Congenital Heart Disease in Taiwan,
Republic of China
By MARY K. M. SHANN, M.B.
SUMMARY
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A review of 5 years' experience with congenital heart disease in National Taiwan
University Hospital, Taipei, Taiwan, Republic of China, shows that congenital aortic
valvar and supravalvar stenosis are not seen and the incidence of coarctation is very
low. Statistics on congenital heart disease as reported in studies on all groups of patients
from several other Asian countries were collected and compared with those from European and North American countries. The low incidence for the two mentioned congenital cardiac defects in the reports from Asian countries is probably not due to chance, but
may represent a true difference in incidence. In the discussion of etiological factors possibly responsible for the low incidence of coarctation and aortic stenosis, it is postulated
that dietary factors may be responsible for the observed differences in incidence.
Additional Indexing Words:
Aortic valvar and supravalvar stenosis
Coarctation of aorta
Rheumatic heart disease
Congenital heart disease in Asian countries
C ONGENITAL HEART DISEASE has
been studied frequently in the Western
countries,'-7 but it has not been studied
thoroughly on the Asian subcontinent. Of the
few studies8-12 made there, many often have
been based on a small number of cases and
incomplete data. The purpose of the present
report is to present our experience with congenital heart disease in Taiwan, based on 5
years of laboratory and clinical studies at the
National Taiwan University Hospital.
Methods
The cardiac catheterization laboratory of the
National Taiwan University Hospital has performed right and left heart catheterizations,
selective angiocardiography, and dye-dilution studies since October 1962. In the 5 years from October 1962 to September 1967, a total of 520 cardiac catheterizations has been done. Definitive
diagnosis of congenital heart disease was made
on 232 patients from these 520 studies and
the case records of the 356 children with
congenital cardiac disease admitted to the pediatric ward of the hospital from January 1963
From the National Taiwan University, School of
Medicine, Taipei, Taiwan, Republic of China.
The Cardiopulmonary Laboratory is donated and
equipped by the China Medical Board of New
York, U.S.A.
Table 1
Total Cardiac Admissions to Pediatric Ward between January 1963 and June 1967
Admissions to pediatric ward
Year
Total
1963
1964
1822
1935
1903
1857
972
8489
1965
1966
1967*
Total
Congenital
heart disease
Other
heart disease
56
74
85
84
6
15
23
35
4
2
3
1
57
356
17
96
10
*January to June 1967.
Csrculation, Volume XXXIX, February 1969
Rheumatic
heart disease
251
0
SHANN
252
Table 2
Relative Incidence of Various Types of Congenital
Heart Diseases among 232 Cases Diagnosed in
the Cardiopulmonary Laboratory of NTUH
Diagnosis
Interventricular septal defect
Tetralogy of Fallot
Patent ductus arteriosus
Interatrial septal defect
Secundum
....
Infundibular
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With
pulmonic
stenosis
19.4
18.1
16.0
8.6
14
6.0
9
3.9
8
8
3.4
3.4
7
3.0
5
2.1
4
1.7
3
1.3
3
1.3
shows the number of cardiac admissions to the hospital in the past 432 years.
Of a total of 8,489 pediatric patients, 356 children had congenital cardiac malformations
and 96 had rheumatic heart disease. The ratio
5
Cardiomyopathy of obscure origin
Without subaortic stenosis ... .3
With subaortic stenosis ........ 4
Pentalogy of Fallot
Pulmonary hypertension of unknown
etiology
Interventricular septal defect
+ aortic insufficiency
1
Without infundibular stenosis
With infundibular stenosis .... 2
Coarctation of aorta
Without patent ductus ........ 1
With patent ductus
........ 2
0.4
0.4
45
42
37
20
2
Transposition of great vessels
Without pulmonic stenosis .... 3
With pulmonic stenosis ........ 6
Dextrocardia with combined anomalies
Double outlet of right ventricle
Without pulmonic stenosis .. 3
%
1
1
232
%
13
Pulmonary stenosis (intact ventricular
septum)
..... 12
Valvar
No.
No.
7
Primum
Diagnosis
Persistent A-V canal
Levocardia
Total
through June 1967 have been collected and analyzed. The cases of congenital cardiac disease
encountered in the medical or surgical wards
and the medical, surgical, or pediatric outpatient clinics of the hospital were excluded. Of the
356 patients admitted to the pediatric ward, 146
(41%) had cardiac catheterization, angiocardiography, or both, and less than 10% had postmortem
examination. For the rest, the diagnosis was made
on the basis of clinical evaluation.
Patients with the incomplete form of atrioventricularis communis were classified as having persistent ostium primum defect. Only the complete
form of atrioventricularis communis was classified
as persistent A-V canal. Major associated congenital anomalies, such as Down's, Marfan's, Turner's or Ellis-van Creveld syndrome, were
searched for.
There is some overlapping between the patients undergoing catheterization and those admitted to the hospital.
solely
Results
Table
1
Persistent truncus arteriosus
Ebstein's malformation
Stenosis of one branch of pulmonary artery
2
0.9
of congenital
2
0.9
Endocardial fibroelastosis
Syndrome of asplenia
Interventricular septal defect
+ pulmonic valvar stenosis
Total anomalous drainage of
pulmonary veins
Hypoplastic left heart syndrome
Interventricular septal defect
+ patent ductus
Tricuspid atresia
Ruptured aneurysm of sinus of Valsalva
into right ventricle
Coronary artery anomaly
Coronary arteriovenous fistula
Complete interruption of aortic arch
Pulmonic stenosis + aortic insufficiency
Pulmonic stenosis + tricuspid insufficiency
Interventricular septal defect
+ interatrial septal defect
2
0.9
4:1. Congenital heart disease accounted for
4.2% of the total pediatric admissions.
2
0.9
2
0.9
2
2
0.9
0.9
1
0.4
0.4
0.9
1
1
1
1
0.4
0.4
0.4
1
0.4
0.4
0.4
1
0.4
1
1
to
rheumatic heart disease
was
Table 3
Sex Distribution of the 232 LaboratoryDiagnosed Cases
Age
and
Age (yr)
0- 6 mo
7-12 mo
13 mo-2
3- 5
6-10
11-15
16-20
21-30
31-40
41-50
51-60
Total
Female
Male
9
4
12
4
6
7
18
32
21
17
13
3
1
1
129
14
28
13
11
11
4
3
0
103
Circulation, Volume XXXIX, February 1969
CONGENITAL HEART DISEASE IN TAIWAN
253
Table 4
Age and Sex Distribution of the Five Major Defects of the 232 Patients with LaboratoryDiagnosed Cardiac Lesion
Age (yr)
F
0- 6 mo
7-12 mo
13 mo-2
3- 5
6-10
11-15
16-20
21-30
31-40
41-50
51-60
3
0
1
3
2
3
2
6
0
0
0
20
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Total
VSD
VSD
M
0
3
1
2
8
2
3
4
1
0
1
25
Tetralogy
M
F
F
1
1
1
2
8
1
1
0
0
0
0
15
0
1
1
6
8
3
3
2
1
0
0
25
2
0
3
4
7
6
4
0
1
0
0
27
PDA
M
F
1
0
0
3
2
3
2
1
0
0
0
12
0
0
0
0
4
1
0
2
0
1
0
8
ASD
M
F
0
0
1
0
3
3
2
4
0
0
0
13
1
0
0
1
1
2
1
0
0
0
0
6
PS
M
0
0
0
1
4
0
2
1
0
0
0
8
ventricular septal defect; tetralogy= tetralogy of Fallot; PDA =patent ductus
= pulmonary stenosis.
arteriosus; ASD = atrial septal defect; PS
The relative frequency of the various types
of congenital heart disease in the 232 cases
diagnosed by the Cardiopulmonary Laboratory is shown in table 2. Interventricular septal
defect was the most frequently encountered
defect, comprising 19.4% of the total. In descending order, tetralogy of Fallot was found
in 18.1%, patent ductus arteriosus in 16.0%,
interatrial septal defect in 8.6%, pulmonic stenosis with intact ventricular septum in 6.0%,
and transposition of the great vessels in 3.9%.
Only three cases of coarctation of aorta with
or without patent ductus (1.3%) were encountered. There were no cases of congenital
aortic stenosis.
Table 3 shows the age and sex distribution
of these 232 patients. The youngest patient
was a 1-month-old baby boy who died 12
hours after cardiac catheterization. Both mitral and aortic atresia with patent ductus were
found at postmortem examination. The oldest
patient was a 57-year-old man with interventricular septal defect. The sex distribution did
not show a significant difference, there being
103 females and 129 males.
Table 4 shows the number, age, and sex of
patients with each of the five major defects
found in this series. Interventricular septal defect, tetralogy of Fallot, and patent ductus arteriosus were usually diagnosed at an earlier
age than were interatrial septal defects.
Circulation, Volume XXXIX, February 1969
The relative incidence of various types of
cardiac defects in the 356 pediatric patients
admitted to the hospital is shown in table 5.
Interventricular septal defect was present
about twice as frequently in this group as in
the group diagnosed at cardiac catheterization. A clinical diagnosis of coarctation of the
aorta was made in four cases (1.1%). Again
there was no case of aortic stenosis. The distribution of the patients according to age and
sex groups among the clinically diagnosed
cases is shown in table 6. There were more
younger patients in this group than in the
laboratory-diagnosed group.
Fifteen patients had Down's syndrome. Of
these patients, nine had interventricular septal defect, three, persistent A-V canal, and
one, an ostium primum defect clinically. The
one who underwent cardiac catheterization
was found to have a patent ductus arteriosus
plus an interventricular septal defect.
Discussion
This study shows a notable increase in the
number of pediatric cardiac admissions to the
hospital in the past 5 years (table 1). This
increase, however, can probably be explained
by improved diagnostic methods and better
medical and surgical prognosis rather than
by any true increase in the prevalence of
heart disease. Thus, the true prevalence of
254
SHANN
Table 5
Relative Incidence of Distribution of Various
Defects among the 356 Cases Diagnosed on
Clinical Findings
No.
%
Age
Female
Male
140
44
35
19
39.3
12.3
9.8
5.3
9
19
2.5
5.3
0- 6 mo
7-12 mo
13-36 mo
3- 5 yr
6-10 yr
11-15 yr
16+ yr
Total
51
28
26
17
31
8
2
163
71
23
17
27
33
20
2
Diagnosis
Interventricular septal defect
Tetralogy of Fallot
Patent ductus arteriosus
Interatrial septal defect
Secundum
Primum
Pulmonary stenosis
I
..
9
10
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Transposition of great vessels
Without pulmonary stenosis ....13
With pulmonary stenosis ..
6
Dextrocardia
Double outlet of right ventricle
Without pulmonary stenosis .... 3
With pulmonary stenosis
... 2
Cardiomyopathy of obscure origin
Pentalogy
Pulmonary hypertension of
unknown etiology
Interventricular septal defect
+ aortic insufficiency
Coarctation of aorta
Without patent ductus ..
With patent ductus
....
With ventricular septal defect
Truncus arteriosus
Ebstein's malformation
Endocardial fibroelastosis
Syndrome of asplenia
Total anomalous drainage of
pulmonary veins
Interventricular septal defect
+ patent ductus
Coronary artery anomaly
Hypoplastic left heart syndrome
Tricuspid atresia
Complete interruption of aortic arch
Interventricular septal defect
+ pulmonary insufficiency
Levocardia
Corrected transposition
Persistent A-V canal
Glycogen storage disease
Aortic runoff (probably aortic
regurgitation)
Cor triatriatum
Congenital heart block
Diagnosis uncertain
Total
...
Table 6
Age and Sex Distribution of the 356 Patients
with Clinically Diagnosed Cases of Congenital
Cardiac Disease
2
1
I
1
1
2
18
356
heart disease in the pediatric age group is still
unknown. Admission for congenital heart disease was four times as frequent as that for
193
rheumatic heart disease and the former was
the main reason for pediatric cardiac admission. In the past it was believed that congenital and rheumatic heart diseases occurred with
equal frequency in the pediatric age
group.13, 14 But more recently Nadas2 has
reported that at least 10 times as many new
patients with congenital cardiac lesions as
compared to those with rheumatic disease
are admitted yearly to Boston Children's Hospital Medical Center in the United States.
Keith and associates4 gave the ratio of congenital to rheumatic heart disease in Toronto
in his series as 20:1.
The present study indicates that rheumatic
heart disease is one of the most important
pediatric cardiac problems in Taiwan although Taiwan is located in the semitropics
and rheumatic fever is considered to be more
common in the temperate zones.15' 16 Reports
from Pakistan17 and the Philippines,'" two
tropical countries, also indicate that rheumatic
heart disease is at least as common in the
tropics as in nations in the temperate zones.
Because the present study is limited to hospital experience only, it may not represent the
incidence in the population; therefore. a
more accurate epidemiological study of the
prevalence of pediatric heart disease in Asian
countries is needed.
It is not unusual to find that interventricular septal defect is the most frequently
seen congenital cardiac defect among the
single anomalies, and tetralogy of Fallot is
most frequent among the multiple anomalies.
Circulation, Volume XXXIX, February 1969
CONGENITAL HEART DISEASE IN TAIWAN
However, the absence of congenital aortic
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valvar and supravalvar aortic stenosis plus
a very low occurrence of coarctation with
or without patent ductus in the clinical and
laboratory series is of interest since both of
these lesions are reported as relatively common congenital cardiac defects by authors
in Europe3 5 and North America.2 4 The
present series, although it includes a smaller
number of cases, covers a long period (5
years) and so the reported differences are of
interest. Additional statistics on congenital
heart disease collected from the reports of
authors from different Asian countries,8-12 and
these figures are shown in table 7.
In the Philippines, Imperial and Felarca8
reported in 1963 a study of 6,000 autopsy
cases. Among these were 67 cases of congenital heart disease. Interventricular septal defect and tetralogy of Fallot were the most
frequently encountered anomalies, but no case
of coarctation of aorta or aortic valvar stenosis
was noted. In 1964, Alimurung'9 made a statement that coarctation of aorta appeared to be
a rare lesion among the Filipinos.
There are a few studies concerning congenital heart disease in Japan.9' 10 In Furuta's
series of 247 cases in which the diagnosis was
confirmed at surgery and postmortem examination, aortic valvar or supravalvar stenosis
was not found and coarctation of aorta was
found in only four (1.5%) cases. This incidence is similar to that found in our series.
Wada'0 reported 322 cases of congenital
heart disease, all of which were diagnosed at
surgery; he observed only one case of aortic
stenosis and one of coarctation of the aorta.
A small series of patients with congenital
heart disease has been reported from Korea",;
this consisted of 132 cases seen both during
hospital admission and in an outpatient department. Of these 132 cases, 4.5% were diagnosed as aortic stenosis but none as coaretation of aorta.
Ongley12 noticed the difference in the frequency of certain forms of congenital cardiac
defects in Thailand as compared with the
frequency in the United States. He stated
that aortic valvar or supravalvar stenosis and
Circulation, Volume XXX1X, February 1969
255
simple coarctation of the aorta were virtually nonexistent in the autopsy materials among
the Thai over the past several years. Only
one patient with coarctation plus patent ductus was operated on at Siriraj Hospital during the same period.
It is not possible to conclude with certainty
that the incidence of aortic valvar and supravalvar stenosis and coarctation is lower in
some Asian countries than in European and
North American countries. However, although
reports of congenital heart disease in Asian
countries have been few, mostly based on
retrospective analysis of small numbers of
clinical or autopsy material, table 7 suggests
that incidence of coarctation and aortic stenosis is much lower in the reported series
from Asian than in those from Western countries.
If the observed low incidence of both aortic valvar stenosis and coarctation in Asian
countries is true, what is the cause? Is it race,
geography, genetics; diet, or some combination
of these and other factors?
The five mentioned Asian countries do not
belong to the same geographic area or the
same race and, therefore, these factors are
probably not responsible for any difference
in frequency of congenital cardiac malformations.
The dietary habits in these five Asian countries and of the greater part of Asia are similar to each other, with milled rice as the chief
food. Although a general diet survey has been
lacking, Huang and Tung20 showed that most
Taiwanese are not adequately nourished and
a high incidence of vitamin deficiency disease
exists, especially those associated with the
vitamin B group. The incidence is not as high
as in Bataan, the Philippines, or Japan.21-24
Vitamin D deficiency is also seen frequently
in Japan.25-28
If Perou's29 theory is correct, coarctation of
aorta and supravalvar aortic stenosis may
be variants of a single lesion of similar etiology. The lower vitamin diet of Asian people
might actually protect their proximal aorta
during the intrauterine or neonatal period.
256
SHANN
Table 7
Percentage Incidence of Major Defect Reported by Authors from Various Countries
Author
Nadas2
Wood, P.3
Keith
et al.4
Kjellberg
et al.5
& Warburg6
Country
U.S.A.
England
Canada
Sweden
Denmark
Source of material
Cardiac
cath.;
surgery;
autopsy
3786
No. of cases
Hansen
Cardiac
cath.
900
6647
668
1678
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Interventricular septal defect
Tetralogy (VSD + PS)
19.97
11.0
25.0
17.5
6.5
14.55
11.0
10.2
9.4
14.3
Patent ductus arteriosus
12.30
15.0
12.1
26.5
21.0
Pulmonary stenosis
Interatrial septal defect
11.97
12.0
8.5
10.5
10.4
10.04
19.5
10.6
11.5
Aortic stenosis
5.73
3.0
5.5
3.4
Coarctation of aorta
4.99
9.0
5.6
10.2
18.0
3.6
7.2
Transposition of great vessels
3.96
1.0
5.4
3.0
A-V communis (or endocardial
cushion defect)
3.94
1.1
Persistent truncus arteriosus
The low incidence of aortic stenosis and coarctation may be related to dietary deficiencies
in vitamins in Asian countries in contrast to
what may be an excessive vitamin D intake
in the United States and other Western countries. This may prove a fruitful avenue for
research into the etiology of congenital heart
disease.
Acknowledgment
The author wishes to express her deepest gratitude
to Dr. Leonard M. Linde, Associate Professor of the
Department of Pediatrics and Physiology, University
of California, Los Angeles, for his advice and encouragement.
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Pulmonary Valve Incompetence
(Graham Steell Murmur)
I wish to plead for the admission among the recognized auscultatory signs of
disease of a murmur due to pulmonary regurgitation, such regurgitation occurring
independently of disease or deformity of the valves, and as the result of long-continued
excess of blood pres.sure in the pulmonary artery.
I am prepared for the objection that the murmur under consideration is only the
murmur of a slight amount of aortic regurgitation, the usual evidence of which in the
pulse is masked by the mitral lesion.
The murmur of high-pressure in the pulmonary artery is not peculiar to mitral
stenosis, although it is most commonly met with, as a consequence of this lesion. Any
long-continued obstruction in the pulmonary circulation may produce it. The pulmonary
valves, like the aortic, do not readily become incompetent, apart from structural change.
Probably no amount of blood pressure in the pulmonary artery will render them so
suddenly, as, at least, theoretically, the mitral valves may be rendered incompetent.
Changes in the vessel, with widening of its channel, and, eventually, of its orifice, long
precede the occurrence of incompetence of its valves.
.. . To those to whom neither the dogmatic assertion that all such murmurs as I have
described, are, in spite of the absence of any confirmatory evidence, aortic in origin,
nor the sceptical non possumus of Doctor Fagge, commends itself, I would urge a
careful consideration of "the high-pressure murmur of the pulmonary artery" as a
feasible explanation. I have stated my own opinion; from others I ask only that this
murmur should find a place-however subordinate-among the physical signs of disease
which they recognise.-GRAHAM STEELL: The Murmur of High-Pressure in the Pulmonary Artery. In WILLIUS, F. A., AND KEYS, T. E.: Classics of Cardiology, vol. 2. New
York, Dover Publications, Inc., 1941, p. 681.
Circulation, Volume XXXIX, February 1969
Congenital Heart Disease in Taiwan, Republic of China
MARY K. M. SHANN
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Circulation. 1969;39:251-258
doi: 10.1161/01.CIR.39.2.251
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Copyright © 1969 American Heart Association, Inc. All rights reserved.
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