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Hospitalizations from Birth to 28 Years in a Population Cohort of Individuals Born with Five Rare Craniofacial Anomalies in Western Australia - 22/08/23

Doi : 10.1016/j.jpeds.2023.113418 
Mohammed Junaid, MDS, MFDS RCPS (Glasg.), DDPH RCS (Eng.), PhD 1, 2, , Linda Slack-Smith, GradDipMedTech, MSc, PhD 1, 2, Kingsley Wong, MBBS, MPH, MMedStat 2, Timothy Hewitt, MBBS (Hons.), FRACS 3, Emma Glasson, BPsych, BSc (Hons), PhD 2, Jenny Bourke, MPH 2, Gareth Baynam, FRACP, PhD 2, 4, Hanny Calache, BDSc, MDSc, DPH 5, 6, Helen Leonard, MBChB, MPH 2, 7
1 School of Population and Global Health, The University of Western Australia, Nedlands, Western Australia, Australia 
2 Telethon Kids Institute, The University of Western Australia, Northern Entrance, Nedlands, Western Australia, Australia 
3 Department of Plastic and Reconstructive Surgery, Perth Children’s Hospital, Nedlands, Western Australia, Australia 
4 Western Australian Register of Developmental Anomalies, Department of Health, Government of Western Australia, Perth, Western Australia, Australia 
5 Deakin Health Economics, Institute of Health Transformation, School of Health and Social Development, Faculty of Health, Deakin University, Geelong, Victoria, Australia 
6 Department of Dentistry and Oral Health, La Trobe Rural Health School, La Trobe University, Bendigo, Victoria, Australia 
7 UWA Centre of Child Health Research, University of Western Australia, Nedlands, Western Australia, Australia 

Reprint requests: Mohammed Junaid, MDS, MFDS RCPS (Glasg.), DDPH RCS (Eng.), PhD, School of Population and Global Health, The University of Western Australia, Clifton Street Building, Clifton St, Nedlands 6009, Western Australia, Australia.School of Population and Global HealthThe University of Western AustraliaClifton Street Building, Clifton StNedlandsWestern Australia6009Australia

Abstract

Objective

To describe trends, age-specific patterns, and factors influencing hospitalizations for 5 rare craniofacial anomalies (CFAs).

Methods

Data on livebirths (1983-2010; n = 721 019) including rare CFA (craniofacial microsomia, mandibulofacial dysostosis, Pierre Robin sequence, Van der Woude syndrome, and frontonasal dysplasia), episodes of death, and demographic and perinatal factors were identified from the Western Australian Register of Developmental Anomalies, Death Registrations and Midwives Notification System. Information on incident craniofacial and noncraniofacial related admissions, length of hospital stay, and intensive care and emergency-related admissions were identified using principal diagnosis and procedural codes were extracted from the Hospital Morbidity Data Collection and linked to other data sources. Associations of hospitalizations by age groups as well as demographic and perinatal factors were expressed as incidence rate ratio (IRR).

Results

The incident hospitalizations were 3 times as high for rare CFA (IRR 3.22-3.72) throughout childhood into adolescence than those without. Children with rare CFA had 3-4 times as many potentially preventable hospitalizations until 18 years of age than those without. Specifically, respiratory infections (IRR 2.13-2.35), ear infections (IRR 7.92-26.28), and oral health–related conditions contributed for most noncraniofacial admissions until the adolescence period. A greater incidence of noncraniofacial related hospitalizations was observed among Indigenous children, births with intrauterine growth restrictions, and families with high socioeconomic disadvantage.

Conclusions

Throughout childhood, individuals with rare CFA had greater hospital service use, specifically for potentially preventable conditions, than those without. These population-level findings can inform new preventive strategies and early disease management targeted toward reducing preventable hospitalizations.

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Keywords : hospital admissions, craniofacial microsomia, Pierre Robin sequence, epidemiology

Abbreviations : APC, CFA, CFM, cLOS, ED, HMDC, ICU, IRR, MNS, PRS, TCS, WARDA-BD


Plan


 MJ is supported by the Australian Government International Research Training Program Scholarship and the Stan and Jean Perron Top-Up Scholarship. HL is funded by National Health and Medical Research Council (NHMRC) Senior Research Fellowship APP1117105. The authors have no conflicts of interest to disclose.


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Vol 259

Article 113418- août 2023 Retour au numéro
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