T h e J o u r n a l of P E D I A T R I C S

643

Symmetrical conjoined twins: An analysis of the

birth records of twenty-two sets
Twenty-two sets o[ symmetrical conjoined twins have been analyzed with the birth
records of the New York State Department o[ Health as a source o[ cases.
Examination o[ the birth certificates in these cases reveal a marked predominance
o[ [emales, an excess o[ stillbirths, a marked tendency toward prematurity and
low combined birth weight, a maternal history o[ previous [etal loss, and a tendency
toward seasonal, geographic, and temporal aggregation o[ such births.

Samuel Milham, Jr., M.D., M.P.H.*

ALBANY,

SINCE

CON

N.

Y.

J GIN

ED

TWINNING

OC-

so infrequently, it is difficult to collect

a series of cases which does not suffer various
ascertainment biases. This report is concerned with an analysis of the birth records
of 22 sets of symmetrically conjoined twins
collected solely through the vital registration
mechanism of the New York State Department of Health. The reporting jurisdiction
of the New York State Department of Health
includes the entire state with the exception
of New York City. The time period encompassed by this study is January, 1945,
through April, 1965. The majority of these
cases were ascertained in the course of two
previously published, retrospective vital record studies: a study of central nervous system malformations, 1 and a study of twin
births. 2 Conjoined twins were included in
curs

From the New York State Department o[

Health, Albany, N. Y.
Supported in part by Public Health Service
Grant No. CH00154-01.
*Address New York State Department o[ Health,
84 Holland Ave., Albany, N; Y. 12208.

the central nervous system malformation

study because the rubric for "monstrosity"
in the International Classification of Diseases
includes both anencephalus and conjoined
twins. Twins born since 1962 were ascertained through an on-going malformation
surveillance system? Birth records were located for each case and abstracted. The
physician who attended delivery was contacted for additional information in one
ease where the anatomic description on the
birth record was vague and in another where
the sex of the twins was in question.

RESULTS AND D I S C U S S I O N
Table I presents information abstracted
from the certificates of birth. Fifteen of 22
sets were stillborn, and the average reported
gestation length for all cases was 33 weeks.
In 10 of 20 sets, the combined birth weight
was below 5 pounds. In 2 sets, the birth
weight was not stated. Only 1 of the 7
live-born sets survived (No. 8 in Table
!). Successful surgical separation was accomplished in this case. ~ The marked

6 44

Milham

October 1966

T a b l e I. D a t a a b s t r a c t e d from b i r t h records of conjoined twins born in upstate New York, January,

Case No.
1
2
3
4
5
6
7
8
9
10
11
12
13
14
15
16
17
18
19
20
21
22

Month and
year of birth

4/48
6/49
9/53
4/54
12/54
5/56
4/57
9/57
12/57
1/58
4/58
5/59
5/59
5/59
9/59
11/59
12/59
4/60
9/60
9/62
5/63
4/65

Race*
W
W
W
W
W
W
W
W
W
W
W
W
W
W
NW
W
W
W
W
NW
W
W

Weeks
gestation
26
24
36
24
32
40
32
39
19
38
38
28
28
37
40
29
40
37
40
40
28
32

Birth statust
SB
SB
SB
SB
SB
SB
SB
LB
SB
SB
SB
LB
LB
SB
LB
SB
SB
LB
SB
SB
LB
LB

Combined birthweigh~
N.S.$
3 lb.
4 lb., 10 oz.
1 lb., 5 oz.
5 lb., 12 oz.
8 lb., 8 oz.
6 lb., 4 oz.
14 lb.
1 lb., 7 oz.
10 lb.
N.S.~
9 lb., 12 oz.
2 lb., 10 oz.
3 lb.
8 lb., 4 oz.
4 lb., 6 oz.
4 lb., 13 oz.
7 lb., 15 oz.
5 lb., 12 oz.
11 lb., 10 oz.
3 lb.
3 lb., 15 oz.

*W, white; NW, non-whlte.

tLB, liveborn; SB, stillborn.
SNot stated.

p r e m a t u r i t y a n d excess of stillbirths are

evidence of the lethality of this m a l f o r m a tion.
T w e n t y of t h e 22 sets were female-female,
1 set was m a l e - m a l e , a n d 1 set (No. 9 in
T a b l e I) was r e p o r t e d as male-female! Since
all available evidence indicates t h a t conj o ' n e d twins a r e i n c o m p l e t e l y s e p a r a t e d
monozygotic or identical twins, the malefemale set is difficult to explain. T h e physician who delivered the twins r e m e m b e r e d
t h e m clearly as being of opposite sex, b u t no
p h o t o g r a p h was taken, a n d the specimen
was n o t saved. I t is possible t h a t the p h e n o typic sex of one m e m b e r of this set differed
from its genotypic sex. T h e sex r a t i o (20
F F : 1 M M ) is striking a n d is in the same
direction b u t of g r e a t e r m a g n i t u d e t h a n
F6rster's 5 extensive collection of conjoined
twins. H e f o u n d a sex ratio a m o n g symm e t r i c a l conjoined twins of 3 females to
1 male. E i t h e r the early female zygote is
m u c h more likely to u n d e r g o this t y p e of
change t h a n the male, or, once formed, male
conjoined twins m a y not be as viable as

female twins a n d are lost early in p r e g n a n c y

before a s c e r t a i n m e n t is possible.
T h e distribution of p a r e n t s ' ages is not
unusual, b u t the r e p r o d u c t i v e history of the
m o t h e r is noteworthy. O f the 32 previous
births r e p o r t e d b y the 15 m u l t i p a r o u s m o t h ers, 7 were stillbirths. Less t h a n 1 stillbirth
would have been expected a m o n g this group
of births, since the stillbirth r a t e for all
births in this state is less t h a n 2 p e r cent.
This would suggest t h a t a m a t e r n a l factor
m a y be of etiologic i m p o r t a n c e in conjoined
twinning.
T w o of the 22 sets were born to N e g r o
mothers. Since a p p r o x i m a t e l y 5 p e r cent of
u p s t a t e N e w Y o r k births a r e to non-white
mothers, the racial incidence in these cases
is not remarkable.
A n a t o m i c a l l y , 17 of 22 sets were joined
v e n t r a l l y (thoracopagus, xiphopagus, abd o m i n o p a g u s ) while there was only a single
case of dorsal union (ischiopagus). T h r e e
sets were of the r a r e dicephalus type, while
one was of t h e equally r a r e cephalothor a c o p a g u s type.

Volume 69

Number 4

EpidernioIogy of c o n j o i n e d twins

64 5

1945, to A p r i l , 1965

Maternal history
Parental age
Father
p Mother

Sex
f

28
28
27
26
32
27
29
29
23
37
29
28
N.S.:~
30
30
34
34
32
33

F F
F F
F F
IaM
F F
F F
F F
F F
F M
F F
F F
F F
F F
F F
F F
F F
F F
F F
F F
F F
F F
F F

Total previous births

Previous
stillbirths

Anatomic description on birth record

I
2
1
0
2
3
1
2
0
3
3
4
0
0
4
1
2
1
2
0
0
0

0
2
0
0
0
0
0
0
0
1
2
0
0
0
2
0
0
0
0
0
0
0

Fused head and chest

Joined at thorax
Thoracopagus
Joined at abdomen; one umbilical cord
Single torso; single sacrum
Joined at sternum; single heart
Joined at chest and abdomen
I schiopagus
Thoracopagus
Dicephalus dipus tetrabrachius
Thoracopagus
Thoracopagus
Conjoined omphalocele
Dicephalus dipus tribrachius
Thoracopagus
Thoracopagus
Abdominopagus
Abdominopagus
Thoracopagus
Xiphopagus
Omphalopagus
Abdominal attachment; common liver

27
29
30
23
23
24
32
27
22
32
28
27
20
25
34
24
32
26
30
21
20
20

N.S.:~
19
22

CANADA

t/
.195,

LAKEONTARIO
~'9~5"9
_ _ ~ _.~, 1949
L~.~'E ~ 1 9 6 0

"1962

,1957

.1963

9 1959

-'2;

.1965

d1954

.1954

VERMONT

J
/

MASSACHUSETTS

91960

59

PENNSYLVANIA

~'~

91959

CONNECTICUT

91948

Fig. 1. Conjoined twin sets by year of birth and residence of mother, upstate New York,
January, 1945, to April, 1965.

64 6

October 1966

Milham

T h e occurrence of these cases by m o n t h

of b i r t h does not a p p e a r to be r a n d o m
( T a b l e I I ) . Sixteen of 22 sets of twins were
b o r n in the three months, April, M a y , a n d
September. F i v e m o n t h s of the y e a r were
w i t h o u t a case birth. I g n o r i n g small differences in length of m o n t h a n d in m o n t h l y
b i r t h rate, the p r o b a b i l i t y t h a t all of 22
cases will fall into seven or fewer of twelve
equally probable categories is quite small (p
= .0045) .6
T h e average c o m b i n e d b i r t h weight of
the 5 sets of twins b o r n in S e p t e m b e r was
8 pounds, 14 ounces, as c o m p a r e d to 5
pounds, 2 ounces, for the A p r i l a n d M a y
births. Since the m e a n of the r e p o r t e d gestation lengths of the twins born in S e p t e m b e r
was 7 weeks longer t h a n t h a t of those born
in A p r i l a n d M a y , there is reason for believing t h a t these 2 groups, which f o r m the
bulk of the series, m a y have been conceived
in the same season of the year. T h e estimated
m o n t h of conception for the 22 sets of twins
is shown in T a b l e I I I . T h e m o n t h of conception was e s t i m a t e d in each case by counting b a c k w a r d in time f r o m the d a t e of birth,
the c a l e n d a r time equivalent of r e p o r t e d
gestation length. Seventeen of 22 sets were
conceived in the 6 months, J u l y t h r o u g h
December. This m a y possibly explain the
u n u s u a l distribution of cases b y b i r t h month.
T h e incidence of these cases over the years
does not a p p e a r to be u n i f o r m (Tables I

I I . C o n j o i n e d twin sets classified b y

m o n t h of b i r t h
Table

Month of birth

January
February
March
April
May
June
July
August
September
October
November
December
Total

a n d I V ) . Six of 22 sets were .born in 1959;

thirteen of 22 were b o r n in t h e four years,
1957 to 1960. T h e statistical significance
achieved in t h e X2 test in T a b l e I V is due
p r i m a r i l y to the 6 sets of conjoined twins
which were b o r n in 1959. This test did not
consider the a d d i t i o n a l fact t h a t five of the
six 1959 sets were b o r n to mothers who
resided an average of 50 miles from one
a n o t h e r in western N e w Y o r k S t a t e (Fig. 1).
This distance was c a l c u l a t e d by t a k i n g the
a r i t h m e t i c m e a n of the ten possible m a p
distances between the five residences of t h e
mothers.
COMMENTS

Because births of less t h a n 20 weeks' gestation a r e e x e m p t from b i r t h r e p o r t i n g in

I I I . C o n j o i n e d twin sets classified by

e s t i m a t e d m o n t h of conception

Table

Month of conception

January
February
March
April
May
June
July
August
September
October
November
December
Total

Table

No. of cases

1
0
1
1
2
0
3
2
3
2
2
5
22

I V . Analysis of incidence d a t a *
Conjoined
twin sets

No. of cases

1
0
0
6
5
1
0
0
5
0
I
3
22

Time period

Total
births

Expected

Observed

1945-1949
710,000
4.28
2
1950-1954
847,568
5.11
2
1955-1959
995,342
6.01
12
1960-June, 1965 1,092,712
6.59
6
Total
3,645,622
21.99
22
X~ = 9.13 p(X~ > 9.13) < . 0 2
*Twenty-two sets of conjoined twins were reported in
a total of 3,645,622 births for an over-all incidence of .6034
cases per I00,000 births. Expected numbers are computed
by multiplying total births by over-all incidence; i.e., for

1945-1949--710,000 X .0634/100,000 = 4.28.

Volume 69 Number 4

upstate New York, it is clear that conjoined

twins will be grossly underreported on birth
records, especially since they show a pror~ounced tendency t o w a r d prematurity. Pott e d has seen only 2 conjoined twin sets in
over 100,000 deliveries at Chicago Lying-in
Hospital since 1931, giving a crude incidence
rate of 1 in 50,000 births. The 22 cases in
this study occurred in a population of
3,645,622 births for an incidence rate of
1 in 165,710 births. By contrast, the six
1959 N e w York cases occurred in 201,192
births for an incidence rate of 1 in 33,532
births, which is high in spite of probable
underreporting.
Since ascertainment of these cases was
solely through vital records, it is difficult
to see how the high 1959 incidence rate
could be the result of a reporting artifact.
This is especially true since the 1959 cases
show geographic as well as temporal aggregation. T h e geographic distribution of cases
in other years was not remarkable. It is
also difficult to see how ascertainment bias
could account for the m a r k e d sex ratio shift
or for the strong maternal history of previous
fetal loss. However, before a firm claim for
either a seasonal effect on incidence or for
time-space clustering can be made, a larger
case series must be analyzed.
SUMMARY

T w e n t y - t w o sets of symmetrically conjoined twins have been identified in 20 years

Epidemiology o[ conjoined twins

64 7

of birth records of the N e w York State

D e p a r t m e n t of Health. Analyses of these
records reveal:
1. A marked predominance of femMes
(20 of 22 sets).
2. An excess of stillbirths (15 of 22 sets).
3. A marked tendency toward prematurity
and low combined birth weight.
4. A maternal history of greater than
expected fetal loss.
5. A possible seasonal effect on incidence,
and possible time-space clustering of cases.
I wish to acknowledge statistical advice given
by Mr. Abraham Kaminsky.
REFERENCES

1. Gittelsohn, A. M., and Milham, S.: Declining

incidence of central nervous system anomalies
in New York State, Brit. J. Prev. & Social
Med. 16: 153, 1962.
2. Gittelsohn, A. M., and Milham, S.: Observations on twinning in New York State, Brit.
J. Prey. & Social Med. 19: 8, 1965.
3. Milham, S.: Congenital malformation surveillance system based on vital records, Pub.
Health Rep. 78: 448, 1963.
4. Koop, C. E.: The successful separation of
pyopagus twins, Surgery 49: 271, 1961.
5. F6rster, A.: Die Missbildungen des Menschen,
systematisch Dargestellt. Jena, 1865, F. Mauke.
6. Feller, W.: An introduction to probability
theory and its applications, ed. 2, New York,
1957, John Wiley & Sons, vol. 1, p. 58.
7. Potter, E. L.: Pathology of the fetus and infant, Chicago, 1961, Yearbook Medical Publishers, Inc., p. 217.