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Survival, hospitalisation and surgery in children born with Pierre Robin sequence: a European population-based cohort study

Michele Santoro Orcid Logo, Ester Garne, Alessio Coi, Joachim Tan, Maria Loane, Elisa Ballardini, Clara Cavero-Carbonell, Hermien EK de Walle, Miriam Gatt, Mika Gissler, Sue Jordan Orcid Logo, Kari Klungsøyr, Natalie Lelong, Stine Kjaer Urhoj Orcid Logo, Diana G Wellesley, Joan K Morris Orcid Logo

Archives of Disease in Childhood, Pages: archdischild - 2022

Swansea University Author: Sue Jordan Orcid Logo

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DOI (Published version): 10.1136/archdischild-2022-324716

Abstract

Objective To evaluate survival, hospitalisations and surgical procedures for children born with Pierre Robin sequence (PRS) across Europe.Design Multicentre population-based cohort study.Setting Data on 463 live births with PRS from a population of 4 984 793 from 12 EUROCAT congenital anomaly regist...

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Published in: Archives of Disease in Childhood
ISSN: 0003-9888 1468-2044
Published: BMJ 2023
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Each registry applied a common data model to standardise the linked data and ran common syntax scripts to produce aggregate tables. Results from each registry were pooled using random-effect meta-analyses.Main outcome measures Probability of survival, proportion of children hospitalised and undergoing surgery, and median length of hospital stay.Results The majority of deaths occurred in the first year of life with a survival rate of 96.0% (95% CI 93.5% to 98.5%); 95.1% (95% CI 92.7% to 97.7%) survived to age 10. In the first year of life, 99.2% (95% CI 95.0% to 99.9%) of children were hospitalised with a median stay of 21.4 days (95% CI 15.6 to 27.2), and 67.6% (95% CI 46.6% to 81.8%) underwent surgery. In the first 5 years of life, 99.2% of children underwent a median of two surgical procedures. Between ages 5 and 9, 58.3% (95% CI 44.7% to 69.7%) were hospitalised with a median annual stay of 0.3 days.Conclusions Children with PRS had high mortality and morbidity with long hospital stays in the first year of life, and almost all had surgery before 5 years of age. Survival improved after infancy with fewer hospitalisations after age 5. 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Re-use permitted under CC BY-NC. No commercial re-use. See rights and permissions. Published by BMJ. http://creativecommons.org/licenses/by-nc/4.0/ This is an open access article distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited, appropriate credit is given, any changes made indicated, and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/.</documentNotes><copyrightCorrect>true</copyrightCorrect><language>eng</language><licence>http://creativecommons.org/licenses/by-nc/4.0/</licence></document></documents><OutputDurs/></rfc1807>
spelling v2 63449 2023-05-12 Survival, hospitalisation and surgery in children born with Pierre Robin sequence: a European population-based cohort study 24ce9db29b4bde1af4e83b388aae0ea1 0000-0002-5691-2987 Sue Jordan Sue Jordan true false 2023-05-12 HNU Objective To evaluate survival, hospitalisations and surgical procedures for children born with Pierre Robin sequence (PRS) across Europe.Design Multicentre population-based cohort study.Setting Data on 463 live births with PRS from a population of 4 984 793 from 12 EUROCAT congenital anomaly registries.Methods Data on children with PRS born 1995–2014 were linked electronically to data on mortality, hospitalisations and surgical procedures up to 10 years of age. Each registry applied a common data model to standardise the linked data and ran common syntax scripts to produce aggregate tables. Results from each registry were pooled using random-effect meta-analyses.Main outcome measures Probability of survival, proportion of children hospitalised and undergoing surgery, and median length of hospital stay.Results The majority of deaths occurred in the first year of life with a survival rate of 96.0% (95% CI 93.5% to 98.5%); 95.1% (95% CI 92.7% to 97.7%) survived to age 10. In the first year of life, 99.2% (95% CI 95.0% to 99.9%) of children were hospitalised with a median stay of 21.4 days (95% CI 15.6 to 27.2), and 67.6% (95% CI 46.6% to 81.8%) underwent surgery. In the first 5 years of life, 99.2% of children underwent a median of two surgical procedures. Between ages 5 and 9, 58.3% (95% CI 44.7% to 69.7%) were hospitalised with a median annual stay of 0.3 days.Conclusions Children with PRS had high mortality and morbidity with long hospital stays in the first year of life, and almost all had surgery before 5 years of age. Survival improved after infancy with fewer hospitalisations after age 5. This study provides reliable estimates of the survival and morbidity of children with PRS for families and healthcare providers. Journal Article Archives of Disease in Childhood archdischild 2022 BMJ 0003-9888 1468-2044 9 5 2023 2023-05-09 10.1136/archdischild-2022-324716 http://dx.doi.org/10.1136/archdischild-2022-324716 COLLEGE NANME Nursing COLLEGE CODE HNU Swansea University Another institution paid the OA fee This project received funding from the European Union’s Horizon 2020 Research and Innovation programme (under grant agreement number 733001, January 2017–December 2021; https://ec.europa.eu/programmes/horizon2020/en). 2023-06-07T11:43:01.6677002 2023-05-12T14:41:20.8344955 Faculty of Medicine, Health and Life Sciences School of Health and Social Care - Nursing Michele Santoro 0000-0003-0676-3036 1 Ester Garne 2 Alessio Coi 3 Joachim Tan 4 Maria Loane 5 Elisa Ballardini 6 Clara Cavero-Carbonell 7 Hermien EK de Walle 8 Miriam Gatt 9 Mika Gissler 10 Sue Jordan 0000-0002-5691-2987 11 Kari Klungsøyr 12 Natalie Lelong 13 Stine Kjaer Urhoj 0000-0002-2069-9723 14 Diana G Wellesley 15 Joan K Morris 0000-0002-7164-612x 16 63449__27740__4f4606c1b8c14461923132ad8a7519a8.pdf 63449.pdf 2023-06-07T11:40:36.3898511 Output 429560 application/pdf Version of Record true © Author(s) (or their employer(s)) 2023. Re-use permitted under CC BY-NC. No commercial re-use. See rights and permissions. Published by BMJ. http://creativecommons.org/licenses/by-nc/4.0/ This is an open access article distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited, appropriate credit is given, any changes made indicated, and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/. true eng http://creativecommons.org/licenses/by-nc/4.0/
title Survival, hospitalisation and surgery in children born with Pierre Robin sequence: a European population-based cohort study
spellingShingle Survival, hospitalisation and surgery in children born with Pierre Robin sequence: a European population-based cohort study
Sue Jordan
title_short Survival, hospitalisation and surgery in children born with Pierre Robin sequence: a European population-based cohort study
title_full Survival, hospitalisation and surgery in children born with Pierre Robin sequence: a European population-based cohort study
title_fullStr Survival, hospitalisation and surgery in children born with Pierre Robin sequence: a European population-based cohort study
title_full_unstemmed Survival, hospitalisation and surgery in children born with Pierre Robin sequence: a European population-based cohort study
title_sort Survival, hospitalisation and surgery in children born with Pierre Robin sequence: a European population-based cohort study
author_id_str_mv 24ce9db29b4bde1af4e83b388aae0ea1
author_id_fullname_str_mv 24ce9db29b4bde1af4e83b388aae0ea1_***_Sue Jordan
author Sue Jordan
author2 Michele Santoro
Ester Garne
Alessio Coi
Joachim Tan
Maria Loane
Elisa Ballardini
Clara Cavero-Carbonell
Hermien EK de Walle
Miriam Gatt
Mika Gissler
Sue Jordan
Kari Klungsøyr
Natalie Lelong
Stine Kjaer Urhoj
Diana G Wellesley
Joan K Morris
format Journal article
container_title Archives of Disease in Childhood
container_start_page archdischild
publishDate 2023
institution Swansea University
issn 0003-9888
1468-2044
doi_str_mv 10.1136/archdischild-2022-324716
publisher BMJ
college_str Faculty of Medicine, Health and Life Sciences
hierarchytype
hierarchy_top_id facultyofmedicinehealthandlifesciences
hierarchy_top_title Faculty of Medicine, Health and Life Sciences
hierarchy_parent_id facultyofmedicinehealthandlifesciences
hierarchy_parent_title Faculty of Medicine, Health and Life Sciences
department_str School of Health and Social Care - Nursing{{{_:::_}}}Faculty of Medicine, Health and Life Sciences{{{_:::_}}}School of Health and Social Care - Nursing
url http://dx.doi.org/10.1136/archdischild-2022-324716
document_store_str 1
active_str 0
description Objective To evaluate survival, hospitalisations and surgical procedures for children born with Pierre Robin sequence (PRS) across Europe.Design Multicentre population-based cohort study.Setting Data on 463 live births with PRS from a population of 4 984 793 from 12 EUROCAT congenital anomaly registries.Methods Data on children with PRS born 1995–2014 were linked electronically to data on mortality, hospitalisations and surgical procedures up to 10 years of age. Each registry applied a common data model to standardise the linked data and ran common syntax scripts to produce aggregate tables. Results from each registry were pooled using random-effect meta-analyses.Main outcome measures Probability of survival, proportion of children hospitalised and undergoing surgery, and median length of hospital stay.Results The majority of deaths occurred in the first year of life with a survival rate of 96.0% (95% CI 93.5% to 98.5%); 95.1% (95% CI 92.7% to 97.7%) survived to age 10. In the first year of life, 99.2% (95% CI 95.0% to 99.9%) of children were hospitalised with a median stay of 21.4 days (95% CI 15.6 to 27.2), and 67.6% (95% CI 46.6% to 81.8%) underwent surgery. In the first 5 years of life, 99.2% of children underwent a median of two surgical procedures. Between ages 5 and 9, 58.3% (95% CI 44.7% to 69.7%) were hospitalised with a median annual stay of 0.3 days.Conclusions Children with PRS had high mortality and morbidity with long hospital stays in the first year of life, and almost all had surgery before 5 years of age. Survival improved after infancy with fewer hospitalisations after age 5. This study provides reliable estimates of the survival and morbidity of children with PRS for families and healthcare providers.
published_date 2023-05-09T11:43:00Z
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