infantile idiopathic scoliosis: can it be prevented?

infantile idiopathic scoliosis: can it be prevented?

INFANTILE
IDIOPATHIC
SCOLIOSIS:
MICHAEL
From
J.
the Edinburgh
CAN
IT BE
PREVENTED?
McMASTER
Scoliosis
Unit
The changing
incidence
of idiopathic
scoliosis
in 672 patients who attended
the Edinburgh
Scoliosis
Clinic between
1968 and 1982 inclusive is reported. Of these patients,
144 had infantile,
51 juvenile and 477
adolescent
idiopathic
scoliosis.
Thirty-seven
of the infantile
curves
progressed
and 107 resolved.
The relative
frequency
of both progressive
and resolving
infantile
idiopathic
scoliosis
(given as a percentage
of the total
number of patients
with idiopathic
scoliosis)
declined from 41.75 per cent in the four years from 1968 to 1971,
to four per cent in the three years from 1980 to 1982. It is suggested
that infantile
idiopathic
scoliosis
is a
preventable
deformity
and that the position in which the infant is laid may be a causative
factor.
Idiopathic
scoliosis
is a structural
deformity
without
any
evident
clinical
cause. Depending
on the age of presentation
it has been classified
into three types : infantile
(presenting
from birth to three years),juvenile
(presenting
from 3 to 10 years) and adolescent
(presenting
from 10
years to skeletal
maturity).
Wynne-Davies
(1968) carried
out a family
survey
of all types of idiopathic
scoliosis
seen in Edinburgh.
She found that there was a genetic
tendency
to develop
scoliosis
but this by itself
was
insufficient
: other factors (still unknown)
were necessary
to “trigger
off”
the deformity.
Infantile
idiopathic
scoliosis
differs
from the adolescent
variety
in several
important
ways. Infantile
scoliosis
is more common
in
boys, and the great
majority
of patients
have a left
thoracic
curve.
During
the juvenile
years there
is a
gradualchange
in the clinical pattern until by adolescence
the majority
of patients
are girls and a high proportion
have
a right
thoracic
scoliosis.
The
most
striking
difference,
however,
is that between
52 and 90 per cent
of infantile
curves resolve spontaneously
(Lloyd-Roberts
and Pilcher
1965; Ferreira
and James
1972) whereas
adolescent
curves do not resolve and nearly all progress
(Ponseti
and Friedman
1950 ; James 1954). These findings
suggest
that although
all idiopathic
curves may have a
common
genetic
basis, the unknown
triggering
factors in
infancy
may be very different
from those in adolescence.
Surveys
in North
America
have
confirmed
the
genetic
nature
of idiopathic
scoliosis
(Cowell,
Hall and
MacEwen
1972) but have revealed
major differences
in
the relative
frequencies
of the different
types.
The
Edinburgh
survey showed
that SO per cent of all patients
with idiopathic
scoliosis
had infantile
curves whereas
in
Boston,
USA, only 0.5 per cent of idiopathic
curves were
M. J. McMaster,
Princess
burgh
EHIO
© 1983 British
030l-620X/83/5
612
MD,
Margaret
7ED,
FRCS,
Rose
Consultant
Orthopaedic
Orthopaedic
Hospital,
Surgeon
Fairmilehead,
Scotland.
Editorial
Society
I24 $2.00
of Bone
and
Joint
Surgery
Edin-
infantile
(Riseborough
and Wynne-Davies
1973). Since
the white
populations
of Edinburgh
and Boston
stem
from similar
genetic
stock, the different
frequencies
of
infantile
idiopathic
scoliosis
suggests
that either
some
beneficial
aspect of the environment
of North
America
actively
prevents
its development
or that some adverse
triggering
factor is present
in Great Britain
but absent in
North America.
Since 1972 I have noted a marked
change
in the
number
of patients
presenting
to the Edinburgh
Scoliosis
Clinic
with infantile
idiopathic
scoliosis.
This clinic is
the only referral
point for all patients
with scoliosis
from
a large area of Scotland
and therefore
reflects any change
in the relative
frequencies
of the different
types
of
idiopathic
scoliosis
in this population.
The purpose
of
this paper is to report on this change and comment
on its
possible
significance.
Table I. The number
ofgirls
scoliosis
between
1968 and
and boys
1982
presenting
with
idiopathic
Number
of patients
Male
Female
Total
Infantile
85
59
144
Juvenile
18
33
51
Adolescent
96
381
477
199
473
672
Type
of idiopathic
scoliosis
Total
MATERIALS
AND
METHODS
A retrospective
study was made of all patients
under the
age of 18 years presenting
for the first time with idiopathic
scoliosis
to the Edinburgh
Scoliosis
Clinic
between
January
1968 and December
1982. Only patients
from
our usual referral
area in Scotland,
for which there is no
other
referral
point,
were included.
There
were 672
patients.
They were classified
into three groups depending
on the age of onset of their curve : 144 patients
with
THE
JOURNAL
OF BONE
AND
JOINT
SURGERY
INFANTILE
IDIOPATHIC
SCOLIOSIS:
infantile
idiopathic
scoliosis
(65 of whom
were seen
before
1972 and have been previously
investigated
by
Wynne-Davies,
1975) ; 5 1 patients
with juvenile
idiopathic
scoliosis ; and 477 patients
with
adolescent
idiopathic
scoliosis.
All patients
in the juvenile
and
adolescent
groups
had progressive
curves
of over 20
degrees.
The sex ratio in each of the three groups
is
shown in Table I.
RESULTS
Incidence
of scoliosis.
The yearly incidence
of patients
presenting
with infantile,
juvenile
and adolescent
idiopathic
scoliosis
is shown in Figure 1.
Infantile
(1L4 pcitientsl
20
:i
17
-
16
16
Resolving
16
Progressive
10
CAN
613
IT BE PREVENTED?
resolved and 22(34 percent)
had curves which progressed.
After 1971 there was a steady decline
in the number
of
patients
presenting
with infantile
scoliosis
with only six
patients
presenting
between
1980 and 1982. Of these six
patients,
three
had resolving
curves
and three
had
progressive
curves.
The declining
incidence
of infantile
idiopathic
scoliosis
has therefore
applied
to both progressive and resolving
curves.
Juvenile.
The yearly
incidence
of juvenile
idiopathic
scoliosis
remained
fairly constant
with between
one and
four patients
presenting
each year from 1968 to 1982
(except
from
1974 to 1976 when
there
was a slight
increase
with six patients
presenting
in each year).
Adolescent.
The yearly incidence
of adolescent
idiopathic
scoliosis
increased
from 18 patients
in 1968 to 31 patients
in 1972. Between
1972 and 1979 there
was a fairly
constant
rate with 31 to 34 patients
presenting
each year.
In the years 1980 to 1983 the yearly rate of incidence
again increased
with 38, 43 and 56 patients
presenting
respectively.
22
,7i,;;;i;;
75
68
69
70
71
72
73
76
77
78
79
80
81
1OC
82
go
7
90
8c
Juvenile
(51 patients)
6C
10
o-oAdotescent
6
3
3
68
69
70
I
22
6
6
II
l_2__1._i_l__Lf3
3
I
72
73
75
7h
76
77
78
79
80
‘
1-i
[‘j
71
5
5c
81
A
I
eLC
82
#{163}7
Adolescent
(77
‘-0
2
‘-S
\.
:2C
56
patients)
Infantile
‘.o,
#{163}5
x
50
Aiuvviile
0----0
.-
‘O-’
i_.
I
8
o..
a...o.
k...
8
#{149}
A..
A.#{149}
68
69
70
‘-.,.
A
#{176}“03
A-A
A
71
72
73
1L
75
76
77
78
79
80
81
82
Year of presentation
‘C
38
33
..
32
31
3o
a
33
-
31
Graph
The
18
1968, 47
infantile
frequency
increase
when 90
10
.-
.
i_
-
9_
-
-
Yearof
yearly
incidence
adolescent
VOL.
65-B,
if
i-
9
i-
ii-.
1
frequency
scoliosis
2
frequency
scoliosis
from
of infantile,
juvenile
from 1968 to 1982.
of the
different
1968 to 1982 is shown
and
types
of
in Figure
juvenile
1982.
and
1968 to 1971 the number
of patients
infantile
idiopathic
scoliosis
remained
17 per year. Ofthe 65 patients
diagnosed
years, 43 (66 per cent) had curves which
No. 5. NOVEMBER
1983
presenting
with idiopathic
scoliosis
in
per cent had adolescent
curves and 45 per cent
curves.
There was then a steady decline
in the
of infantile
curves
and
a corresponding
in the frequency
of adolescent
curves until 1982
per cent of the patients
with idiopathic
scoliosis
had adolescent
curves and three per cent infantile
curves.
ofjuvenile
curves between
1968 and 1982
fairly constant
at around eight per cent.
idiopathic
scoliosis.
Of the 144 patients
with
The frequency
1
of patients
presenting
with infantile,
idiopathic
scoliosis
between
1968 and
From
presenting
with
constant
at 16 to
during these four
Infantile.
-
presentation
Fig.
The
relative
idiopathic
2. Of the patients
16
i-
the
relative
idiopathic
20
20
illustrating
adolescent
31
27
.
I
-
Fig.
.-.
remained
Infantile
.
.
.
.
.
.
infantile
idiopathic
scohosis
107 (74 per cent) had curves
which resolved
spontaneously
and 37 (26 per cent) had
curves
which
progressed.
The clinical
details
of these
patienta
are shown in Table II. The ratio of boys to girls
was 3 : 2 and was the same for both progressive
and
resolving
curves.
614
M. J.
McMASTER
One hundred
and twenty-one
infants
(84 per cent)
developed
curves in the first six months
of life ; of these
patients
83 per cent had curves that resolved
and only 17
per cent had curves that progressed.
In contrast,
30 per
cent of the 23 children
whose
scoliosis
was diagnosed
after one year of age had curves that resolved
and 70 per
cent had curves that progressed.
A single structural
scoliosis
occurred
in 138 patients
(96 per cent); 1 30 infants
had thoracic
curves, seven had
thoracolumbar
curves and only one had a lumbar
curve.
Eighty-two
per cent of these single curves were left-sided
and only 18 per cent right-sided.
Seventy-eight
per cent
of single curves resolved
and 22 per cent progressed.
A double structural
scoliosis
affecting
the thoracic
and lumbar
regions
was less common
and occurred
in
only six infants
(four per cent). Double curves were most
frequently
diagnosed
after the first year oflife. They were
equally
distributed
on either
side of the spine and all
progressed,
but more
slowly
than
single
progressive
curves.
Table
II.
Clinical
details
of
144 patients
with
infantile
Sift
of patients
Progressive
curves
Resolving
curves
28(22L.6R)
102(85L,
Total
Percentage
130
90
of curvature
Thoracic
Thoracolumbar
2 ( I L. I R)
5 (4L,
17R)
I R)
7
5
Lumbar
1 (L)
none
I
I
Double
6
none
6
4
24
61
85
59
I3
46
59
41
16
82
98
68
5
18
23
16
16
7
23
16
26
98
124
86
Absent
2
7
9
6
tinknown
9
2
11
8
Se.
Male
Female
Age
at presentatu)?:
Birthto#{244}months
7monthsto
I year
I yearto3years
Plagiocephalv
Present
*3 right
thoracic,
left lumbar.
and
3 left thoracic,
right
r
4
(
‘
Fig. 3
Left-sided
plagiocephaly
lumbar
Plagiocephaly
(Figs 3 and 4) was present
in 124
infants
(86 per cent) and absent in nine (six per cent), all
with resolving
curves ; no clinical
record had been made
in the remaining
1 1 infants
(eight
per cent).
In the
patients
with progressive
curves, either single or double,
the “recessed”
side of the plagiocephaly
always
corresponded
with
the convex
side of the thoracic
or
thoracolumbar
curve.
In the patients
with resolving
curves,
the “recessed”
side of the head corresponded
with
contralateral
bat ear.
with
the convex side of the curve in all but four patients.
All patients
with progressive
infantile
idiopathic
scoliosis
required
treatment
(McMaster
and Macnicol
1979).
DISCUSSION
idiopathic
scoliosis
Number
4.,’
Over the last 10 years there
has been an increasing
awareness
of the need for early recognition
and referral
of all types of scoliosis
to a specialised
clinic
where
accurate
diagnosis
and treatment
can be carried
out. As
a result, increasing
numbers
of patients
with idiopathic
scoliosis
have presented
to the Edinburgh
Scoliosis
Clinic
but, paradoxically,
there has been a marked
decline
in
the number
with infantile
idiopathic
scoliosis
and this
has applied
to both progressive
and resolving
curves (Fig.
1). Progressive
infantile
curves
are never
overlooked
because
they produce
a severe deformity
at an early age,
and it is very unlikely
that more resolving
curves are now
being missed
than in the past. A similar
decline
in the
previously
high incidence
of infantile
idiopathic
scoliosis
has been observed
in West Germany
(Mau 1981).
If we look at the relative
frequency
of the different
types of idiopathic
scoliosis
presenting
each year, we see
an even more dramatic
change
(Fig. 2). Of the 157
patients
with idiopathic
scoliosis
seen between
1968 and
1971 ; 41 per cent had infantile
curves,
seven per cent
juvenile
curves and 52 per cent adolescent
curves. All the
juvenile
and adolescent
curves progressed,
whereas
only
34 per cent of infantile
curves progressed
and 66 per cent
resolved.
Since 1971 there has been a continuous
gradual
change
in the relative
frequency
of the different
types of
idiopathic
scoliosis.
The frequency
ofadolescent
scoliosis
has increased,
the frequency
of juvenile
scoliosis
has
remained
static
and that
of infantile
scoliosis
has
decreased
markedly.
Of the 153 patients
with idiopathic
scoliosis
seen between
1980 and 1982 only four per cent
had infantile
curves,
seven per cent had juvenile
curves
and 90 per cent had adolescent
curves.
All the juvenile
and adolescent
curves progressed
whereas
only three of
the infantile
curves
progressed
and three resolved.
The
relative
frequency
of the different
types of idiopathic
THE
JOURNAL
OF BONE
AND
JOINT
SURGERY
INFANTILE
IDIOPATHIC
SCOLIOSIS:
scoliosis
seen in Edinburgh
now approaches
that found
in North
America
: a survey
in Boston
of 208 patients
with idiopathic
scoliosis
found
that 0.5 per cent had
infantile
curves (one patient),
10.5 per cent had juvenile
curves and 89 per cent had adolescent
curves (Riseborough
and
Wynne-Davies
1973).
The
frequency
of
adolescent
idiopathic
scoliosis
in the two cities is now
virtually
identical,
but infantile
scoliosis
is still more
common
in Edinburgh
than
in Boston
and juvenile
scoliosis
is less common
(Fig. 5).
EDINBURGH
BOSTON
1968-1971
1967-1970
1980-1982
(153 patients)
(l57potients)
I 208
patients)
Fig. 5
Bar
chart
showing
the
incidence
and
of idiopathic
Boston.
scoliosis
in Edinburgh
Wynne-Davies
(1975) suggested
that the aetiology
of infantile
idiopathic
scoliosis
is multifactorial,
with a
genetic
tendency
to develop
the deformity
which
is
“triggered
off” by different
medical
or environmental
factors. The balance
in each infant islikely to be different,
an infant
with a Strong genetic
tendency
needing
very
little triggering
action and vice versa. From this study it
would
appear
that although
the genetic
tendency
to
develop
infantile
idiopathic
scoliosis
remains
constant,
the triggering
factors are being removed,
or alternatively
some beneficial
factor presumably
present in the environment
of North
America
is now being
applied
in
Edinburgh.
There
is a very close association
between
plagiocephaly
and both resolving
tnd progressive
infantile
idiopathic
scoliosis.
Plagiocephaly
is a plastic deformation of the skull. It is thought
to occur when an immobile
infant
habitually
lies towards
one side. The action
of
gravity
on the plastic skull causes the uppermost
side of
the face and head
to flow backwards
and become
recessed
; the lower ear is pushed
forwards
producing
the
commonly
associated
contralateral
“bat ear” (Fig. 6).
This deformity
is not usually present
at birth (Hay 1971)
but was found
in 28 per cent of 223 normal
infants
examined
in Edinburgh
between
the ages of six weeks
and six months.
The recessed
side of the head was on the
VOL.
65-B.
No. 5, NOVEMBER
1983
CAN
615
IT BE PREVENTED?
left in 83 per cent (Wynne-Davies
1975). The incidence
of plagiocephaly
in 63 premature
Edinburgh
babies was
63 per cent ; this higher incidence
is thought
to be due to
their greater
immobility
(Brown,
Bell and Fulford
1982).
Plagiocephaly,
however,
rarely
persists
and once the
child becomes
mobile it usually resolves
by the age of six
years.
In my study
of infantile
idiopathic
scoliosis,
plagiocephaly
occurred
in 96 per cent ofthe children
seen
in the first six months
of life and in 86 per cent of all the
children.
The recessed
side of the head was on the left in
81 per cent. The scoliosis
in these infants
was rarely
noted
at birth
but, like the plagiocephaly,
developed
within
the first six months
of life in 70 per cent. The
convex side of the curve corresponded
with the recessed
side of the head in all except four infants
with resolving
curves. This close association
between
the presence,
time
of presentation
and side of the deformity
of both
plagiocephaly
and infantile
idiopathic
scoliosis
suggests
a common
pathogenesis.
Wynne-Davies
(1975)
found
that in infants
with idiopathic
scoliosis
there
was a
greater
than normal
incidence
of prematurity,
low birthweight,
mental
retardation
(in boys),
older
mothers,
poorer
homes,
congenital
heart
disease,
and delayed
maturation
ofmusc!es
and ligaments
indicated
by herniae
and congenital
dislocation
of the hip (Hooper
1980). All
of these factors contribute
to immobility
of the newborn
and,
when
combined
with posture,
may trigger
the
development
of scoliosis
in a genetically
predisposed
infant.
Observations
in Edinburgh
(Brown
et al. 1982) and
elsewhere
(Mau 1972) have shown that during
the first
few months
of life there is a natural
tendency
for all
infants
to turn towards
their right side when laid supine.
This would account
for the predominance
of left-sided
plagiocephaly
in both normal
infants
and those with
scoliosis.
In my opinion,
when an immobile
infant is laid
supine
and partly
turns
towards
the right
side, the
Fig.
Diagram
illustrating
plagiocephaly
and
postural moulding
contralateral
bat
6
ofthe skull producing
left-sided
ear. Note the moulding
of the
thorax.
immature
thorax also undergoes
plastic deformation
due
to the influence
of gravity.
The uppermost
hemithorax,
like the skull,
tends
to flow backwards
causing
the
thoracic
vertebrae
to rotate backwards
on their long axis
(Figs 7 and 8). In the normal
spine there is a biomechanical coupling
of axial rotation
and lateral bending.
White
(1978) has stated that axial rotation
of the midthoracic
616
M. J. McMASTER
vertebrae
can on occasion
induce
a lateral curvature
of
the spine in which the anterior
aspects
of the vertebrae
point
towards
the convexity
of the curve.
Backward
vertebral
rotation
in the infant could therefore
induce an
upward
curvature
of the spine and a left-sided
thoracic
scoliosis
in infants
lying on their right side. The tendency
to produce
an upward
curve is increased
by pressure
from
the mattress
on the ribs of the lower hemithorax.
Once
the central nervous
system and the vertebral
musculature
mature,
the infant
becomes
much more mobile
and the
idiopathic
scoliosis.
The prone position
is theoretically
advantageous
because
the child cannot
roll onto its side
and this prevents
asymmetric
moulding
ofthe thorax and
promotes
early maturation
ofthe spinal extensor
muscles.
In the past, infants
in Great Britain
were usually laid on
their back or towards
one side and were often wrapped
tightly in blankets.
However,
in the last six to eight years
there has been an increasing
tendency
to advise mothers
to nurse their babies
prone and, because
more people
now have central
heating,
there is less need to wrap
Fig. 7
Figure
7-Diagram
laid supine
and
months
partly
who presented
illustrating
turned
postural
moulding
of the thorax
when
towards
its side. Figure
8-A
baby boy
with a left thoracic
been laid supine
and partly
of his head and trunk.
The
The head shows
a left-sided
idiopathic
scoliosis.
a baby is
aged four
He has habitually
turned
towards
his right side resulting
in moulding
creases
on his trunk
indicate
his relative
immobility.
plagiocephaly
and there
is a right bat ear which
has been against
J
the mattress.
Fig. 8
tendency
to spinal
curvature
is usually
overcome
; this
accounts
for the high incidence
of resolving
curves.
If,
however,
there is a marked
vertebral
rotation
associated
with the genetic,
medical
and environmental
factors
discussed
earlier,
it is possible
that the spinal balance
could
be irreversibly
affected
and the scoliosis
would
progress
rather than resolve. The prognostic
rib-vertebral
angle difference
at the apex of the curve, as described
by
Mehta (1972), is produced
by a combination
of vertebral
rotation
and the action of gravity causing
the ribs on the
convexity
to droop.
The greater
the tendency
for the
upper
hemithorax
to grow backwards,
the greater
the
degree ofvertebral
rotation
(and the rib-vertebral
angle);
the greater
the rotation
the more likely is the scoliosis
to
progress.
Why there should
be such a marked
decline
in the
number
of children
presenting
in Edinburgh
with
infantile
idiopathic
scoliosis
is not fully understood.
In
North America
newborn
children
have always been laid
prone and it has been suggested
that this position
could
account
for the much
lower
incidence
of infantile
babies
in restrictive
blankets.
However,
the majority
of
babies
are still not laid prone : only 25 per cent of 568
normal
infants
examined
in their cots in Edinburgh
had
been laid prone and 75 per cent were supine
(Brown
et
al. 1982). Seventy
per cent of the babies
who were laid
supine
showed
a strong
preference
to turn towards
the
right. In my series, none of the infants
who developed
idiopathic
scoliosis
in the last six years had been laid
prone.
Wynne-Davies,
in her series, found that before
1972 only three out of the 97 Edinburgh
infants
with
scoliosis
had been laid prone and all three had resolved
spontaneously.
This study
has shown
a marked
decline
in the
incidence
of infantile
idiopathic
scoliosis
in the community and it is therefore
concluded
that this is a preventable
deformity.
Evidence
suggests
that the onset of scoliosis
may be associated
with immobility
in the supine
or
lateral
position
and it is possible
that by laying
the
newborn
infant
in the prone position
and encouraging
much freer movement
the incidence
of this potentially
severely
crippling
disorder
may be further
decreased.
THE
JOURNAL
OF BONE
AND
JOINT
SURGERY
INFANTILE
IDIOPATHIC
SCOLIOSIS:
CAN
617
IT BE PREVENTED?
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