2006 Wiley-Liss, Inc.

Birth Defects Research (Part A) 76:170 174 (2006)

Original Articles

Epidemiologic Characteristics of Congenital

Diaphragmatic Hernia among 2.5 Million California
Births, 1989 1997
Wei Yang,* Suzan L. Carmichael, John A. Harris, and Gary M. Shaw
March of Dimes Birth Defects Foundation, California Birth Defects Monitoring Program, Berkeley, California
Received 14 July 2005; Revised 29 August 2005; Accepted 14 September 2005

BACKGROUND: Congenital diaphragmatic hernia (CDH) is a severe birth defect of unknown etiology. The
aims of the current report were to extend the literature on the descriptive epidemiology of CDH and to
determine whether its mortality rate decreased in California during the study period. METHODS: Using data
from a large population-based registry, we ascertained 631 CDH cases from 1989 to 1997. We also classied
cases as isolated or nonisolated based on the presence and type of major accompanying malformations.
Approximately 2.5 million live and stillbirths occurred during the ascertainment period. Multivariate Poisson
regression analysis was applied to examine the time trend and risk factors. RESULTS: The overall prevalence
of CDH was 2.49 per 10,000 live and stillbirths and did not vary over the study period. Isolated cases, which
accounted for 58% of cases, revealed a prevalence of 1.45 per 10,000. Heart malformations were the most
frequent major anomalies accompanying CDH. We observed a lower risk of isolated CDH among blacks.
Advanced maternal age groups had a higher risk for nonisolated CDH. Multiparous women tended to have a
lower risk for nonisolated CDH. Male infants and multiple births had an increased risk for isolated and
nonisolated CDH. Infant mortality was slightly decreased over the study period. CDH cases with additional
anomalies had higher mortality rates than isolated cases. CONCLUSION: Our observations add to the
relatively few population-based descriptive epidemiologic studies of the prevalence and mortality, and maternal and infant characteristics of CDH. Birth Defects Research (Part A) 76:170 174, 2006.
2006 Wiley-Liss, Inc.
Key words: birth defects; congenital diaphragmatic hernia; epidemiology; prevalence; trends

INTRODUCTION
Congenital diaphragmatic hernia (CDH) is a severe birth
defect in which some portion of the abdominal contents
protrudes into the thoracic cavity, with failure of development of the left (85%), right (13%), or both (2%) diaphragms (Stevenson et al., 1993; Rudolph et al., 2003). CDH
has been phenotypically described as an absence of the
diaphragm, Bochdalek hernia (posterolateral), Morgagni
hernia (anterior), and paraesophageal hernia (Stevenson et
al., 1993). The prevalence of CDH varies from 1 in 2,000 to
1 in 4,200 live births among different regions and across
time periods (Torfs et al., 1992; Cannon et al., 1996; Kaiser
and Rosenfeld, 1999; Stege et al., 2003; Tonks et al., 2004).
Despite improved prenatal diagnostic techniques and ad-

vances in neonatal intensive care, the mortality for CDH

has been estimated to be as high as 89%, depending on the
presence of additional structural or chromosomal anomalies (Kaiser and Rosenfeld, 1999). Moreover, infants who
survive have persisting morbidity with poor prognoses
(Huddy et al., 1999).

Grant sponsor: Centers for Disease Control and Prevention: Centers of Excellence; Grant number: U50/CCU913241.
*Correspondence to: Wei Yang, California Birth Defects Monitoring Program,
1917 Fifth Street, Berkeley, CA 94710. E-mail: wya@cbdmp.org.
Published online 1 March 2006 in Wiley InterScience (www.interscience.wiley.
com).
DOI: 10.1002/bdra.20230

Birth Defects Research (Part A): Clinical and Molecular Teratology 76:170 174 (2006)

RISK FACTORS FOR DIAPHRAGMATIC HERNIA

CDH results from failure of the posterolateral pleuroperitoneal canals to separate and develop the thoracic and
abdominal cavities during the eighth week of gestation
(Behrman et al., 2000). Studies with nitrofen-induced CDH
animal models have suggested that CDH may be due to a
primary disturbance of pulmonary growth into the pleuroperitoneal canal, which then disturbs the growth of the
posthepatic mesenchymal plate, the main origin of the
diaphragm (Iritani, 1984; Keijzer et al., 2000; Rottier and
Tibboel, 2005). The etiologies of CDH remain unknown,
although both genetic and environmental factors have
been implicated. Friend of gata 2 (FOG2) was the rst gene
implicated in the pathogenesis of nonsyndromic human
CDH (Ackerman et al., 2005). Genes that regulate the retinoid signaling pathway may contribute to the etiology of
CDH as well (Greer et al., 2003). Polybrominated diphenyls, thalidomide, quinine, nitrofen, phenmetrazine, and
vitamin A deciency have been reported to cause CDH in
various experimental animal models (Tibboel and Gaag,
1996; Greer et al., 2003).
Several epidemiologic studies have focused on the relationship of maternal and infant characteristics and risk for
CDH, but have produced inconsistent results (Torfs et al.,
1992; Robert et al., 1997; Riley et al., 1998; Hollier et al.,
2000; Lary and Paulozzi, 2001; Dott et al., 2003). The aims
of the current study were to extend the California component of the Robert et al. (1997) and Torfs et al. (1992)
studies of the descriptive epidemiology of CDH by using
data from a population-based registry linked with 2.5 million California births that occurred in the period of 1989
1997, controlling for additional covariates, substantially
enlarging the study case population, and incorporating
more recent births. In addition, we assessed whether the
mortality rate decreased in California during the study
period.

MATERIALS AND METHODS

Cases were obtained from the California Birth Defects
Monitoring Program (CBDMP), a population-based registry that actively collects information about births with
structural and chromosomal congenital malformations
from medical records of the nonmilitary hospitals in California. This study focused on infants (cases) who were
delivered between 1 January 1989 and 31 December 1997,
and included all live and stillbirths (dened as 20 weeks
of gestation) (Croen et al., 1991). Data-collection specialists
reviewed medical records at hospitals and genetic centers
in selected California counties to identify all structural
malformations diagnosed within 1 year of age. The overall
ascertainment has been estimated to be 97% complete
(Schulman and Hahn, 1993).
Live and stillbirths diagnosed as British Pediatric Association (BPA) codes 756.60 (absence of diaphragm),
756.610 756.615 (Bochdalek diaphragmatic hernia), and
756.617 (absence of hemidiaphragm), with conrmation by
surgery or autopsy, were eligible as cases. Additionally,
infants who did not have surgery or autopsy conrmation
but died within 1 year of birth were also considered eligible. For complex cases, charts were reviewed by 1 of the
authors (J.H.A.). Infants and fetuses diagnosed with Morgagni hernia (756.616) and diaphragmatic eventration
(756.620) or with known autosomal single-gene disorders
were excluded because the etiologies of these phenotypes
were believed to differ from those of the remaining cases

171

(Torfs et al., 1992). Of the 2,537,099 births that occurred

from 1989 to 1997, 631 infants with CDH were identied
and met our case eligibility criteria. Of these 631 case
infants, 300 were included in the previous study conducted
by Robert et al. (1997). Cases were further classied into 3
mutually exclusive groups: isolated (n 369), which includes cases with lung anomalies (748.3748.9), malrotation (751.4), or Ladds bands (751.100); nonisolated (n
210), which includes cases with at least 1 additional major,
apparently unrelated anomaly; and chromosomal abnormalities (n 52).
Cases were linked to California live birth, fetal death,
and infant death certicates to obtain maternal and infant
characteristics. The relative risks (RRs) for the ratio of
prevalences comparing an at-risk group to its reference,
and corresponding 95% condence intervals (CIs) were
calculated (Poisson regression, Statistical Analysis Software, version 9.1; SAS Institute, Cary, NC) for the following variables: maternal race/ethnicity (non-Hispanic
white, U.S.-born Hispanic white, foreign-born Hispanic
white, black, Asian, or other), maternal age (20, 20 24,
2529, 30 34, 3539, or 40 years), maternal education (12,
12, or 12 years), infant sex, parity (0, 1, 2 or 3) and
plurality (singleton or multiple).

RESULTS
The prevalence of CDH per 10,000 live and stillbirths
was 2.49. It was 1.45 for isolated cases and 0.83 for nonisolated cases. The birth prevalence did not vary by years
during the study period, that is, annual prevalence did not
change substantially over the 9-year study period (RR/
year, 0.98; 95% CI, 0.951.01). Of the 492 cases with known
laterality, 78% (383/492) were left-sided, 20% (101/492)
were right-sided, and 2% (8/492) were bilateral.
Among the 52 CDH cases with chromosomal anomalies,
the numbers of cases for trisomy 18, trisomy 13, trisomy 21,
trisomy 22, trisomy 9, and nontrisomy chromosomal
anomalies were 27 (52%), 6 (12%), 1(2%), 2 (4%), 1 (2%),
and 15 (29%), respectively.
Table 1 displays the percentages of the 210 nonisolated
cases with particular diagnoses for diagnostic groups at the
4-digit level BPA codes that included 3% (7) of cases.
Among these infants, 23.3% had other specied anomalies
of the heart (746.8), 16.2% had other anomalies of the ribs
and sternum (756.3), 15.7% had anomalies of the spine
(756.1), 14.8% had other specied anomalies of the brain
(742.4), and 10.5% had congenital hydrocephalus (742.3).
Of the 579 cases of isolated and nonisolated CDHs that
occurred from 1989 through 1997, 29 were excluded from
the multivariate Poisson regression because they were
missing covariate data. The results of the crude and adjusted RRs were similar; therefore, only the adjusted RRs of
the study factors for the occurrence of isolated and nonisolated CDH are presented in Table 2. We considered adjusted RRs with magnitude 1.5 or 0.7 worthy of comment. After adjustment for other maternal and infant
characteristics, Blacks had a 37% lower risk of developing
isolated CDH than non-Hispanic Whites. For nonisolated
cases, relative risks were elevated at least 50% in maternal
age 35 years or older, compared with the group aged 20 24
years. Previous live births of two showed a 48% decreased
risk in nonisolated cases. These associations with maternal
age and parity were not observed among isolated cases.
Birth Defects Research (Part A) 76:170 174 (2006)

172

YANG ET AL.
Table 1
Frequency of Accompanying Malformation Groups in 210 Infants with Congenital
Diaphragmatic Hernia among Liveborn and Stillborn Infants Observed in This
Study in California 1989 1997
Malformation group (BPA code)a
741.9
742.0
742.1
742.2
742.3
742.4
745.1
745.2
746.0
746.4
746.8
747.1
747.2
747.3
747.4
750.3
751.2
753.0
753.1
753.2
753.3
754.0
755.1
755.2
755.5
756.0
756.1
756.3
756.7
759.0
759.3
759.8

Spina bida w/o hydrocephalus

Encephalocele
Microcephalus
Reduction deformities of brain
Congenital hydrocephalus
Other specied anomalies of brain
Transposition of great vessels
Tetralogy of fallot
Anomalies of pulmonary valve
Congenital insufciency of aortic valve
Other specied anomalies of the heart
Coarctation of aorta
Other anomalies of aorta
Anomalies of pulmonary artery
Anomalies of great veins
Tracheo-esophageal stula, esophageal, atresia and stenosis
Atresia and stenosis of large intestine, rectum and anal canal
Renal agenesis and dysgenesis
Cystic kidney disease
Obstructive defects of renal pelvis and ureter
Other specied anomalies of kidney
Musculoskeletal deformities of skull, face and jaw
Syndactyly
Reduction defects of upper limb
Other anomalies of upper limb, including shoulder girdle
Anomalies of skull and face bones
Anomalies of spine
Other anomalies of ribs and sternum
Anomalies of abdominal wall
Anomalies of spleen
Situs inversus
Other specied anomalies and syndromes

Count

Percent (%)b

7
7
11
10
22
31
12
8
8
11
49
10
20
9
12
9
14
15
8
11
11
10
10
18
8
8
33
34
18
17
7
11

3.33
3.33
5.24
4.76
10.48
14.76
5.71
3.81
3.81
5.24
23.33
4.76
9.52
4.29
5.71
4.29
6.67
7.14
3.81
5.24
5.24
4.76
4.76
8.57
3.81
3.81
15.71
16.19
8.57
8.10
3.33
5.24

a
Grouping derived from 4-digit denitions used in the British Pediatric Association classication of
disease.
b
Percent reects number of infants among 210 with that particular diagnosis. A case with 1
diagnosis was counted in each corresponding group.

Male infants and multiple births had increased risk of

isolated and nonisolated CDH.
Mortality within the rst year among 584 live births
signicantly decreased 8% per year on average over the
study period, from 47% in 1989 to 28% in 1997 (RR/year,
0.92; 95% CI, 0.86 0.98). These reductions in mortality
were similar for isolated (RR/year, 0.91; 95% CI, 0.831.01)
and nonisolated (RR/year, 0.96; 95% CI, 0.851.07) cases.
Overall, the mortality of infants with CDH from 1983
through 1997 was 38.9% for total live births (n 584),
23.5% for isolated live births (n 358), 57.2% for nonisolated live births (n 187), and 92.3% for live births with
chromosomal anomalies (n 39). Compared with the risk
of death for isolated cases, the risk of death was 4-fold
higher for nonisolated cases and 39-fold higher for cases
with chromosomal anomalies (data not shown).

DISCUSSION
In this population-based study we observed that the
prevalence of CDH was 2.49 per 10,000 live and stillbirths.
Isolated CDH represented 55% of the total prevalence, and
the prevalence of isolated CDH was decreased among
Birth Defects Research (Part A) 76:170 174 (2006)

blacks. The prevalence of nonisolated CDH was increased

in offspring whose mothers were 35 years old. The prevalence of both isolated and nonisolated CDH was elevated
in male infants and multiple births. Additionally, the infant mortality was slightly decreased over the study period
(1989 1997).
The prevalence of CDH observed in this study is close to
that reported by Dott et al. (2003), which employed similar
case classication criteria, but the prevalence is lower than
that reported by others (Torfs et al., 1992; Stege et al., 2003;
Tonks et al., 2004). Tonks et al. (2004) and Stege et al. (2003)
included terminations of pregnancy, whereas Torfs et al.
(1992) included anterolaternal hernia, Morgagni hernia,
and pars sternalis hernia, which were excluded in our
study.
The increased frequency of cardiovascular anomalies
co-occurring with CDH observed in our study is consistent
with that of previous investigations (Benjamin et al., 1988;
Cunniff et al., 1990; Cannon et al., 1996; Dott et al., 2003).
Such an increased frequency does not directly indicate a
potentially common factor between CDH and certain heart
anomalies because cardiovascular anomalies are the most

RISK FACTORS FOR DIAPHRAGMATIC HERNIA

173

Table 2
Adjusted Relative Risks (RRs) for Isolated and Non-Isolated CDH with Maternal and Birth Characteristics Among
Liveborn and Stillborn Infants in California (1989 1997)
Isolated

Race/ethnicity
Non-Hispanic white
U.S.-born Hispanic white
Foreign-born Hispanic white
Black
Asian
Other
Education (years)
12
12
12
Age
2024
20
2529
3034
3539
4055
Parity
0
1
2
3
Plurality
Singleton
Multiple
Infant sex
Female
Male
a

Non-isolated

No. of births
(n 2,506,128)

No. of cases
(n 355)a

Adjusted RR
(95% CI)b

No. of cases
(n 195)a

Adjusted RR
(95% CI)b

772,481
357,302
886,375
200,042
182,831
107,097

115
54
130
19
22
15

Ref.
0.97 (0.691.37)
0.91 (0.671.22)
0.63 (0.381.03)
0.78 (0.491.24)
0.96 (0.561.64)

64
29
69
14
12
7

Ref.
1.00 (0.631.60)
0.95 (0.631.43)
0.88 (0.491.59)
0.75 (0.401.39)
0.80 (0.371.75)

959,352
727,277
819,499

145
107
103

Ref.
0.95 (0.721.26)
0.76 (0.551.06)

78
55
62

Ref.
0.93 (0.631.37)
0.82 (0.521.28)

636,964
314,160
705,875
547,291
248,587
53,311

85
49
102
74
38
7

Ref.
1.08 (0.741.56)
1.15 (0.851.55)
1.11 (0.801.56)
1.27 (0.841.92)
1.08 (0.492.38)

44
31
41
45
26
8

Ref.
1.24 (0.762.02)
0.92 (0.591.43)
1.37 (0.872.14)
1.77 (1.043.00)
2.51 (1.145.54)

981,090
761,750
418,673
344,615

147
106
51
51

Ref
0.91 (0.711.18)
0.77 (0.551.08)
0.91 (0.631.31)

88
57
21
29

Ref
0.82 (0.581.17)
0.52 (0.320.86)
0.77 (0.471.26)

2,449,451
56,677

343
12

Ref
1.54 (0.872.74)

187
8

Ref
1.80 (0.883.66)

1,224,872
1,281,256

131
224

Ref
1.64 (1.322.03)

74
121

Ref
1.56 (1.172.09)

A total of 29 isolated and nonisolated cases were excluded due to missing covariate data.
RR was adjusted for all other variables in the table.

frequent group of anomalies, and therefore one would

expect this group to be more frequently represented even if
no co-occurring association existed. However, we previously observed that cardiovascular anomalies did not occur more frequently in infants with cleft palate and cleft lip
with or without cleft palate (Shaw et al., 2004). Thus, the
increased frequency of cardiovascular anomalies co-occurring with CDH merits additional study for potential etiologic clues. For example, as genetic contributions for certain types of cardiovascular malformations become
known, an association between cardiac and CDH defects
might be helpful in identifying the genes that are responsible for CDH.
The results from previous studies that examined CDH
risks associated with race/ethnicity were inconsistent, and
none of those studies adjusted for other risk factors (Torfs
et al., 1992; Yang et al., 1994; Robert et al., 1997; Dott et al.,
2003). After we adjusted for other maternal and infant
characteristics, we observed that non-Hispanic whites
were at higher risk for isolated CDH than blacks, in agreement with previous study ndings (Yang et al., 1994; Robert et al., 1997).
Our study showed a higher risk of nonisolated CDH
among older mothers, which was observed in 2 previous
studies (Yang et al. 1994; Hollier et al., 2000), but not all
(Torfs et al., 1992; Robert et al., 1997; Dott et al., 2003). We

hypothesized that different patterns of co-occurring anomalies between the different maternal age groups might
explain why we observed an elevated risk with increasing
age only among the nonisolated group. However, no particular BPA diagnosis appeared to be more common in the
younger or older age groups to raise the risk in those
groups above that of the 20 to 24-year age group.
The higher risk of CDH observed for multiple births was
consistent with limited data from previous studies. Studies
conducted by Mastroiacovo et al. (1999) and Robert et al.
(1997) of multiple registries and countries with 40 multiple births and 1000 singletons indicated an elevated risk
with increased plurality. Risk estimates were imprecise in
studies by Torfs et al. (1992) and Riley et al. (1998), owing
to a small number of twins available for study.
Our nding that males are more likely than females to
have isolated and nonisolated CDH is in general agreement with previous studies (Torfs et al., 1992; Yang et al.,
1994; Robert et al., 1997; Dott et al., 2003).
The infant mortality observed in this study within 1 year
of delivery was within the range reported previously
(Torfs et al., 1992; Cannon et al., 1996; Riley et al., 1998;
Kaiser and Rosenfeld, 1999; Dott et al., 2003; Stege et al.,
2003; Tonks et al., 2004). We observed that the mortality of
infants with CDH decreased over the study period from
1989 to 1997. The prevalence of CDH did not vary signifBirth Defects Research (Part A) 76:170 174 (2006)

174

YANG ET AL.

icantly over the years; therefore, the decrease in mortality

was probably not attributable to a change in case ascertainment (such as severely affected infants who died immediately after birth without being adequately diagnosed).
Medical management and surgical strategies for CDH infants changed substantially beginning in 1990, including
the use of exogenous surfactant, inhaled nitric oxide, highfrequency oscillatory ventilation, delayed surgery, permissive hypercapnia ventilation, extracorporeal membrane
oxygenation, and abandonment of conventional hyperventilation (Muratore and Wilson, 2000). Our study did not
collect information on these strategies. Therefore, we were
unable to investigate their inuence on survival for infants
with CDH.
This population-based study is strengthened by its large
sample size, diverse population, near-complete case ascertainment, and adjustment for several potential maternal
and infant characteristics. Therefore, this study provides
accurate prevalence and mortality data for CDH, a rare
disease. However, the ascertainment of malformations was
conditional on the completeness and accuracy of the medical records. Moreover, this study did not include ascertainment of malformations of fetuses that were electively
terminated. If diagnoses of malformed fetuses or decisions
to electively terminate differed substantially among the
study population groups, such differences could have biased the estimated risks. The extent of this potential bias on
the current results is unknown.
Despite these limitations, this large population-based
study estimates the prevalence, mortality, and demographic risk factors associated with CDH after adjusting
for other maternal and infant characteristics, and extends
the limited descriptive epidemiologic information available on CDH.

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