TY - JOUR
T1 - Elevated Infant Mortality Rate among Dutch Oral Cleft Cases
T2 - A Retrospective Analysis from 1997 to 2011
AU - van Nunen, Daan P.F.
AU - van den Boogaard, Marie José H.
AU - Don Griot, J. Peter W.
AU - Rüttermann, Mike
AU - van der Veken, Lars T.
AU - Breugem, Corstiaan C.
N1 - Funding Information: We would like to thank Dr. Christl Vermeij-Keers of the NVSCA for the provision of data from the NVSCA cleft registry. Publisher Copyright: © Copyright © 2014 van Nunen, van den Boogaard, Don Griot, Rüttermann, van der Veken and Breugem. Copyright: Copyright 2020 Elsevier B.V., All rights reserved.
PY - 2014/12/4
Y1 - 2014/12/4
N2 - Objectives: First, to determine the infant mortality rate (IMR) for Dutch patients with isolated oral clefts (OC) as well as for patients with clefts seen in association with other malformations. Second, to conduct a similar analysis per cleft type: cleft lip with or without cleft palate (CP), CP (including Robin sequence). Third, to examine the underlying causes of death. Material and Methods: A retrospective review of the charts of patients with OC born in the period 1997–2011 and treated in three regional cleft centers in the Netherlands. Results: One thousand five hundred thirty patients with OC were born during the study period and treated in the cleft centers. The overall IMR for all clefts was 2.09%, significantly higher than the general Dutch IMR of 0.45%. In a subanalysis per cleft type, the IMRs were 1.22, 1.38, 2.45, and 3.62% for cleft lip, cleft lip with CP, CP, and Robin sequence, respectively. The mortality rates for isolated OC did not differ significantly from the general Dutch rate. Causes of death were congenital malformations of the heart in 40.6%, airway/lungs in 15.6%, nervous system in 15.6%, infectious disease in 12.5%, and other or unknown in 15.6%. Conclusion: The elevated IMR observed in Dutch patients with OC is almost exclusively caused by associated congenital malformations. After diagnosis of an oral cleft an in-depth medical examination and a consult by the pediatrician and clinical geneticist is imperative to instigate the appropriate medical management.
AB - Objectives: First, to determine the infant mortality rate (IMR) for Dutch patients with isolated oral clefts (OC) as well as for patients with clefts seen in association with other malformations. Second, to conduct a similar analysis per cleft type: cleft lip with or without cleft palate (CP), CP (including Robin sequence). Third, to examine the underlying causes of death. Material and Methods: A retrospective review of the charts of patients with OC born in the period 1997–2011 and treated in three regional cleft centers in the Netherlands. Results: One thousand five hundred thirty patients with OC were born during the study period and treated in the cleft centers. The overall IMR for all clefts was 2.09%, significantly higher than the general Dutch IMR of 0.45%. In a subanalysis per cleft type, the IMRs were 1.22, 1.38, 2.45, and 3.62% for cleft lip, cleft lip with CP, CP, and Robin sequence, respectively. The mortality rates for isolated OC did not differ significantly from the general Dutch rate. Causes of death were congenital malformations of the heart in 40.6%, airway/lungs in 15.6%, nervous system in 15.6%, infectious disease in 12.5%, and other or unknown in 15.6%. Conclusion: The elevated IMR observed in Dutch patients with OC is almost exclusively caused by associated congenital malformations. After diagnosis of an oral cleft an in-depth medical examination and a consult by the pediatrician and clinical geneticist is imperative to instigate the appropriate medical management.
KW - Netherlands
KW - Pierre Robin syndrome
KW - Robin sequence
KW - cleft lip
KW - cleft palate
KW - epidemiology
KW - infant mortality
UR - http://www.scopus.com/inward/record.url?scp=84924035277&partnerID=8YFLogxK
U2 - https://doi.org/10.3389/fsurg.2014.00048
DO - https://doi.org/10.3389/fsurg.2014.00048
M3 - Article
SN - 2296-875X
VL - 1
JO - Frontiers in Surgery
JF - Frontiers in Surgery
M1 - 48
ER -