therapeutic exercise children spastic spinal paraplegia
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mp7eszaee eg 23 Uas 8 il 497 -- 506 R' (1996 F)
Report
Therapeutic Exercise for Children with Spastic Spinal Paraplegia"
Masamichi FURUSAWA'", Nobuhiro HIURA, Masahiro TAKESHITA,
Yuki NOMOTO, and Kanako IZUMI
Abstract '
The purpose of this research is to ascertain the characteristics that differentiate
spastic spinal paraplegia (SSP) from spastic diplegia of cerebral palsy (CP ・ SD), an.dthe points that should be taken into consideration in the treatment of SSP by physi-
cal therapists, We report on ways of physical therapists can prevent, by therapeutic
exercise, abnormal development of SSP children. Eleven SSP children are analyzed
through clinical charts, and with video-films, photographs, and Bobath's postural
tone test in comparison with CP (SD) children who had had lumbago and foot pain,
No child with SSP was born at a birth weight of less than 2,500 g, unlike the typical
scenario for CP (SD) children (p<O,Ol), All children acquired the ability to walk at
2.6± 1.4 years on average (ranging from 1 year to 5 years 1 month). Eight achieved
independent without braces, one with short leg braces, two with Lofstrand crut6heS
and short leg braces. SSP children were clearly superior to diplegic children in
terms of normal extensor patterns of the lower extremities and selective movements
of individual joints. Lumbago was present in 5 (45.4%) at 7.8± 1.3 years and foot
pain was present in 4 (36.3%) at 8.5± 2.6 years of our 11 cases. Postural tone of the
lower trunk tended to be low in SSP. In order to obtain walking and prevent lum-
bago, physical therapists should make efforts to promote sustained coactivation of
the lower trunk musculature early in the course of treatment,
Key words Children with spastic spinal paraplegia, Spastic diplegia, Therapeutic
exerclse
Introduction
Spastic spinal paraplegia (SSP) is paralysisdue to degeneration in the spinal cord mainly
'
,JivaffM,trpm,msWzzfiAdiffbltwix'S
Bebath Memorial HospiLal (1-6-5 Higashinakahama, Joto-ku, Osaka 536, Japan) (Received June 21, 19941Accepted August 31, 1996}
with bilateral involvement to the cor-
ticospinal tractsi). It has recently been
reported that spastic spinal paraplegia in pe-
diatric patients is not necessarily limited to
the spinal cord but possibly includes
degenerative changes in higher centers of the
brain2-5). Differential diagnosis from other
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498 wa7diza\
diseases is important because of the different
clinical picture that ultimately emerges.
Most children with SSP who received thera-
peutic exercise at eur hospital were original-
ly diagnosed elsewhere as having spastic di-
plegia of cerebral palsy (CP・SD). Physical
therapists should differentiate SSP from
other conditions and proceed with treatment
after having obtained a 'thorough
knowledge
of thc characteristics of abnormal develop-
ment in SSP. In this study, we ascertain the
characteristics that differentiate SSP from CP
(SD), and the points that should be taken into
consideration in the treatment of SSP by
physical therapists,
Subjects and Methods
The subjects were eleven children with
SSP, who received therapeutic exercise at
Bobath Mernorial Hospital between August
1982 and July 1993 (Fig. 1). Prior to receiv-
ing treatment at our hospital, cight children
(72,7%) had originally been diagnosed as
having had CP (SD>, one (9.0%) as having
SSP, and the condition had been evaluated as
unknown in two (18.2%). Thus, the rnajority
of these children had been treated by physi-
cal therapists under the diagnosis of CP (SD),
They were seven boys and four girls. None
had Segawa disease (hereditary progressive
dystonia)6} or juvenile Parkinsonism7), both of
which can produce symptorns similar to
those of SSP. Neurogenic bladder8) was not
observed in any of these ehildren. The mean
age at initial examination was 6.8± 2.3 years
(ranging 'from
2 to 13 years). Five children
were treatcd on beth an inpatient and an out-
patient basis, and 6 werc treated by thera-
peutic exercise only on an outpaticnt basis.
Diagnosis prior to treatment at our hospi-
ee 23 igag 8 e
Fig. 1. Pattern of walking in children with spastic
spinaE paraplegia. Lordosis is remarkable and compensatory sideflexion of trunk is
observed when he shifts his weight to one
leg.
tal, birth history, family history, locomotor
development, presence or absence and the oc-
casion of lumbago and foot pain, and any
previous surgical opcrations were ascertaincd
from medical charts. Standing posture and
walking patterns were assessed from video-
films (VTR) and photographs in order to find
out abnormal patterns caused by spasticity
in legs and compensatory movements espe-
cially in trunks, Postural tone was assessed
with the postural tone test advocated by
Bobath9). Points that should be taken into
consideration in treating pediatric patients
with SSP by our approachiO} were in-
vestigated by comparing our data on SSP
children with the report of CP (SD} children
by Suzukiii), many clinical features of which
resemble those of SSP. We checked on the
differentiation of both groups in the birth
weights and the gestational ages. Lumbago
and foot pain of SSP were compared with
those of CP <SD) in scanning the clinical
records. They were also tested to have
speech disturbance or not,
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Therapeutic Exercise for Children with Spastic Spinal Paraplegia 499
Results
Eight children had blood relatives who
either had definitive SSP or were suspegted
of having SSP. SSP was a known entity in
two pairs of twins (four children), two
fathers, a mother, and an eider sister. One
uncle was suspected of having SSP. Among
the eight children who had blood relatives
with SSP, six had been diagno$ed as having
CP (SD) and one as having SSP, and the con-
dition had been evaluated as unknown in
one. Of the three children who had no
known blood relatives with SSP, two were
referred to our hospital under the diagnosis
of CP (SD) and one remained undiagnosed.
Cephalic presentation was common to the
birth of all the children, The values of jaun-dice were within normal range. Four of the
childrcn weighed 2,500-3,OOOg at birth, and
the other seven 3,OOO-3,500g, so none of
them had abnormally low birth weight. As-
phyxia was present in 3 children (27.2%),who were maintained in incubators fer less
than 7 days after birth, and their subsequent
conditions was favorable. Gestation had
been 36 weeks for two children, 38 weeks for
one, 39 weeks for four, and 40 weeks for
four. The three children with asphyxia had
been born at gestations of 39 weeks and 40
weeks. Three children had speech disturb-
ance. A pair of 8 year old twin boys and a
6,5 year old girl had dysarthria. Locomotor
development is shown in Fig, 2, The chil-
dren could roll over at 7,l± 2.4 months on
the average (ranging from 4 to 12 months).
Sitting was achieved at 7.7± 2.6 months
(ranging from 5 to 13 months), although the
age was uncertain in one child. Crawling
was achieved at 11.8± 4.8 months (ranging
WALKINGWITHOUTBRACES
WALKING WITH SHORT L.EG BRACES
WALKINGWITHLOFSTRAND CRUTCHESANDSHORTLEa BRACESWALKINGWITHWALKER
CRUTSINGLOCOMOTIONASSESSMENT
F --o" :--oH --o 1H ,M ,
'111'''/11''1T''b'
IN:TtAL
ASSESSMENTF]NALASSESSMENT
Fig. 2. Change of locomotion.
from 6 to 16 months), but with the ages un-
certain in two children. The children could
cruise along the furniture at 18.8± 11.1
month (ranging from 9 months to 2.6 years),
All children acquired the ability to walk,
As shown in Flg. 2, nine ambulated inde-
pendently, but two needed Lofstrand
crutches. They became able to walk at 2,6±
1.4 years (ranging from 1 year to 5 years 1
month).
Six of the children underwent surgical
proceduresi2)i3). The Baker operation to
lengthen the gastrocnemius was performed
bilaterally in five children, and the surface of
adductor longus was released bilaterally to
alleviate flexion and adduction contracture of
the hip joints in one child. Medial ham-
strings were clongated bilaterally in one
child to increase the ability to extend the
knees. The flexor hallucis longus was
released bilaterally in one child for treatment
of hammer toes.
Pain was noted in seven (63.6%) of the
children with SSP. Five (45.4%) had lumba-
ge at 7.8± 1.3 years (ranging from 6 years to
10 years), and four (36.3%) had foot pain at
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500
Table
ve\wtzaeig ee23igees・g-
l Comparison of birth weight between SSP and CP {SD)
Birth weight (g)
1,ooe-1,soe 1,500-2,OOO 2,OOON2,500 2,500--・3,OOO 3,OOO<
SSP (N-11)CP (SD) (N- 9)'
o3 o2 o2 4o 72
'Suzuki
19goll).
8.5± 2.6 years (ranging from 5 years to 12
years), In scanning our clinical records of
thirteen children with cerebral palsy, we
found that eight diplegic children and one
diplegic adult had suffered Iumbago at 12.9±
5,6 years (ranging from 5 years 2 months to
22 years), and five diplegic children had had
foot pain at 9.4± 3,1 years (ranging from 5
years 9 rnonths to l3 years). All of three
children with CP (SD) could walk independ-
ently without crutches,
When spasticity of the lower extremities
was reduced by the therapisVs handling, chil-
dren with SSP were better able than children
with CP (SD) to maintain the lower ex-
tremities extended against gravity, which is
necessary for standing and walking. The
chi!dren with SSP also dernonstrated greater
ability for selective movement at the hips,
knees, and ankles, especially in movement in-
volving fiexion and extension of the knee
and dorsifiexion of the ankle. In contrast, the
children with SSP had diMculty with
coactivation in thc lower trunk, particularly
with sustaining contraction of the abdominal
muscles during extension of the trunk
against gravity.
Our clinical observations, gait analysis
using VTR and photographs, and postural
tone testing have confirmed that postural
tone of the lower trunk in children with SSP
is lower than that in children with CP (SD),and that SSP is associated with a tendency
(P< O.Ol)
for marked lumbar lordosis.
Discussion
1. Knowledge of SSP in terms of physical
therapy
Some recent studies using diagnostic im-
aging, such as computed tomography and
magnetic resonance imaging, as well as cere-
brospinal fluid testing, have Shown that the
degeneration Qf SSP is not necessarily limit-
ed to the spinal cord but can also include the
brain2-5). Two brothers, 10 and 8 years old,
did indeed exhibit slight intellectual handi-
caps. However, they were independent in ac-
tivities of daily living and they could socially
interact with other persons.
Eight (72.5%) of the 11 children with SSP
had been treated by therapeutic exercise
under the diagnosis of CP (SD) prior to beingbrought to our hospital, in day-care centers
for disabled preschool children. Birth weight
was investigated in comparison with the
report by Suzukiii} (Table 1). Arnong the
nine children observed and fellowed by
Suzuki in a neenatal intensive care unit and
finally diagnosed as having CP (SD), seven
(77,7%) were born at birth weights below
2,OOO g, and the remaining two (22,3%)weighed 3,OOOg or more. AII of the children
with SSP, howeyer, weighed 2,500g or more
at birth, so none werc low birth weight in-
fants. This difference in distribution be-
tween the CP (SD) and SSP children was sig-
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Table 2 Comparisen of gestational ages between SSP and CP CSD)
501
Gestational age (weeks)
2728293031323334353637383940
SSP (N- 11)
CP (SD) (N == 9)*o1o1o1o1o1ooo1ooeo2ooo1o4!42
"Suzuki
19goil),
nificant (p<O,Ol) according to the Mann-
Whltney U test.
The CP CSD) children and SSP children
were compared according to gestational age
(Table 2). Nine SSP children (8i.8%) were
born at gestational ages of 38 weeks or more,
According to the report by Suzuki, however,
only three children (33.3%) with CP (SD)were born at 38 we6ks or rnore, This differ-
ence between the diagnostic grouPs was sig-
nificant <p<O.05) according to the U test.
Bobath and Blecki4) have also described
many children with CP (SD) as having been
premature babies. Hagberg et al.i5) reported
that 88 (45.5%) of 207 children with CP (SD)were born at full term,
From the viewpoint of motor development,
rolling over and sitting were achieved very
late only in the twin boys with SSP, The
period of achievement of these behaviors was
normal or only slightly delayed in the other
children. Six (60%) of the 10 children
achieved cruising by one year of age, except
possibly for one child whose data were un-
known. The other four acquired cruising at
1 year 3 months to 3 years 2 months. These
findings suggest the occult presentation of
SSP in some children.
In Japan, differentiating SSP from CP (SD)is difiicult because of the small number of
day-care centers for preschool handicapped
children that have full-time specialist doctors.
It is therefore important, in differentiating
(p< O.05)
SSP from CP (SD), for physical therapists to
investigate gestational age at birth, birth
weight, birth history, presence or absence qf
SSP in blood relatives, course of motor devel-
opment, and presence or absence Qf
dysarthria. Pediatric patients' responses to
stimulation during therapeutic exercise
should also be observed carefully.・ SSP is
characterized by deficiency of sustained con-
traction of the abdominal muscles during
antigravitational extension of the trunk and
by more selective movement in the lower ex-
tremities than typically found in spastic di-
plegia SSP should be suspected after a com-
prehensive assessment of all of the informa-
tion described above, and physicians should
be requested to make a differential diagnosis,
attempting to rule out CP (SD}. Besides the differentiation of SSP from CP
(SD), it is also important in planning treat-
ment to differentiate these disorders from
human T-lymphotropic virus-type associated
myelopathy (HAM), as reported by Shimazaki
et al,i6) When a physical therapist suspects
that the condition of a child may not be cere-
bral palsy with spastic diplegia, the therapist
should clearly describe the various responses
obtained through therapeutic exercise and
speculate Qn the reasons for those responses.
2. Essential points on therapeutic exercise
The distribution of spasticity in the lower
extremities of children with SSP and their
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abnormal walking patterns resembled the ab-
normal developmenti7) in CP (SD). Eight of
the 11 SSP children had been treated by
therapeutic exercise under a diagnosis of CP
<SD) before being brought to our hospital,
Bealsi8} reported that 52.6% of children
with CP (SD) achieved independent ambula-
tion and 20.4% became able to walk with
crutches. Kajiurai9) similarly reported that
50% of children with CP (SD) became able to
walk independently while another 25%
became ambulatory with crutchcs. From
these reports, the rate of acquiring the ability
to walk in children with CP (SD) can be con-
sidered to be about 75%. In contrast, all of
the children with SSP acquired the ability to
walk, although 2 (18.1%) were limited to
crutch arnbulation. None of the patients
with SSP showed severe spasticity20), enough
to prevent movement, although moderate
spasticity was present in the lower ex-
tremities of all SSP children. According to
Bealsi8) and Okawa et al.i3>, some children
with CP require iliopsoas release because the
muscle is shortened due to severe spasticity.
None of the present children with SSP under-
went this operation. It is particularly
diMcult, in treating SSP, to obtain sustained
coactivation of the abdominal and gluteal
muscles, necessary for extending the pelvis
and trunk against gravity during standing
and walking. The diMculty in sustaining
contraction of the abdominal muscles in an
attempt to extend the trunk in therapeutic
exercise was confirmed by comments of
therapists in our group. Physical therapists
should promote continuous contraction of
the child's lower trunk, particularly the ab-
dominal muscles, when attempting to elicit
antigravitational extension of the trunk
ee 23 gee 8 e
during the walking portion of treatment.
The appearance of lumbar and foot pain in
seven (63.6%) of the SSP children, ranging in
age from five years to twelve years of age,
can be considered unusually frequent. These
children could walk fast or even run, Ac-
cording to the Welch test, SSP children
suffered lumbago significantly earlier than
the nine diplegic children (p<O,05), but foot
pain showed no such significant difference
between the two groups. The results of the
analysis suggest that pain, especially lumba-
go, tends to appear in pediatric patients with
SSP earlier than in those with CP (SD),
Lumbar lordosis was particularly remarka-
ble in nine clients (Fig. 1). Postural tone test-
ing revealed slight to moderate spasticity in
flexors and adductors of the hip joints. The
spasticity of these muscles was particularly
marked in the outer range of hip joints.Spasticity of the hip flexors and adductors
interferes with antigravitational posterior tilt
of the pclvis and extension of the lower ex-
tremities. Since righting activities from the
pelvis and lower trunk appear late as a re-
sponse, however, unilateral loading of body
weight onto a lower extremity and the pro-
pulsion necessary for walking are promoted
by hyperextension and lateral flexion of the
lumbar spine by excessively using the back
muscles, All five patients who complained of
lumbago showed lumbar lordosis in standing
as well as during walking. The lumbago
might have developed as a result of repeti-
tion of compensatory movements.
Treatment of lumbago is incorporated into
systematic therapeutic exercise. Contraction
of the abdominal muscles is sustained during
extension of the trunk and lumbar lordosis is
thereby reduced (Fig. 3). In this process, mo-
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Fig.3. Promotion of sustained coactivation in trunk musculatures.
bility improves in painful lumbar extensors.
Whcn occasien demands, soft tissue mobiliza-
tion is transiently performcd in the lurnbar
region2i) to reduce pain prior to continuing
therapeutic exercise. As a result, lumbago
may disappear after this approach. To im-
prove gait and prevent lumbago in SSP,
physical therapists should reduce the
spasticity of flexors and adductors of the hip
joints, promote sustained contraction of the
abdominal muscles during coactivation of the
lewer trunk, starting in infancy, particularly
during extension of the trunk, and must con-
sciously pursuc preventjon of lumbar lordo-
sis.
With regard to foot pain, evaluation of pos-
tural tone revealed spasticity with a slight to
moderate degree of resistance9) in the tibialis
anterior, the tibialis posterior and
gastrocnemius muscles, Postural tone testing
revealed moderate spasticity in the
plantarflexors. Intrinsic musclcs of the foot,
however, exhibited low tone. The arrange-
ment of the longitudinal arch of the foot was
disrupted and pes pianovalgas was observed.
503
These findings were considered to be respon-
sible for pain resulting from loading of body
weight onto the feet, particularly the soles.
Before wcight loading onto the lower ex-
tremities, the hypersensitivity of feet was
desensitized and the difference in distribution
of postural tone of the lower cxtrernities and
feet was cqualized while selective movements
of the lower extremities were performed in
supine or sitting during treatment (right and
left parts of Fig. 4)22}. Loading in the stand-
ing position was then gradually increased
(right and left parts of Fig. 5), The foot pain
disappeared with stepwise progression of this
Fig. 4.Desensitization of hypersensitivity in solesand triceps suraes to weight on hee]s.
Fig. 5.Treatrnent in walking. In order to de-crease lordosis and to prevent compensato-
ry sidefiexion of his trunk, contraction of
abdominal muscles is sustained jn stand-ing and walking.
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program.
A pair of 8 year-old twin boys and one 6.5
year-old girl underwent speech therapy be-
cause all three had dithculty with speech due
to dysarthria, They were suggested
supraspinal involvement, but their intellectu-
al abilities were normal. Takahashi et at.3>5)
reported autosomal recessively inherited SSP
with dysarthria. Physical therapists should
help the SSP child to sustain a syrnmetrical
neutral position of the head and neck during
walking, in conjunction with speech therapy.
The pelvis in such a child is insuMciently
mobile because of spasticity in the muscles
surrounding the hip joint. During walking,
the center of gravity moves in a compensato-
ry manner, with lateral fiexion and anterior
flexion of the trunk, head and neck, and with
horizontal instability of the jaw (Fig. 1). As a
result, fine independent skillful movements
of the lower jaw, tongue, and lips, necessary
for articulation, do not develop adequately,
exacerbating the diMculty with speech devel-
opment. Physical therapists carry out treat-
ment by reducing the spasticity of the hip
flexors and adductors and eliciting sustained
coactlvation of the abdominal muscles and
gluteal muscles with the aim of helping the
child rnaintain the head and neck in neutral
position without compensatory movements
during walking. With these ways physical
therapists must thus cooperate with speech
therapists in planning and administering
treatment.
Conclusion
Eleven pediatric patients with SSP were
studied. The points to which physical
therapists should pay attention in ad-
ministering therapeutic exercise for acquisi-
eg 23 tseg 8 -ij
tion of the ability to walk were also
described. Physical therapists can contribute
to the preventien of abnormal development,
Iumbago, and foot pain in SSP by applying
therapeutic exercise based on an adequate
knowledge of the characteristjcs and differ-
ences in the individual clinical features of
SSP and CP (SD).
References
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506 理学療法学 第 23巻第 8 号
〈要 旨〉
小児脊髄性痙性対麻痺へ の運動療 法
古澤正道, 月浦伸祐,竹下正弘,野本由紀,泉佳奈子
ボ バ ース 記念病院
本研究の 目的は小児 の 脊髄性痙性対麻痺 (SSP) の 特徴を脳性麻痺痙 直型両麻痺 (CP ・
SD ) と の 対比で 明確に す る こ とと, SSP の 運動療法上の 留意点 を明 らかにす る こ とで ある。
対象は 11名の SSP で, 方法は ビ デ オ と写真に よ る歩行パ タ
ーン の 分析, ボ バ ー
ス の 姿勢
緊張テ ス ト, 診療録か らの 出産歴 ・腰痛 ・足部痛 の 調査で あ る 。 来院前診 断名 は,CP
(SD) 8名, SSP l名,不明 2名で あ っ た 。 SSP に は 2,500 g 未満の低 出生体重児 は いず,
在胎週で は 9 名が満期産で あ っ た 。 歩行は ロ フ ス トラ ン ド杖を使用 した 2 名を含め て ,2.6
± 1.4歳 (1歳〜 5歳 1カ月)で 全 員が獲得 した 。 両下肢の 正常 な伸展運動 と, 遊脚相で の
個 々 の 関節 の 選択運動 は CP (SD )よ り高い能力をも っ て い た 。 SSP の 腰痛 は 5 名 (45.4%,
7.8 ± 1.3 歳), 足部痛は 4 名 (36.3% ,8.5 ± 2.6歳)に生 じた 。 CP (SD)に比べ SSP の 腰
痛は有意に早 く生 じた 。 腰部 の 運動性を回復さ せ腰椎前弯の軽減を図 る こ とと, 足部内の
姿勢緊張 を整え た上 で 踵 に体重 を荷重す る こ とで 疼痛は緩解 した。安定 した歩行の 獲得と
腰痛防止の た め に は , 早 くか ら下部体幹 の 持続 した同時活動を促通 し, 腰椎前弯を軽減す
る こ とが重要 で あ る 。
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