Hospital length of stay and surgery among European children with rare structural congenital anomalies – A population-based data linkage study

Ester Garne; Joachim Tan; Mads Damkjaer; Elisa Ballardini; Clara Cavero-Carbonell; Alessio Coi; Laura Garcia-Villodre; Mika  Gissler; Joanne Given; Anna  Heino; Sue Jordan; Elizabeth Limb; Maria Loane; Amanda J. Neville; Anna Pierini; Anke Rissmann; David Tucker; Stine Kjaer  Urhoj; Joan Morris

doi:10.3390/ijerph20054387

Hospital length of stay and surgery among European children with rare structural congenital anomalies – A population-based data linkage study

Ester Garne, Joachim Tan, Mads Damkjaer, Elisa Ballardini, Clara Cavero-Carbonell, Alessio Coi, Laura Garcia-Villodre, Mika Gissler, Joanne Given, Anna Heino, Sue Jordan, Elizabeth Limb, Maria Loane, Amanda J. Neville, Anna Pierini, Anke Rissmann, David Tucker, Stine Kjaer Urhoj, Joan Morris

Research output: Contribution to journal › Article › peer-review

4 Citations (Scopus)

35 Downloads (Pure)

Abstract

Little is known about morbidity for children with rare structural congenital anomalies. This European, population-based data-linkage cohort study analysed data on hospitalisations and surgical procedures for 5948 children born 1995–2014 with 18 rare structural congenital anomalies from nine EUROCAT registries in five countries. In the first year of life, the median length of stay (LOS) ranged from 3.5 days (anotia) to 53.8 days (atresia of bile ducts). Generally, children with gastrointestinal anomalies, bladder anomalies and Prune-Belly had the longest LOS. At ages 1–4, the median LOS per year was ≤3 days for most anomalies. The proportion of children having surgery before age 5 years ranged from 40% to 100%. The median number of surgical procedures for those under 5 years was two or more for 14 of the 18 anomalies and the highest for children with Prune-Belly at 7.4 (95% CI 2.5–12.3). The median age at first surgery for children with atresia of bile ducts was 8.4 weeks (95% CI 7.6–9.2) which is older than international recommendations. Results from the subset of registries with data up to 10 years of age showed that the need for hospitalisations and surgery continued. The burden of disease in early childhood is high for children with rare structural congenital anomalies.

Original language	English
Article number	4387
Pages (from-to)	1-11
Number of pages	11
Journal	International Journal of Environmental Research and Public Health
Volume	20
Issue number	5
Early online date	1 Mar 2023
DOIs	https://doi.org/10.3390/ijerph20054387
Publication status	Published online - 1 Mar 2023

Bibliographical note

Funding Information:
This project has received funding from the European Union’s Horizon 2020 research and innovation programme under grant agreement No. 733001. The funders had no role in the design and conduct of the study; the collection, management, analysis, and interpretation of the data; the preparation, review, or approval of the manuscript; and the decision to submit the manuscript for publication.

Publisher Copyright:
© 2023 by the authors.

Funding

Funding Information: This project has received funding from the European Union’s Horizon 2020 research and innovation programme under grant agreement No. 733001. The funders had no role in the design and conduct of the study; the collection, management, analysis, and interpretation of the data; the preparation, review, or approval of the manuscript; and the decision to submit the manuscript for publication.

Keywords

rare congenital anomalies
morbidity
length of stay
surgery
Health, Toxicology and Mutagenesis
Public Health, Environmental and Occupational Health
Length of Stay
Parturition
Humans
Information Storage and Retrieval
Child, Preschool
Infant
Pregnancy
Congenital Abnormalities
Hospitals
Female
Registries
Child
Cohort Studies
Article

UN SDGs

This output contributes to the following UN Sustainable Development Goals (SDGs)

Access to Document

10.3390/ijerph20054387Licence: CC BY

ijerph-20-04387Final published version, 317 KBLicence: CC BY

Cite this

Garne, E., Tan, J., Damkjaer, M., Ballardini, E., Cavero-Carbonell, C., Coi, A., Garcia-Villodre, L., Gissler, M., Given, J., Heino, A., Jordan, S., Limb, E., Loane, M., Neville, A. J., Pierini, A., Rissmann, A., Tucker, D., Urhoj, S. K., & Morris, J. (2023). Hospital length of stay and surgery among European children with rare structural congenital anomalies – A population-based data linkage study. International Journal of Environmental Research and Public Health, 20(5), 1-11. Article 4387. Advance online publication. https://doi.org/10.3390/ijerph20054387

@article{9748e8e9c2e640ac8d3f6fb491715315,

title = "Hospital length of stay and surgery among European children with rare structural congenital anomalies – A population-based data linkage study",

abstract = "Little is known about morbidity for children with rare structural congenital anomalies. This European, population-based data-linkage cohort study analysed data on hospitalisations and surgical procedures for 5948 children born 1995–2014 with 18 rare structural congenital anomalies from nine EUROCAT registries in five countries. In the first year of life, the median length of stay (LOS) ranged from 3.5 days (anotia) to 53.8 days (atresia of bile ducts). Generally, children with gastrointestinal anomalies, bladder anomalies and Prune-Belly had the longest LOS. At ages 1–4, the median LOS per year was ≤3 days for most anomalies. The proportion of children having surgery before age 5 years ranged from 40\% to 100\%. The median number of surgical procedures for those under 5 years was two or more for 14 of the 18 anomalies and the highest for children with Prune-Belly at 7.4 (95\% CI 2.5–12.3). The median age at first surgery for children with atresia of bile ducts was 8.4 weeks (95\% CI 7.6–9.2) which is older than international recommendations. Results from the subset of registries with data up to 10 years of age showed that the need for hospitalisations and surgery continued. The burden of disease in early childhood is high for children with rare structural congenital anomalies.",

keywords = "rare congenital anomalies, morbidity, length of stay, surgery, Health, Toxicology and Mutagenesis, Public Health, Environmental and Occupational Health, Length of Stay, Parturition, Humans, Information Storage and Retrieval, Child, Preschool, Infant, Pregnancy, Congenital Abnormalities, Hospitals, Female, Registries, Child, Cohort Studies, Article",

author = "Ester Garne and Joachim Tan and Mads Damkjaer and Elisa Ballardini and Clara Cavero-Carbonell and Alessio Coi and Laura Garcia-Villodre and Mika Gissler and Joanne Given and Anna Heino and Sue Jordan and Elizabeth Limb and Maria Loane and Neville, \{Amanda J.\} and Anna Pierini and Anke Rissmann and David Tucker and Urhoj, \{Stine Kjaer\} and Joan Morris",

note = "Funding Information: This project has received funding from the European Union{\textquoteright}s Horizon 2020 research and innovation programme under grant agreement No. 733001. The funders had no role in the design and conduct of the study; the collection, management, analysis, and interpretation of the data; the preparation, review, or approval of the manuscript; and the decision to submit the manuscript for publication. Publisher Copyright: {\textcopyright} 2023 by the authors.",

year = "2023",

month = mar,

day = "1",

doi = "10.3390/ijerph20054387",

language = "English",

volume = "20",

pages = "1--11",

journal = "International Journal of Environmental Research and Public Health",

issn = "1660-4601",

publisher = "MDPI",

number = "5",

}

Garne, E, Tan, J, Damkjaer, M, Ballardini, E, Cavero-Carbonell, C, Coi, A, Garcia-Villodre, L, Gissler, M, Given, J, Heino, A, Jordan, S, Limb, E, Loane, M, Neville, AJ, Pierini, A, Rissmann, A, Tucker, D, Urhoj, SK & Morris, J 2023, 'Hospital length of stay and surgery among European children with rare structural congenital anomalies – A population-based data linkage study', International Journal of Environmental Research and Public Health, vol. 20, no. 5, 4387, pp. 1-11. https://doi.org/10.3390/ijerph20054387

Hospital length of stay and surgery among European children with rare structural congenital anomalies – A population-based data linkage study. / Garne, Ester; Tan, Joachim; Damkjaer, Mads et al.
In: International Journal of Environmental Research and Public Health, Vol. 20, No. 5, 4387, 01.03.2023, p. 1-11.

Research output: Contribution to journal › Article › peer-review

TY - JOUR

T1 - Hospital length of stay and surgery among European children with rare structural congenital anomalies – A population-based data linkage study

AU - Garne, Ester

AU - Tan, Joachim

AU - Damkjaer, Mads

AU - Ballardini, Elisa

AU - Cavero-Carbonell, Clara

AU - Coi, Alessio

AU - Garcia-Villodre, Laura

AU - Gissler, Mika

AU - Given, Joanne

AU - Heino, Anna

AU - Jordan, Sue

AU - Limb, Elizabeth

AU - Loane, Maria

AU - Neville, Amanda J.

AU - Pierini, Anna

AU - Rissmann, Anke

AU - Tucker, David

AU - Urhoj, Stine Kjaer

AU - Morris, Joan

N1 - Funding Information: This project has received funding from the European Union’s Horizon 2020 research and innovation programme under grant agreement No. 733001. The funders had no role in the design and conduct of the study; the collection, management, analysis, and interpretation of the data; the preparation, review, or approval of the manuscript; and the decision to submit the manuscript for publication. Publisher Copyright: © 2023 by the authors.

PY - 2023/3/1

Y1 - 2023/3/1

N2 - Little is known about morbidity for children with rare structural congenital anomalies. This European, population-based data-linkage cohort study analysed data on hospitalisations and surgical procedures for 5948 children born 1995–2014 with 18 rare structural congenital anomalies from nine EUROCAT registries in five countries. In the first year of life, the median length of stay (LOS) ranged from 3.5 days (anotia) to 53.8 days (atresia of bile ducts). Generally, children with gastrointestinal anomalies, bladder anomalies and Prune-Belly had the longest LOS. At ages 1–4, the median LOS per year was ≤3 days for most anomalies. The proportion of children having surgery before age 5 years ranged from 40% to 100%. The median number of surgical procedures for those under 5 years was two or more for 14 of the 18 anomalies and the highest for children with Prune-Belly at 7.4 (95% CI 2.5–12.3). The median age at first surgery for children with atresia of bile ducts was 8.4 weeks (95% CI 7.6–9.2) which is older than international recommendations. Results from the subset of registries with data up to 10 years of age showed that the need for hospitalisations and surgery continued. The burden of disease in early childhood is high for children with rare structural congenital anomalies.

AB - Little is known about morbidity for children with rare structural congenital anomalies. This European, population-based data-linkage cohort study analysed data on hospitalisations and surgical procedures for 5948 children born 1995–2014 with 18 rare structural congenital anomalies from nine EUROCAT registries in five countries. In the first year of life, the median length of stay (LOS) ranged from 3.5 days (anotia) to 53.8 days (atresia of bile ducts). Generally, children with gastrointestinal anomalies, bladder anomalies and Prune-Belly had the longest LOS. At ages 1–4, the median LOS per year was ≤3 days for most anomalies. The proportion of children having surgery before age 5 years ranged from 40% to 100%. The median number of surgical procedures for those under 5 years was two or more for 14 of the 18 anomalies and the highest for children with Prune-Belly at 7.4 (95% CI 2.5–12.3). The median age at first surgery for children with atresia of bile ducts was 8.4 weeks (95% CI 7.6–9.2) which is older than international recommendations. Results from the subset of registries with data up to 10 years of age showed that the need for hospitalisations and surgery continued. The burden of disease in early childhood is high for children with rare structural congenital anomalies.

KW - rare congenital anomalies

KW - morbidity

KW - length of stay

KW - surgery

KW - Health, Toxicology and Mutagenesis

KW - Public Health, Environmental and Occupational Health

KW - Length of Stay

KW - Parturition

KW - Humans

KW - Information Storage and Retrieval

KW - Child, Preschool

KW - Infant

KW - Pregnancy

KW - Congenital Abnormalities

KW - Hospitals

KW - Female

KW - Registries

KW - Child

KW - Cohort Studies

KW - Article

UR - https://pure.ulster.ac.uk/en/publications/9748e8e9-c2e6-40ac-8d3f-6fb491715315

UR - https://www.scopus.com/pages/publications/85149912958

U2 - 10.3390/ijerph20054387

DO - 10.3390/ijerph20054387

M3 - Article

C2 - 36901396

SN - 1660-4601

VL - 20

SP - 1

EP - 11

JO - International Journal of Environmental Research and Public Health

JF - International Journal of Environmental Research and Public Health

IS - 5

M1 - 4387

ER -

Hospital length of stay and surgery among European children with rare structural congenital anomalies – A population-based data linkage study

Abstract

Bibliographical note

Funding

Keywords

UN SDGs

Access to Document

Other files and links

Fingerprint

Cite this