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Carpal fusions in children of four South African populations.

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Carpal Fusions in Children of Four South African
Populations
ERROL L E V I N E
D e p a r t m e n t of Dingnostic Rndiology a n d D e p a r t m e n t of A?iritomy,
U ? ~ i u r r s i tof
y the W i t w a t e r s r a n d , J o h a n n e s b u r g , Soutlz Africri
KEY WORDS Carpal fusions . Hand-wrist X-rays . Children .
South Africa . Genetic differences among populations.
ABSTRACT
The incidence of carpal fusions was determined from radiographs of both hands of 2,250 Pretoria schoolchildren aged 72 to 191 months.
The group included White, Negro, Coloured and Indian subjects.
Of the many possible varieties of carpal fusion, only lunato-triquetral fusion
and capitate-hamate fusion were encountered.
No examples of lunato-triquetral fusion were encountered i n children less
than ten years of age. I n Pretoria Negroes aged 10 to 15 years, the incidence
of lunato-triquetral synostosis is 4.5796, while Pretoria Coloureds show a n
incidence of 1.51 % . This type of fusion was not noted i n the White or Indian
populations. Two examples of capitate-hamate synostosis were encountered i n
Pretoria Negroes. The available evidence suggests that these two varieties of
carpal fusion may be added to the list of discrete traits varying in frequency
from population to population and probably reflecting different underlying
gene frequencies.
No significant sex difference in incidence of lunato-triquetral fusion was
noted in the present study. Lunato-triquetral fusion is about twice as commonly
bilateral as unilateral i n occurrence.
By carpal fusion is meant the cohesion
of two or more normally adjacent, but
separate elements of the carpus. Fusions
have been encountered in almost every
possible combination in the carpus and
the recorded varieties of fusion have been
reviewed by O’Rahilly (‘53, ’57). The present study has been carried out with the
objective of determining the nature and
incidence of carpal synostoses in children
belonging to the four main population
groups of South Africa.
M A T E R I A L S AND M E T H O D S
During the years 1962 to 1965, the National Nutrition Research Institute of the
Council for Scientific and Industrial Research (CSIR) carried out an extensive
cross-sectional nutritional status survey
on the school children of Pretoria. The
survey included the four main population
groups of the Republic of South Africa
namely, Southern African Bantu-speaking
Negroes, Whites (Caucasoids of European
extraction), Indians or Asians (descendants of immigrants from India), and ColAM. J. PHYS.ANTHROP.,37: 75-84.
oureds (persons of mixed extraction).
Anthropometric, clinical, biochemical, haematological, dietetic, radiological and socio-economic data were collected for a
total of 2,250 children aged six to fifteen
years. The statistical planning of the survey has been described by Fellingham
(‘66).
Radiographs of both hands of 2,250 Pretoria children were used in the present
study. The children included 808 Whites
(413 males, 395 females), 585 Negroes
(288 males, 297 females), 450 Coloureds
(225 males, 225 females), 407 Indians (202
males, 205 females). They ranged in age
from 72 to 191 months.
The children were grouped according
to age, sex and population group and the
frequency of carpal fusions in each group
was determined. Skeletal age determinations were made by means of the GreulichI This paper is based on part of a thesis submitted
for the degree of Doctor of Philosophy at the University
of the Witwatersrand i n 1969. The study was supported i n part by the National Nutrition Research
Institute of the Council for Scieiitific and Industrial
Research, Pretoria, South Africa.
75
76
ERROL LEVINE
Fig. 1 Early stage of lunato-triquetral fusion i n a Negro m a l e aged 161 months. There
elongation of the triquetral a n d the two bones a r e i n close apposition.
IS
Pyle Radiographic Atlas of the Skeletal
Development of the Hand and Wrist ('59).
1. Lunato-triqzietral fusion in
Pretoria children
FINDINGS
I n all, 22 Pretoria children showed the
presence of lunato-triquetral fusion in
either one or both wrists.
Serial radiographic studies by other
workers (Silverman, '55; Dean and Jones,
'59; Hughes and Tanner, '66) have shown
Only two varieties of carpal fusion have
been detected in the present study, namel y fusion of the lunate and triquetral and
fusion of the capitate and hamate.
CARPAL FUSIONS I N SOUTH AFRICAN C H I L D R E N
77
Fig. 2 Lunato-triquetral fusion in a Negro male aged 138 months. There is a line of
increased density at the site offusion of the two centres.
that fusion of the two centres progresses
with increasing age. Figure 1 shows a n
early stage of lunato-triquetral synostosis
in a Pretoria Negro male aged 161
months. There is close apposition of the
adjacent surfaces of the two centres and
the radiological appearance resembles
that of a pseudarthrosis. Figure 2 shows
a somewhat later stage of fusion with a
dense line at the site of junction of the
two centres in another child. Figure 3
shows complete synostosis of the two centres in a Negro female.
The incidence of fusion i n the different
population groups is shown i n table 1.
As no example was encountered under
ten years of age, the incidence has been
determined for children aged 10 to 15
years. Fusion was noted in 4.57% of
Negro children and i n 1.51% of Coloured
78
ERROL LEVINE
Fig. 3
Complete lunato-triquetral fusion in a Negro female aged 133 months
children. No examples of fusion were
encountered in the White or Indian
groups.
Negro and White children differ significantly i n incidence of lunato-triquetral fusion, in that the observed differences between the percentage incidences
in the two groups exceeds three times
the standard error of the difference. No
significant difference in incidence of this
variant exists between Coloured and
White children and Negro and Coloured
children.
Hence, on the basis of the data, it may
79
CARPAL FUSIONS IN SOUTH AFRICAN CHILDREN
TABLE 1
Incidence of liinato-triquetral f u s i o n in Pretoria children aged 10 to 15 gears
Population
group
Total
number
Individuals with
fusion (male
and female)
Of
White
Negro
Coloured
Indian
children
No.
%
445
394
265
240
0
18
4
0
0.00
4.57
1.51
0.00
Total
number
of
inales
Males with
No.
%
Total
number
of
females
0
7
1
0
0.00
3.48
0.75
0.00
219
193
132
120
fusion
226
201
133
120
Females with
fusion
No.
5
0
11
3
0
0.00
5.70
2.27
0.00
TABLE 2
The frequency of unilatercil a n d bilateral lirnnto-triquetral fusioii in Pretoria
Negro a n d Colorired children
Negro
Laterality of lunatotriquetral fusion
NO.
Coloured
cc
Total
No
7%
No
%
~~
Bilateral fusion
Unilateral fusion
13
72.2
2
50.0
15
68.2
Right carpus
Left carpus
3
2
16.7
11.1
2
0
50.0
0.0
5
2
22.7
9.1
Total
5
27.8
2
50.0
7
31.8
be concluded that Negro children manifest the 0s lunato-triquetrum significantly
more frequently than White children.
The data in table 1 suggest that lunatotriquetral fusion is more common in Negro
and Coloured females than in Negro and
Coloured males. However, in both Pretoria Negroes and Coloureds the sex difference in incidence of the variant is
not significant, the observed difference
between the percentages in each instance
being less than three times the standard
error of the difference. The number of
cases in the present study is clearly too
small to pennit any conclusion regarding
the significance of the observed sex difference in the incidence of fusion.
Table 2 shows the frequency of unilateral and bilateral distribution of the
0 s lunato-triquetrum in Pretoria children.
In 15 of the 22 children with this variant
(68.2%), fusion was present in both
wrists. In seven children (31.8%) fusion
occurred on one side only. In five of the
seven instances of unilateral occurrence,
fusion was on the right and in two instances it was on the left side. Because
of the small size of the sample, these
data were not subjected to statistical analysis. Furthermore, the incidence of bilateral occurrence of lunato-triquetral
synostosis in this series of children may,
in fact, be higher. Some children with
unilateral fusion at the time the radiographs were taken may subsequently have
developed fusion in the opposite carpus.
However, the available data suggest that
fusion is about twice as commonly bilateral as unilateral.
The data in the present study show a
general trend for synostosis of the two
ossific centres to commence in the circumpubertal period. No instances of fusion were detected in children between
six and nine years of age. The median
skeletal age of the 22 children exhibiting
fusion is 154 months, the range being
87 to 204 months. The median chronological age of the 22 children is 167
months with a range of 126 to 191 months.
2. Fusion of the capitate and hnmate
in Pretoria children
Fusion of the capitate and hamate is
less common than fusion of the lunate
and triquetral. In the present study two
examples of bilateral capitate-hamate fusion were encountered in 585 Negro children -an
incidence of 0.34%. This variety of fusion was not detected in the
White, Coloured or Indian population
groups.
Figure 4 shows partial fusion of the
80
ERROL LEVINE
Fig. 4 Partial capitate-hamate fusion in a Negro female aged 189 months. There is complete fusion of the lunate and triquetral
capitate and hamate in a Negro female
aged 189 months. The fusion is bilateral
and the child has, in addition, bilateral
lunato-triquetral synostosis. The other
example of fusion was noted in a Negro
female aged 133 months. The fusion i n
this iiistance is likewise bilateral and incomplete.
DISCUSSION
Carpal fusions may occur as isolated
anatomical variants or they may be associated with generalised skeletal disorders
(Poznanski and Holt, ’71). Generally the
isolated carpal fusions involve bones in
the same row, while the syndrome-associated fusions not infrequently involve co-
hesion of adjacent carpals in the proximal
and distal rows (Poznanski and Holt, ’71).
It is considered that the term “fusion”
applied to the isolated anatomical variant
is a misnomer (O’Rahilly, ’57; Cockshott,
’59). According to O’Rahilly (’57), the
first step i n the development of carpal
fusions is either failure of separation of
adjacent cartilages in the embryo or possibly fusion of cartilaginous anlagen at an
early stage in embryonic development.
Ossification begins from two centres in a
single cartilaginous primordium. The existence of a single cartilage, where usually there are two, then inevitably leads
to the subsequent fusion of the two ossific
centres concerned. Thus the variant may
be regarded not as a “fusion,” but rather
81
CARPAL FUSIONS IN SOUTH AFRICAN CHILDREN
TABLE 3
The incidence of the 0s lu n a t o - t r i q u m x m zn dzflerext populations
Individuals with fusion
Population
Size of series
Negroid
West African
Negroes
Baganda
Baganda
Wadigo
Yoruba
American
Negroes
South African
Negroes
Caucasoid
Scandinavian
Whites
German Whites
American Whites
American Whites
South African
Indians
South African
Whites
No.
%
3
40
5
3.75
5.88
2.12
Dean and Jones ('59)
24
14
74
6.67
1.03
8.01
Davies ('59)
Mackay ('52)
Cockshott ('59)
7543 subjects
119
1.58
Garn et al. ('71)
394 children
18
4.57
Present study
2100 adults
5000 subjects
743 subjects
11663 subjects
3
4
12
0.14
0.08
0.13
0.10
Lonnerblad ('36)
Arens ('50)
ORahilly ('53)
Garn et al. ('71)
240 children
0
0.00
Present study
445 children
0
0.00
Present study
80 children
680 adults
236 children
360 children
and adults
1360 children
923 adults
as a non-separation of cartilaginous carpals.
There is evidence to suggest a genetic
basis for certain types of carpal synostosis. There is now abundant evidence that
lunato-triquetral fusion may be regarded
as a familial trait (Dean and Jones, '59;
Garn et al., '71). Cockshott ('59) has encountered capitate-hamate fusion in identical twins. Carpal fusions are frequently
associated with hereditary skeletal disorders including symphalangism (Harle
and Stevenson, '67; Geelhoed et al., '69),
the Ellis-van Creveld syndrome (McKusick,
'66) and the hand-foot-uterus syndrome
(Poznanski et al., '70; Stern et al., '70).
Certain types of carpal fusion vary in
incidence from population to population.
Lunato-triquetral synostosis is apparently
rare in Mongoloid peoples. Only one example was found in 1,400 Japanese children, an incidence of 0.071 % (Wetherington, '61). The present study and other
studies have shown that lunato-triquetral
fusion is undoubtedly more common in
Negroid than in Caucasoid subjects (table 3). The observed frequency in the
African Negro is higher than that in the
American Negro. The writer has been
unable to find any record of lunato-trique-
1
Authors
Smitham ('48)
tral fusion in Indian subjects. No example
of fusion was encountered in Pretoria
Indian children. In Pretoria Coloureds,
the incidence of lunato-triquetral synostcsis is higher than in Whites, but lower
than in Negroes. Since the Coloureds
include a fairly high incidence of Negroid
genes, and the Indians are Caucasoid, the
findings in the present study may be interpreted as suggesting strongly that lunato-triquetral fusion is genetically determined with a higher frequency of allele(s)
in Negroid than in Caucasoid subjects.
Capitate-hamate fusion also appears to
be more common in Negroid than in Caucasoid subjects. It is the second most
common variety of carpal synostosis in the
African Negro (Smitham, '48; Beresowski
and Lundie, '52; Mackay, '52; Dean and
Jones, '59; Cockshott, '59). Capitatehamate fusion appears to be rare in White
subjects. Isolated instances in Whites have
been mentioned in the literature (White,
'44), but Kohler ('56), in his radiological
text, does not mention having encountered an example.
It seems appropriate that these two varieties of carpal fusion should be added
to the list of discrete traits differing in
incidence from population to population
82
ERROL LEVINE
and probably reflecting different underlying gene frequencies.
In the present study, lunato-triquetral
synostosis appears to be more common in
females, while in the Yorubas of Nigeria
(Cockshott, '59) and i n West African Negroes (Smitham, '48), there is a tendency
for the variant to be more common in
males. However, in none of the groups is
the sex difference statistically significant.
Of 119 instances of fusion encountered
in 7,543 American Negroes, 86 instances
occurred i n females (Garn et al., '71).
A female preponderance for the trait in
American Negroes thus seems firmly established.
Very little information is available regarding the relative frequency of unilateral and bilateral lunato-triquetral fusion.
This is because in some studies only one
wrist has been X-rayed. Even when both
wrists have been X-rayed, authors sometimes have not documented whether fusion was unilateral or bilateral. The finding in the present study that fusion is
about twice as commonly bilateral as unilateral is confirmed by other studies. Of
his 74 cases, Cockshott ('59) found bilateral fusion in 47 (63.5%), right-sided fusion in 12 (16.2%) and left-sided fusion i n
15 (20.3%). In all five cases of Gean and
Jones ('59) synostosis was bilateral. Smitha m ('48) found that the variant was most
frequently bilateral, but when unilateral,
it occurred only i n the right carpus in
his Negro subjects.
Very few data dealing with the age at
which fusion of the lunate and triquetral
ossific centres occurs have been published.
Table 3 indicates that the maximuin incidence of synostosis is found in adult Negroes. In the present study no example
of fusion was encountered below ten years
of age and a general trend was noted for
synostosis to become apparent in the circumpubertal period. However, it seems
that the chronological age at which fusion
first becomes evident radiologically is variable. Examples of fusion have been noted
at 102 months in a Japanese boy (Wetherington, '61), and at six years in a
Yoruba girl (Cockshott, '59). Since synostosis is progressive with the maturation
of the wrist (Silverman, '55; Dean and
Jones, '59; Hughes and Tanner, '66), i t
would seem likely that the onset of fusion
may correlate better with skeletal age
than with chronological age.
ACKNOWLEDGMENTS
I wish to thank Professor Phillip V. Tobias, Head of the Anatomy Department,
University of the Witwatersrand, for his
considerable help in the production of this
paper; Dr. J. J. Theron, former Director
of the National Nutrition Research Institute, Pretoria, and Dr. J. F. Potgieter,
former Head of the Field Studies Division
of the National Nutrition Research Institute, for allowing me freely to study their
collection of radiographs.
LITERATURE CITED
Arens, W. 1950 Uber die angeborene synostose
zwischen dem 0 s lunatum und dem 0 s triquetrum. Fortschr. Rontgenstr., 73: 772 (quoted
by O'Rahilly, 1953).
Beresowski, A., and J. K. Lundie 1952 Sequence in the time of ossification of the carpal
bones in 705 African children from birth to
6 years of age. S. Afr. J. Med. Sci., 1 7 : 25-31.
Cockshott, W. P. 1959 Carpal anomalies amongst
Yorubas. W. Afr. Med. J.,8: 1 8 5 1 9 0 .
Davies, A . G . M. 1959 Unpublished data quoted
by Cockshott, 1959.
Dean, R. F. A., and P. R. M. Jones 1959 Fusion
of triquetral and lunate bones shown in serial
radiographs. Am. J . Phys. Anthrop., 17: 279281.
Fellingham, S. A. 1966 Statistical planning of
the nutrition status surveys on Pretoria schoolchildren. S. Afr. Med. J., 2: 228-234.
Garn, S . M., A . Frisancho, A. K. Poznanski, J.
Schweitzer a n d M. B. McCann 1971 Analysis of triquetral-lunate fusion. Am. J . Phys.
Anthrop., 34: 4 3 1 4 3 4 .
Geelhoed, G., J. V . Nee1 and R. T. Davidson 1969
Symphalangism and tarsal coalitions: hereditary syndrome; report on two families. J. Bone
Jt. Surg., 51 B: 278-289.
Greulich, W. W., a n d S. I. Pyle 1959 Radiographic Atlas of Skeletal Development of the
Hand and Wrist. Second edition. Stanford University Press, Stanford, California, pp, 1-256.
Harle, T. S. , a n d J. R. Stevenson 1967 Hereditary symphalangism associated with carpal and
tarsal fusions. Radiology, 89: 91-94.
Hughes, P. C. R., a n d J. M. Tanner 1966 Development of carpal bone fusion as seen i n
serial radiographs. Br. J . Radiol., 39: 943-949.
Kohler, A. 1956 Borderlands of the Normal
a n d Early Pathologic in Skeletal Roentgenology.
Revised by E. A . Zimmer. Grune and Stratton,
New York, pp, 1-723.
Lonnerblad, L. 1936 Uber zwei anomalien im
carpus. Acta Radiol., 16: 682 (quoted by Minnaar, 1952).
Mackay, l3. H. 1952 Skeletal maturation i n the
hand: a study of development in East African
CARPAL FUSIONS IN SOUTH AFRICAN CHILDREN
children. Trans. R. SOC.Trop. Med. Hyg., 46:
135-150.
McKusick, V. A. 1966 Heritable Disorders of
Connective Tissue. The C. V. Mosby Company,
St. Louis, pp. 1-499.
Minnaar, A . B. D. 1952 Congenital fusion of
the lunate and triquetral bones in the South
African Bantu. J. Bone Jt. Surg., 348: 45-48.
O’Rahilly, R. 1953 Epitriquetmm, hwotriquetrum and 1UnatOtriqUetrUm. Acts Radial., 39:
401410.
957 Developmental deviations in the
carpus and tarsus. Clin. Orthop., 10: 9-18.
Poznanski, A. K., and J. F. Holt 1971 The
carpals i n congenital malformation syndromes.
Am. J. Roentg., 112: 44-59,
Poznanski, A. K., A. M. Stern and J . C. Gall, Jr.
1970 Radiographic findings in the hand-footuterus syndrome (HFUS). Radiology, 96: 129134.
83
Silverman, F. N. 1955 A note on the 0 s lunatotriquetrum. Am. J. Phys. Anthrop., 13: 143145.
Smitham, J. H. 1948 Some observations on
certain congenital abnormalities of the hand
in African natives. Br. J . Radiol., 21: 513-518.
Stern, A, M., J . c. G&, J ~ . ,B. L. perry, C. W.
Stimson, L. R. Weitkamp and A . K. Poznan1970 Hand-foot-uterus syndrome; new
hereditary disorder characterised by hand and
foot dysplasia, dermatoglyphic abnormalities,
and partial duplication of female genital tract.
J . Pediat., 77: 109-116.
Wetherington, R. K. 19C1 A note on the fusion
of the lunate and triquetral centres. Am. J .
Phys. Anthrop., 19: 251-253.
White, E. H. 1944 Bilateral congenital fusion
of carpal capitate and hamate. Am. J. Roentg.,
52: 4 0 6 4 0 8 .
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