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Cone-shaped epiphyses in Japanese children.

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AMERICAN JOURNAL O F PHYSICAL ANTHROPOLOGY 57:117-121(1982)
Cone-Shaped Epiphysas in Japanese Children
RONALD K. WETHERINGTON
Department af Anthropology, Southern Methodist Uniuersity, Dallas, Texas
75275
KEY WORDS
Cones, Dysplasia, Radiographs, Hand-wrist, Japanese
ABSTRACT
Cone-shaped epiphyses (CSE) are reported in left-hand
radiographs of a sample of 1,399 otherwise normal Japanese children aged 5-11
years. CSE occurred in only three centers: distal thumb, mid-index finger, and
mid-fifth finger. The overall frequency of 23% shows a female bias (32%to l6%),
which disappears when mid-5 cones are excluded. Chi-squared analyses show
significant gender bias for mid-5 cones alone or in combination with the other
phalanges, and show significant center associations: CSE in distal-1 andlor mid-2
are more common in association with mid-5 cones than they are without mid-5
cones. The CSE female bias in mid-5 is essentially unchanged across agecohorts
when bone-age is controlled for precocity, and no significant trends in CSE frequency with age are noted for either gender.
Among the more frequent osseous anomalies
occurring in the hand is that commonly referred to as cone-shaped epiphysis (CSE).The
reciprocal dysplastic shaping of epiphysis and
diaphysis creates a “cone-and-crater” radiographic image and generally results in premature fusion of the two osseous centers. Studies
that report CSE in the middle segment of the
fifth finger (MP-5),where it appears ta be most
common, show substantis? variation in population frequencies and generally higher frequencies in females.
Although CSE occurs less frequently in
other phalangeal segments, its association in
these sites with its occurrence in MP-5, and
with gender, have not been fully explored. Furthermore, the possibility that CSE may be reversible as maturation proceeds, thus affecting observed frequencies, has yet to be
established.
In the study reported here, the occurrence of
CSE was tabulated in a large sample of Japanese children by sex, age, and osseous center in
order to determine (1)whether a gender and/or
age bias for CSE is evident, and (2)whether
significant associations exist among osseous
centers displaying CSE.
METHODS AND MATERIALS
The sample consists of left-hand radiographs
of 1,399 native Japanese children from Hiroshima, ages 5 through 11 years. The films were
taken during 1958-59 for the Child Health
0002-948318215701-0117$02.00 0 1982 ALAN R. LISS, INC.
Survey of the Atomic Bomb Casualty Commission. They represent a control group for a
study of the effects of consanguineous marriage on child growth and development. The
sample includes 742 males and 657 females.
The age and sex distribution of the sample
(with the exception of the oldest cohort of 4
males and 6 females) is presented in Figure 1.
Cone epiphyses were recorded as a single
class of phenomena, but included two variants:
the more common central cone-and-crater (Fig.
2) and a less common radial dysplasia (Fig. 3).
In the latter, the reciprocal diaphyseal-epiphyseal shaping occurs on the radial side of the
segment. In both forms, brachyphalangia is
common along with notching at the distal diaphyseal margin, although these were separately recorded only when they occurred without cones. In radial cones the notching is
radial, usually contributing to marked clinodactyly as the condition progresses. Premature epiphyseal union is common in both forms
and was tabulated when any portion of the
epiphyseal margin was fused with the
diaphysis.
RESULTS
Of the 1,399 children, CSE is present in 326,
or 23% of the sample. Of these, 118 are males
and 208 are females. By sex, then, 16% of the
males and 32% of the females are affected-a
female:maleratio of 1.76:l (Table 1).
Received June 8. 1981; accepted September 1 , 1981.
R.K. WETHERINGTON
118
MALES
FEMALES
123128
117122
111116
105110
-g
-
99
104
In
9398
U
a
3
2 8;i
(3
u
(3
8186
-
75
80
6974
-
63
68
1
Fig. 1. Age and sex distribution of sample. Shaded region represents those affected. Age Cohort 129-134 months
(4 males and 6 females, 3 and 2 affected, respectively) is not shown.
Fig. 2. Representative radiographs showing central
coneand-crater development in four children. Initial epiphyseal invasion of metaphysis is shown in .4. premature fusion in D.
Fig. 3. Medial (radial)dysplasia in two children, showing
(A)epiphyseal-metaphyseal remodeling with distal notch.
and (B)resulting clinodactyly. premature fusion, and
brach yphalangy.
CONE-SHAPED EPIPHYSES IN JAPANESE CHILDREN
TABLE 1. Frequency
119
of cone-epiphyses in the sample by sex, age-groups, and affected segments
Affected segment(s1 - _ _
____
DPI MP2 MP5 DP1- DP1- MP2- All Total DP1 Total MP2 Total MP5 Total affected
only only only MP2 MP5 MP5
3
no.
'70 ~no. _ 70
%
no.
%
_ no.
~
Age group
(months)
Males
129-133
123-128
117-122
111-116
105-110
99-104
93-98
87-92
81-86
75-80
69-74
63-68
Total
Percentage
Females
129-133
123-128
117-122
111-116
105-110
99-104
93-98
87-92
81-86
75-80
69-74
63-68
Total
N
~
4
51
101
75
82
67
85
63
63
62
59
3
742
0
0
0
0
2
1
2
3
1
1
1
0
0
20
2.69
6
1
46
1
87
1
70
3
66
1
67
1
66
3
55
0
58
0
46
1
4 6 0
4 4 0
657
Percent age
Total
Percent age
1
1
4
3
1399
1
3
11
10
7
8
7
7
9
7
4
1
0
0
0
0
0
0
0
0
0
0.0
0
0
0
0
1
0
0
1
0
0
0
0
2
0
2
0
0
0
0
0
14
1.88
4
0.53
4
0.53
0
1
4
1
3
1
0
0
0
1
0
1
0
0
0
0
1
0
0
0
0
0
0
0
0
0
0
1
12
18
16
14
26
19
18
15
12
12
0
0
1
0
2
0
1
0
0
0
0
0
0
0
75
1
10.10 0.13
0
0
0
0
1
1
3
3
3
1
0
0
1
0
0
0
0
1
0
1
0
0
0
0
0
0
0
3
1
1
0
0
0
1
0
0
0
0
1
0
0
0
0
0
0
0
2
0
0
0
2 5 0 . 0 0 0
2
3.921
8
7.92
1
6
8.00
2
7
8.542
2
2.991
4
4.712
3
4.760
2
3.170
1 1 . 6 1 0
2
3.390
0
.
0
0
39
0
1
0
1
1
0
0
0
1
2
0
0
6
0
0.0
15
2.28
2
0.30
3
0.45
30
32
2.29
1
250
1
0.07 17.87 0.07
29
2.07
6
0.43
7
0.50
69
4.57
0.0
2.17
0.0
1.43
1.52
0.0
0.0
0.0
1.72
4.35
0.0
0.0
1
14
22
18
18
27
19
21
17
14
12
12
15
16.67
30.43
25.29
25.71
27.27
40.30
28.79
38.18
29.31
30.43
26.09
27.27
6
1
75.00
11.76
18.81
24.00
17.07
16.42
12.94
15.87
17.46
12.90
10.17
3.33
15.90
2
15
23
21
19
28
22
21
17
16
12
12
33.33
32.61
26.44
30.00
28.79
41.79
33.33
38.18
29.31
34.78
26.09
27.27
208
29.68
292
1.07
3
6
19
18
14
11
11
10
11
8
118
195
0.91
4.93
50.00
9.80
13.86
20.00
14.63
14.93
10.59
11.11
15.87
11.29
8.47
3.33
13.07
1.21
1 1 6 . 6 7
3
6.52
5
5.75
5
7.14
4
6.06
2
2.99
3
4.55
3
5.45
2
3.45
2
4.35
0 0 . 0
0 0 . 0
1
175
0.15 23.63
2
5
14
15
12
10
9
7
10
7
5
1
97
9
5.26
12
1.82
0.0
1.96
0.99
2.67
2.44
1.49
2.35
0.0
0.0
0.0
0.0
0.0
31.66
326
20.87
23.30
DP1 = distal phalanx. thumb: MP2 = mid-phalanx. index finger: MP5 = mid-phalanx. fifth finger
In 90% of the affected ( N = 292) the middle
fifth finger (MP-5)was involved, and in 77%
(N = 250) MP-5 was the only affected segment.
In all cases involving MP-5, the frequency in
females is approximately twice that in males,
comprising 67% of all individuals in which
MP-5 is involved and 70% of all individuals
displaying CSE in MP-5 alone.
Among all remaining digital segments, CSE
occurred in only two. The distal phalanx of the
thumb (DP-1) was affected in 69 children
(4.9%) and the middle phalanx of the index
finger (MP-2) was affected in 15 (1.1%).In
neither segment is the difference in frequency
by gender significant by sign test. The association of CSE with gender when MP-5is involved
is significant. The chi-squared value for the
association of gender with CSE in MP-5 alone is
45.03; for the gender association with all CSE
cases involving MP-5 (including MP-5 alone),
x 2 = 37.96; and for gender with total CSE (including cases where MP-5 is not involved) x2 =
29.99. Hence, CSE has a distinct gender bias
where MP-5 is involved (d.f. = 1, p < 0.001),
and no bias when MP-5 is not involved.
To further test the association, age-cohort
analysis was performed to determine whether
the gender bias of CSE is stable throughout
the sample. As is apparent in Figure 1, males
outnumber females in almost all cohorts. Only
in the youngest and in the oldest (129-133
months) age cohorts is the female sample
larger, yet females outnumber males in all
cohorts in the number of affected. Sign tests
by 6-month cohort reveal significant differences favoring females for ages 87-104
months (p < 0.005) and for ages 63-80 and
117-128 months (p < 0.05), but no significant
differences for the 105-116 month cohorts.
Since female precocity in development extends to hand growth, unadjusted age-cohort
comparisons are not satisfactory if CSE is a
function of skeletal development - as it indeed
is for ages at which secondary ossification
120
R.K. WETHERINGTON
centers are becoming radio-opaque. Consequently, agecohort comparisons were made
using age-adjusted mean skeletal maturity
scores derived from skeletal age analysis. This
analysis (Wetherington, 1961)involved assigning maturity scores for each bone center and
calculating a total maturity score for the individual. Mean scores by month of age and by
six-month cohort were derived for the total
sample. Comparisons of these scores between
sexes thus allow measurement of female precocity in terms of chronological age. The male
sample, matched with the female by maturity
score, results in a difference of about 1.5 years
during the age range considered here. The adjustment truncates the top three age cohorts for
males, and sign tests show significant gender
differences (p < 0.05) for all but the earliest age
cohort. Hence, when matched for bone age, the
sample shows even stronger CSE-gender
association for MP-5 involvement.
Having established that a gender bias for
CSE exists across age-cohorts, it was next important to test the stability of CSE within
gender across the age range. In lieu of longitudinal data to indicate the progress of CSE, a
comparison was made across age cohorts of the
frequency of CSE involving MP-5. Applying
the chi-squared statistic for goodness of fit
among cohorts from 63 through 128 months
(d.f. = lo), neither the males (x’= 7.05)nor the
females (x’ = 3.87) show any significant trend
with age.
The lower frequency of CSE among males in
the first two cohorts (i.e., ages 5.5-6 years) is
due to the later appearance of epiphyses in
males. This relative delay in growth likewise
apparently delays the appearance of CSE. The
earliest recognized affect in males occurs at
age 64 months (1case), while 3 cases were observed in females at age 63 months. Through
70 months only 2 males display CSE, while 17
females are affected.
At least through 10.5 years, then, neither
males nor females demonstrate any reversible
tendency in CSE. In females, epiphyseal fusion
is often precocious: 17% of the MP-5 cases over
105 months show epiphyseal union, while no
union is present in any of the male cases.
Finally, in order to determine whether cone
epiphyses in the three observed segments are
associated, chi-squared tests were again applied. The strong gender bias for CSE involving MP-5, and the lack of such bias when MP-5
is not involved, suggests that no association
exists between MP-5 and either DP-1 or MP-2.
However, when the Occurrence of CSE in DP-1
(and not simultaneously in MP-5) is compared
with its co-occurrence in DP-1 and MP-5, the
association is significant at the 0.05 level in
females (x’= 4.05) but not in males (x’= 0.12).
Similarly, the test for association of CSE in
MP-2 and in MP-5 is significant at the 0.001
level for both males (x’= 50.42) and females (x’
= 16.12). On the other hand, association of
CSE in DP-1 with CSE in MP-2 is not significant for either sex. Hence, cone epiphyses in
either DP-1 or MP-2 or both are significantly
more common in association with MP-5 cones
than without such association.
DISCUSSION
Dysplastic modeling of epiphyses and adjacent metaphyseal margins of the diaphyses
probably occurs sporadically in all populations
of clinically normal individuals (Giedion, 1965;
Greulich, 1970, 1973; Hertzog, 1967; Hertzog
et al., 1968; Rondon de Iturriza and Tanner,
19691, with higher frequencies in Asians and
populations of Mongoloid ancestry.
Although numerous forms of the condition
have been recognized (Giedion, 1967; Rondon
de Iturriza and Tanner, 1969), the term “coneshaped epiphysis” has been its general
designation. While all phalangeal segments are
susceptible to the anomaly in both the hand
and foot, its more common occurrence is in the
middle fifth phalanx of the hand, where it is
variously associated with brachyphalangia,
clinodactyly, and distal diaphyseal notching
(“pseudoepiphysis”)(Garn et al, 1967, 1972a,b;
Greulich, 1973; Hertzog, 1967; Buschang and
Malina, 1980).
While the patterns of dysmorphogenesis involving these anomalies in MP-5 are apparently different in clinically normal individuals
than in specific disease syndromes (cf.Garn et
al., 1972a; Greulich, 1970, 1973),there is considerable variation within otherwise normal individuals. In the present study, CSE does not
apparently involve early timing of epiphyseal
ossification, but does lead to premature diaphyseal union. The Fels longitudinal series,
however, revealed precocious appearance of
both CSE and brachyphalangia-5 as well as
delayed epiphyseal fusion (Hertzog et al.,
1968).
What appears rather consistent, however, is
that in clinically normal samples, both CSE in
general and particularly CSE in MP-5 are most
common where Asian ancestry is strongest
and, where CSE affects MP-5, either alone or in
combination, females have significantly higher
cone frequencies than do males.
CONE-SHAPED EPIPHYSES IN JAPANESE CHILDREN
Hence, our overall frequency of 21% (13%
males, 30% females) compares with other
native-born Japanese samples: Pryde and
Kitabatake (in Greulich, 1973) report 17%,
14%, and 21%, respectively, and Eyman (in
Hertzog et al., 1968) reports three samples
from Nagasaki and Hiroshima with overall frequencies of 15-21% (8-13% males, 22-29%
females). Greulich (1973) reports similar frequencies in American-born Japanese (21%,
1370, and 29%, respectively), with considerably lower frequencies among several
American Indian groups (approximately 8%,
6%, and 9%). Comparable frequencies of coma
bined CSE and brachymesophalangia of MP-5
are reported for a Oaxacan Ladino group
(Buschang and Malina, 1980), while frequencies of mid-5 cones in 6 Central American
samples range from 5% in Guatemala, with the
most predominant indigenous contribution, to
2% in Nicaragua (Hertzog et al., 1968).By contrast, populations with no significant Asian
ancestry generally show frequencies under 1YO.
In almost every sample, regardless of
ancestry, females significantly outnumber
males in frequencies of CSE in MP-5. Correspondingly, cone epiphyses in other
phalangeal segments show no gender association when MP-5 is not also involved, and tend
to occur most frequently when in combination
with MP-5 cones.
ACKNOWLEDGMENTS
The data in this report were collected as one
aspect of the Child Health Survey sponsored
by the U.S. Atomic Energy Commission, the
Rockefeller Foundation, the Association for
Aid to Crippled Children, and the National Institute of Dental Research (USPHS). The
Atomic Bomb Casualty Commission and the
121
Japanese National Institute of Health contributed data, facilities, and personnel. The support of the Japan Science Council’s Committee
on Genetics is also gratefully acknowledged, as
is the material support of James V. Neel.
LITERATURE CITED
Buschang, PH, and Malina, RM (1980) Brachymesophalangia-V in five samples of children: a descriptive and
methodological study. Am. J. Phys. Anthrop. 53:189-195.
Garn, SM, Fels, SL, and Israel, H (1967) Brachymesophalangia of five in ten populations. Am J. Phys. Anthrop.
27205-209.
Garn, SM, Gall, JC. and Nagy. J M 11972a) Brachymesophalangia-5 without cone-epiphysis mid-5 in Down’s Syndrome. Am. J. Phys. Anthrop. 36.253-255,
Garn. SM, Poznanski, AK, Nagy, JM, and McCann. MB
(1972b) Independence of brachymesophalangia-5 from
brachymesophalangia-5 with cone mid-5. Am. J. Phys.
Anthrop. 36.295-298.
Giedion, A (1965) Cone-shaped epiphyses (C.S.E.). Ann.
Radiol. (Fr)8:135-145.
Giedion. A (1967) Coneshaped epiphyses of the hands and
their diagnostic value: The tricho-rhino-phalangal syndrome. Ann. Radiol. (Fr)10:322.
Greulich, WW 11970) Incidence of dysplasia of the middle
phalanx of the fifth finger in normal Japanese, in some
American Indian groups, and in Caucasians with Down’s
Syndrome. In N Kretchmer and DN Walcher (eds):Environmental Influence on Genetic Expression. Washington, D.C.: U S . Government Printing Office, pp. 91-105.
Greulich, WW (1973)A comparison of the dysplastic middle
phalanx of the fifth finger in mentally normal Caucasians,
Mongoloids, and Negroes with that of individuals of the
same racial groups who have Down’s Syndrome. Am. J.
Roent. 118:259-281.
Hertzog, K P 11967) Shortened fifth middle phalanges. Am.
J. Phys. Anthrop. 27:113-118.
Hertzog, KP. Garn,SM. and Church, SF 119681 Cone-shaped
epiphyses in the hand. Invest. Radiol. 3:433-441.
Rondon de Iturriza. J. and Tanner, J M (1969)Cone-shaped
epiphyses and other minor anomalies in the hands of
normal British children. J. Pediat. 75.965-272.
Wetherington, RK (1961) An alternate method of assessing skeletal maturity in the hand and wrist. Pap., Mich.
Acad. Sci., Arts, Ltrs.. 46:419-433.
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