 436-448. https://doi.org/10.1111/ppe.12589

Standard

Analysis of Mortality among Neonates and Children with Spina Bifida : An International Registry-Based Study, 2001-2012. / Bakker, Marian K; Kancherla, Vijaya; Canfield, Mark A; Bermejo-Sanchez, Eva; Cragan, Janet D; Dastgiri, Saeed; De Walle, Hermien E K; Feldkamp, Marcia L; Groisman, Boris; Gatt, Miriam; Hurtado-Villa, Paula; Kallen, Karin; Landau, Daniella; Lelong, Nathalie; Lopez Camelo, Jorge S; Martínez, Laura; Morgan, Margery; Mutchinick, Osvaldo M; Nembhard, Wendy N; Pierini, Anna; Rissmann, Anke; Sipek, Antonin; Szabova, Elena; Tagliabue, Giovanna; Wertelecki, Wladimir; Zarante, Ignacio; Mastroiacovo, Pierpaolo.

In: Paediatric and Perinatal Epidemiology, Vol. 33, No. 6, 11.2019, p. 436-448.

Research output: Contribution to journalArticleAcademicpeer-review

Vancouver

Bakker MK, Kancherla V, Canfield MA, Bermejo-Sanchez E, Cragan JD, Dastgiri S et al. Analysis of Mortality among Neonates and Children with Spina Bifida: An International Registry-Based Study, 2001-2012. Paediatric and Perinatal Epidemiology. 2019 Nov;33(6):436-448. https://doi.org/10.1111/ppe.12589


BibTeX

@article{cc7ffb6a67e043e89063cdcd59558683,
title = "Analysis of Mortality among Neonates and Children with Spina Bifida: An International Registry-Based Study, 2001-2012",
abstract = "BACKGROUND: Medical advancements have resulted in better survival and life expectancy among those with spina bifida, but a significantly increased risk of perinatal and postnatal mortality for individuals with spina bifida remains.OBJECTIVES: To examine stillbirth and infant and child mortality among those affected by spina bifida using data from multiple countries.METHODS: We conducted an observational study, using data from 24 population- and hospital-based surveillance registries in 18 countries contributing as members of the International Clearinghouse for Birth Defects Surveillance and Research (ICBDSR). Cases of spina bifida that resulted in livebirths or stillbirths from 20 weeks' gestation or elective termination of pregnancy for fetal anomaly (ETOPFA) were included. Among liveborn spina bifida cases, we calculated mortality at different ages as number of deaths among liveborn cases divided by total number of liveborn cases with spina bifida. As a secondary outcome measure, we estimated the prevalence of spina bifida per 10 000 total births. The 95{\%} confidence interval for the prevalence estimate was estimated using the Poisson approximation of binomial distribution.RESULTS: Between years 2001 and 2012, the overall first-week mortality proportion was 6.9{\%} (95{\%} CI 6.3, 7.7) and was lower in programmes operating in countries with policies that allowed ETOPFA compared with their counterparts (5.9{\%} vs. 8.4{\%}). The majority of first-week mortality occurred on the first day of life. In programmes where information on long-term mortality was available through linkage to administrative databases, survival at 5 years of age was 90{\%}-96{\%} in Europe, and 86{\%}-96{\%} in North America.CONCLUSIONS: Our multi-country study showed a high proportion of stillbirth and infant and child deaths among those with spina bifida. Effective folic acid interventions could prevent many cases of spina bifida, thereby preventing associated childhood morbidity and mortality.",
author = "Bakker, {Marian K} and Vijaya Kancherla and Canfield, {Mark A} and Eva Bermejo-Sanchez and Cragan, {Janet D} and Saeed Dastgiri and {De Walle}, {Hermien E K} and Feldkamp, {Marcia L} and Boris Groisman and Miriam Gatt and Paula Hurtado-Villa and Karin Kallen and Daniella Landau and Nathalie Lelong and {Lopez Camelo}, {Jorge S} and Laura Mart{\'i}nez and Margery Morgan and Mutchinick, {Osvaldo M} and Nembhard, {Wendy N} and Anna Pierini and Anke Rissmann and Antonin Sipek and Elena Szabova and Giovanna Tagliabue and Wladimir Wertelecki and Ignacio Zarante and Pierpaolo Mastroiacovo",
note = "{\circledC} 2019 The Authors. Paediatric and Perinatal Epidemiology published by John Wiley & Sons Ltd.",
year = "2019",
month = "11",
doi = "10.1111/ppe.12589",
language = "English",
volume = "33",
pages = "436--448",
journal = "Paediatric and Perinatal Epidemiology",
issn = "0269-5022",
publisher = "Wiley",
number = "6",

}

RIS

TY - JOUR

T1 - Analysis of Mortality among Neonates and Children with Spina Bifida

T2 - An International Registry-Based Study, 2001-2012

AU - Bakker, Marian K

AU - Kancherla, Vijaya

AU - Canfield, Mark A

AU - Bermejo-Sanchez, Eva

AU - Cragan, Janet D

AU - Dastgiri, Saeed

AU - De Walle, Hermien E K

AU - Feldkamp, Marcia L

AU - Groisman, Boris

AU - Gatt, Miriam

AU - Hurtado-Villa, Paula

AU - Kallen, Karin

AU - Landau, Daniella

AU - Lelong, Nathalie

AU - Lopez Camelo, Jorge S

AU - Martínez, Laura

AU - Morgan, Margery

AU - Mutchinick, Osvaldo M

AU - Nembhard, Wendy N

AU - Pierini, Anna

AU - Rissmann, Anke

AU - Sipek, Antonin

AU - Szabova, Elena

AU - Tagliabue, Giovanna

AU - Wertelecki, Wladimir

AU - Zarante, Ignacio

AU - Mastroiacovo, Pierpaolo

N1 - © 2019 The Authors. Paediatric and Perinatal Epidemiology published by John Wiley & Sons Ltd.

PY - 2019/11

Y1 - 2019/11

N2 - BACKGROUND: Medical advancements have resulted in better survival and life expectancy among those with spina bifida, but a significantly increased risk of perinatal and postnatal mortality for individuals with spina bifida remains.OBJECTIVES: To examine stillbirth and infant and child mortality among those affected by spina bifida using data from multiple countries.METHODS: We conducted an observational study, using data from 24 population- and hospital-based surveillance registries in 18 countries contributing as members of the International Clearinghouse for Birth Defects Surveillance and Research (ICBDSR). Cases of spina bifida that resulted in livebirths or stillbirths from 20 weeks' gestation or elective termination of pregnancy for fetal anomaly (ETOPFA) were included. Among liveborn spina bifida cases, we calculated mortality at different ages as number of deaths among liveborn cases divided by total number of liveborn cases with spina bifida. As a secondary outcome measure, we estimated the prevalence of spina bifida per 10 000 total births. The 95% confidence interval for the prevalence estimate was estimated using the Poisson approximation of binomial distribution.RESULTS: Between years 2001 and 2012, the overall first-week mortality proportion was 6.9% (95% CI 6.3, 7.7) and was lower in programmes operating in countries with policies that allowed ETOPFA compared with their counterparts (5.9% vs. 8.4%). The majority of first-week mortality occurred on the first day of life. In programmes where information on long-term mortality was available through linkage to administrative databases, survival at 5 years of age was 90%-96% in Europe, and 86%-96% in North America.CONCLUSIONS: Our multi-country study showed a high proportion of stillbirth and infant and child deaths among those with spina bifida. Effective folic acid interventions could prevent many cases of spina bifida, thereby preventing associated childhood morbidity and mortality.

AB - BACKGROUND: Medical advancements have resulted in better survival and life expectancy among those with spina bifida, but a significantly increased risk of perinatal and postnatal mortality for individuals with spina bifida remains.OBJECTIVES: To examine stillbirth and infant and child mortality among those affected by spina bifida using data from multiple countries.METHODS: We conducted an observational study, using data from 24 population- and hospital-based surveillance registries in 18 countries contributing as members of the International Clearinghouse for Birth Defects Surveillance and Research (ICBDSR). Cases of spina bifida that resulted in livebirths or stillbirths from 20 weeks' gestation or elective termination of pregnancy for fetal anomaly (ETOPFA) were included. Among liveborn spina bifida cases, we calculated mortality at different ages as number of deaths among liveborn cases divided by total number of liveborn cases with spina bifida. As a secondary outcome measure, we estimated the prevalence of spina bifida per 10 000 total births. The 95% confidence interval for the prevalence estimate was estimated using the Poisson approximation of binomial distribution.RESULTS: Between years 2001 and 2012, the overall first-week mortality proportion was 6.9% (95% CI 6.3, 7.7) and was lower in programmes operating in countries with policies that allowed ETOPFA compared with their counterparts (5.9% vs. 8.4%). The majority of first-week mortality occurred on the first day of life. In programmes where information on long-term mortality was available through linkage to administrative databases, survival at 5 years of age was 90%-96% in Europe, and 86%-96% in North America.CONCLUSIONS: Our multi-country study showed a high proportion of stillbirth and infant and child deaths among those with spina bifida. Effective folic acid interventions could prevent many cases of spina bifida, thereby preventing associated childhood morbidity and mortality.

U2 - 10.1111/ppe.12589

DO - 10.1111/ppe.12589

M3 - Article

C2 - 31637749

VL - 33

SP - 436

EP - 448

JO - Paediatric and Perinatal Epidemiology

JF - Paediatric and Perinatal Epidemiology

SN - 0269-5022

IS - 6

ER -

ID: 101119182