Elevated infant mortality rate among Dutch oral cleft cases: a retrospective analysis from 1997 to 2011

被引:18
作者
van Nunen, Daan P. F. [1 ]
van den Boogaard, Marie-Jose H. [2 ]
Griot, J. Peter W. Don [3 ]
Ruttermann, Mike [4 ]
van der Veken, Lars T. [2 ]
Breugem, Corstiaan C. [1 ]
机构
[1] Univ Med Ctr Utrecht, Div Plast & Reconstruct Surg, Utrecht, Netherlands
[2] Univ Med Ctr Utrecht, Dept Med Genet, Utrecht, Netherlands
[3] Vrije Univ, Univ Med Ctr Amsterdam, Dept Plast Reconstruct & Hand Surg, Amsterdam, Netherlands
[4] Univ Med Ctr Groningen, Dept Plast Surg, Groningen, Netherlands
关键词
cleft lip; cleft palate; Robin sequence; Pierre Robin syndrome; infant mortality; epidemiology; Netherlands;
D O I
10.3389/fsurg.2014.00048
中图分类号
R61 [外科手术学];
学科分类号
摘要
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.
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相关论文
共 19 条
[1]   THE PIERRE-ROBIN-SEQUENCE - REVIEW OF 125 CASES AND EVOLUTION OF TREATMENT MODALITIES [J].
CAOUETTELABERGE, L ;
BAYET, B ;
LAROCQUE, Y .
PLASTIC AND RECONSTRUCTIVE SURGERY, 1994, 93 (05) :934-942
[2]   Elevated Infant Mortality Rates Among Oral Cleft and Isolated Oral Cleft Cases: A Meta-Analysis of Studies From 1943 to 2010 [J].
Carlson, Lucas ;
Hatcher, Kristin Ward ;
Vander Burg, Richard .
CLEFT PALATE CRANIOFACIAL JOURNAL, 2013, 50 (01) :2-12
[3]  
Druschel CM, 1996, CLEFT PALATE-CRAN J, V33, P400, DOI 10.1597/1545-1569(1996)033<0400:FYOLMA>2.3.CO
[4]  
2
[5]   FURTHER EPIDEMIOLOGICAL DIFFERENTIATION OF CLEFT-LIP AND PALATE - POPULATION STUDY OF CLEFTS IN KING-COUNTY, WASHINGTON, 1956-1965 [J].
EMANUEL, I ;
CULVER, BH ;
ERICKSON, JD ;
GUTHRIE, B ;
SCHULDBERG, D .
TERATOLOGY, 1973, 7 (03) :271-281
[6]  
Eurocat Nederland, 2013, UPD ACT CIJF AANG AA
[7]   First-year mortality rates for selected birth defects, Hawaii, 1986-1999 [J].
Forrester, MB ;
Merz, RD .
AMERICAN JOURNAL OF MEDICAL GENETICS PART A, 2003, 119A (03) :311-318
[8]   Pierre Robin sequence: A series of 117 consecutive cases [J].
Holder-Espinasse, M ;
Abadie, V ;
Cormier-Daire, V ;
Beyler, C ;
Manach, Y ;
Munnich, A ;
Lyonnet, S ;
Couly, G ;
Amiel, J .
JOURNAL OF PEDIATRICS, 2001, 139 (04) :588-590
[9]  
HUJOEL PP, 1992, CLEFT PALATE-CRAN J, V29, P451, DOI 10.1597/1545-1569(1992)029<0451:FYMAIW>2.3.CO
[10]  
2