1. The study evaluated the motor outcomes of 10 children with spastic diplegia who received different intensities of early onset physiotherapy. 2. Children who received a full course of intensive physiotherapy for over 52 months on average (full training group) had better motor outcomes than those who received inadequate or no therapy (insufficient training group). 3. At the 5-year follow up evaluation, 4 of the 5 children in the full training group could stand or walk, compared to none of the 5 children in the insufficient training group, showing a statistically significant difference.

1. Brain & Development 26 (2004) 118–126
www.elsevier.com/locate/braindev
Original article
Motor outcome differences between two groups of children
with spastic diplegia who received different intensities
of early onset physiotherapy followed for 5 years
Toyoko Kandaa,*, Frank S. Pidcockb, Katumi Hayakawac, Yuriko Yamoria, Yuko Shikatad
a
Department of Pediatric Neurology and Rehabilitation at St. Joseph’s Hospital for People with Handicaps, 6 Higashikobai-cho,
Kitano, Kita-ku, Kyoto, 603-8323, Japan
b
Department of Physical Medicine and Rehabilitation, Johns Hopkins University School of Medicine, Kennedy Krieger Institute,
707 North Broadway, Baltimore, MD 21205, USA
c
Department of Radiology, Kyoto City Hospital, Kyoto, Japan
d
Department of Medicine, Kamigyo Hospital, The Second Central Hospital of Kyoto Miniren, Kyoto, Japan
Received 9 December 2002; received in revised form 21 May 2003; accepted 26 May 2003
Abstract
The objective of this study is to determine the clinical effectiveness of early onset long-term intensive physiotherapy on motor
development in children with spastic diplegic cerebral palsy (CP). The study was a non-randomized cohort study with 62 months (mean)
follow-up. The participants were ten infants who were first examined before 3 months of age corrected for prematurity. All had a gestational
age of less than 33 weeks and a birth weight of less than 2000 g. Brain magnetic resonance imaging revealed periventricular white matter
injury in nine subjects and moderate grade bilateral porencephaly in one. Five completed a full course of training of 52 months (mean), two
did not receive therapy, and three received an insufficient course of therapy. The study was conducted at the Regional Center for Children
with Disabilities including outpatient clinics and a school for children with special needs. The Vojta Method was used, which is an extensive
family oriented physiotherapy program which uses isometric strengthening of muscles with tactile stimulation. Subjects were evaluated for
the highest motor developmental level at the outcome evaluation 59 months (mean) after initiation of therapy. Four of the five who completed
training could either stand still for 5 s or walk at the time of the outcome evaluation 52 months after the beginning of the therapy program.
None of the five subjects with no training or insufficient training could accomplish this task when evaluated 64 months following therapy
initiation. This was a statistically significant difference (P ¼ 0:0278). A consistently applied physiotherapy program resulted in better motor
outcomes in this group of children at risk for developing spastic diplegic CP.
q 2003 Elsevier B.V. All rights reserved.
Keywords: Long-term therapy; Intensive physiotherapy; Early onset of therapy; Vojta Method; Spastic diplegia; Magnetic resonance imaging; Cohort study
1. Introduction The aim of this study is to evaluate whether there is an
effect of early onset long-term intensive physiotherapy on
Establishing the scientific validity of physiotherapy the motor development of premature infants at risk for
programs for premature infants at risk for motor problems cerebral palsy. Infants who received therapy were compared
is a difficult task. Although a randomized clinical trial to those who either received inadequate therapy or no
would provide the most definitive proof, there are many therapy.
obstacles to successfully completing studies of this kind
over a prolonged period of time [1]. This report describes a
1.1. Background
group of children who were offered therapy starting at a very
young age and then consistently followed for 5 years. Motor
outcomes at the end of the study period are described. Specially trained physicians in regional training centers
in each of Japan’s 47 prefectures perform assessments in
* Corresponding author. Fax: þ 81-75-464-2760. infants to determine their eligibility for financial aid from
E-mail address: azatuji@d1.dion.ne.jp (T. Kanda). government or private medical insurance. Aid is given if
0387-7604/$ - see front matter q 2003 Elsevier B.V. All rights reserved.
doi:10.1016/S0387-7604(03)00111-6

2. T. Kanda et al. / Brain & Development 26 (2004) 118–126 119
the risk of disability is strongly suspected in children even when younger than a corrected age of 3 months. Assess-
less than 6 months of age based on the likelihood of walking ments occurred once every 2 months if the corrected age
at 3 years of age. was between 3 and 6 months and once every 3 months until
This study is from the referral center in the Kyoto entering primary school if the corrected age was 6 months or
Prefecture, which includes a population of 2.56 million older. Physiotherapy was discontinued and assessments
people. Referrals come from hospitals and regional health were conducted once a year until the age of 6 if a child
care centers that have government contracts to screen started walking in a stable way, without evidence of toe
healthy infants and toddlers. walking or valgus.
2.4. Follow-up study
2. Method
The total number of patients who were first assessed in
2.1. Neurological assessment the Department of Pediatrics at our hospital between 1991
and 1995 was 2449. Among them, there were 37 children
Pediatric neurologists with at least 10 years of pediatric who were born at less than 33 weeks of gestation,
neurological experience performed evaluation of the subjects. weighing , 2000 g, and who were assessed before they
Physical therapy was recommended based on Vojta’s motor reached a corrected age of 3 months. Assessment of the
risk criteria or central coordination disturbance criteria, study parameter items from patients’ charts was done in
which includes spontaneous movements, postural reactions October 1998, and additional information was obtained by
and primitive reflexes [2]. The decision to recommend calling families or making assessments at the hospital if
therapy was based on either the initial consultation or a necessary. The decision as to whether or not patients had
repeat examination within 1 month of the first consultation. received continuing full physiotherapy depended on
information provided by the therapist in charge. Not
2.2. Treatment only did they ask families if they were conducting the
therapies as recommended but they also evaluated the
Physiotherapy intervention was based on the method of ongoing skill of the parents in providing therapeutic
Vojta [3]. Therapists who had completed an 8 week training exercises during return visits to the hospital. To provide
course and who had at least 5 years of clinical experience appropriate interventions at the correct locations on the
with this method participated in this study. The same child for a sufficient duration requires continuing practice
therapist was responsible for therapy for each child in this much like practicing playing the piano. It is difficult to
report throughout the course of treatment. Movement and maintain the proper skills if sufficient intervention is not
reflex patterns were taught to the parents over a 3 month being done at home.
period which included 1 month of inpatient teaching during
which parents received daily training about the Vojta 2.5. Selection of the study group
Method, and about feeding and playing with their children.
Children were treated in their homes during the training Of the 37 very premature infants who were initially
period and after the patterns were learned. Parents evaluated before a corrected age of 3 months, 34 were
subsequently visited the hospital for instruction from followed-up until at least 3 years of age. Two children
therapists twice a month before the child was 1 year of moved away from the Prefecture and were lost to follow-up,
age, and once a month thereafter. They were instructed to and one severely affected infant died. The rest were divided
treat their children three to four times daily. Fully treated into three groups by the neurological findings within 1 year
children received 90– 120 sessions per month. Therapy after the first evaluation. The most severe group included
sessions took 30 min or more to complete, which included three children who showed very abnormal muscle tone with
time for displays of affection and affirmation to the children paucity of spontaneous movements and seven abnormal
at the completion of the therapy. Other interventions for postural reactions. They had profound mental retardation,
assisting these families in the daily care of their children severe visual disorders, and epilepsy. All three children in
were provided after 1 year of age. They included this group would not be expected to roll over or crawl. These
occupational therapy and feeding therapies. children had greater than eight abnormal primitive reflexes
as described by Vojta [2]. Twenty-one children had a slight
2.3. Follow-up decrease in spontaneous movements, less than seven
abnormal postural reactions and showed less than four
Each assessment was performed by Vojta’s system [2], abnormal primitive reflexes [2]. These children were
which included voluntary movements, postural reactions classified as the ‘mild group’. Six of these children were
and primitive reflexes. Assessments were recorded on charts diagnosed as at risk for cerebral palsy at the first evaluation.
and photographs were taken of voluntary movements and All of these children started walking without standing on
postural reactions. Each patient was assessed once monthly their toes or other motor disturbances during the study

3. 120 T. Kanda et al. / Brain & Development 26 (2004) 118–126
period. The remaining ten children had patterns of Table 1
spontaneous movement, postural reactions, primitive Characteristics of the study group
reflexes, and muscle tone abnormalities which were Item Group Statistics
intermediate between the severe and mild groups. Eight of
these children were diagnosed as at risk for cerebral palsy Full training Insufficient
based on these neurological findings. Two of these children training
showed increased asymmetry of spontaneous movements,
Number of cases 5 5
postural reactions, and primitive reflexes. They were Mean gestation (weeks) 30.6 (29–32) 30.4 (30–31) n.s.
diagnosed as at risk for central coordination disturbance [2]. Mean birth weight (g) 1474.8 1544.8 n.s.
This last group is the subject of this report. Five of these (1230–1680) (1262– 1806)
children received a full course of training for not less than 3 Cases whose birth weight 3 (60%) 2 (40%) n.s.
years and 3 months (full training (FT) group) and five had was less than 1500 g
Corrected months of age 1.2 (0–2) 1.0 (0–2) n.s.
abbreviated or no training due to parental reluctance first checked
(insufficient training (IT) group). Age at outcome evaluation 56.2 (42–82) 67.4 (48–95) n.s.
(months)
2.6. Description of the study group Mean trained term (months) 52.2 (40–77) 25.2 (0–69) P , 0:05
on one side
Complications
At the first assessment (mean age of 1.1 corrected Mental development
months), eight children in this group were at risk for DQ . 80 2 3 n.s.
cerebral palsy and two were at risk for a central coordination 80 ^ DQ . 70 2 0
disturbance. All ten cases were diagnosed with spastic 70 ^ DQ . 50 0 0
50 ^ DQ . 30 1 2
diplegia at the time of the outcome evaluation at a mean age
30 ^ DQ 0 0
of 62 months. Epilepsy 0 0 n.s.
Clinical characteristics and brain MRI findings of the FT Visual disorder 3 2 n.s.
and IT groups are described in Tables 1– 5 and Fig. 1. The Microcephalus 1 0 n.s.
mean gestational age of the FT group was 30.6 weeks Diagnosis at first check
Risk for cerebral palsy 4 4 n.s.
compared to 30.4 weeks for the IT group. The mean birth
Risk for central 1 1 n.s.
weight was 1474 g (FT group) and 1544 g (IT group) with coordination
three children with a birth weight , 1500 g in the FT group disturbance
and two in the IT group. Mental development in the two Diagnosis at the time of
groups was not significantly different. Three children in the outcome evaluation
Spastic diplegia 5 5 n.s.
FT group and two children in the IT group had a visual
disorder. One child in the FT group had microcephaly. All
cases showed distinct neurological signs at the first clinical
Table 2
examination (Table 2). All except one case showed paucity Neurological signs before corrected 3 months of age at the first clinical
of spontaneous movements of legs (Table 3). check
Rolling over, supported sitting, and unsupported sitting
did not occur at significantly different ages between the Group Cases Corrected Corrected Corrected
0 months 1 months 2 months
groups that received full therapy or partial therapy (Mann–
Whitney U-test; Table 4). Rolling over developed after the Full training 1 b, d, f, i
age when parents decided to discontinue physiotherapy or 2 a, b, c, h, i
were unable to continue with the full therapy regimen. 3 b, d, h
MRI findings included end stage of periventricular 4 a, b, c, d, e, g, h, i
leucomalacia in four children in the FT group and five 5 a, b, h, i
Insufficient 6 b, d, h
children in the IT group. One child in the FT group had training
moderate grade bilateral porencephaly. 7 a, b, d, e, i
The socioeconomic background was similar for both 8 a, b, c, h
groups. 9 c, d, h, I
10 a, b, g, h, i
(a) Hypertone; (b) all the seven postural reactions by Vojta are
3. Results abnormal; (c) body and four extremities move totally unstably without
separate movement; (d) fencing position with clear asymmetric position in
head, upper and lower extremities; (e) sudden jerky movement occurs
The mean corrected age at first assessment was 1.2
which makes supine position unstable and makes him/her cry; (f)
months for the FT group and 1.0 months for the IT group. opisthotonus; (g) abnormality of Moro reflex; frequency or too much
The mean age at outcome evaluation was 56 months for the extended elbows; (h) reflex of foot grasp is inhibited or delayed either on
FT group and 67 months for the IT group. The mean both or one side; (i) other pathologic primitive reflexes are found.

4. T. Kanda et al. / Brain & Development 26 (2004) 118–126 121
Table 3 Table 5
Patterns of spontaneous movements of legs before corrected 3 months of Findings of magnetic resonance in the study group
age at the first clinical check
Findings of magnetic resonance Group Statistics
Group Cases Corrected Corrected Corrected
0 months 1 months 2 months Full training Insufficient
training
Full training 1 a, b, c
2 a, b, e Number of cases 5 5
3 a, b, c Mean examined age (months) 29.2 (22–40) 26.4 (7–54) n.s.
4 a, b, c, d Subdural effusion 0 cases 1 casea n.s.
5 a, b, f, g Periventricular leucomalacia 4 cases 5 cases n.s.
Insufficient 6 a, b, c, d Bilateral porencephaly 1 case 0 cases n.s.
training Width between anterior 0.23 ^ 0.05 0.24 ^ 0.04 n.s.
7 a, b horn/maximum width of the brain
8 a, b, c Peritrigonal white matter reduced 5 cases 3 casesc n.s.
9 b, c in both hemispheresb
10 a, b, d High intensity aread
þ 5 cases 3 cases n.s.
(a) Spontaneous movement of legs is rare; (b) no elevation of legs is þA 1 case 0 cases
seen; (c) plantar flexion is seen at the extended legs; (d) hyperabduction and þP 0 cases 0 cases
hyperrotation outward of legs are seen; (e) only bilateral total flexion or þ AP 4 cases 2 cases
total extension of legs are seen; (f) stepping like movement is seen in both
a
legs; (g) plantar flexion is seen while legs are extended or flexed. Complicated.
b
Peritrigonal white matter reduced in both hemispheres is evaluated by
duration of training was longer in the FT group (52 months) comparing the normal control in the same age which appears in the
than in the IT (25 months) (P , 0:05) (Table 1). textbook of ‘Pediatric Neuroradiology’ written by A.J. Barkovich.
c
The diagnosis at first assessment was similar in both It was unclear in the other two cases since the checking was under 1
year of age.
groups. Four cases were diagnosed as at risk for cerebral d
þ means that there were high intensity areas at the body of lateral
palsy and one as at risk for central coordination disturbance ventricles; þA means that the high intensity area was also seen at the
in each group. anterior horn; þ P, at the posterior horn as well; þ AP means the high
Of the five families who administered full physiother- intensity areas at the body, anterior horn and posterior horn as well.
apy training, two mothers recognized abnormal lower
sibling. The remaining case in which full physiotherapy
extremity movement patterns and sought early attention.
occurred was in a family where one twin survived and
This also occurred in two other families where twins or
the other died. This mother dedicated herself to providing
triplets were born and the mother could compare the
therapy to the surviving child.
child with motor problems with a normally developing
Reasons for poor compliance with the physiotherapy
Table 4 in the IT group varied. They included severe asthma in
Months of age motor development acquired and months of age when one child, insufficient time or commitment by the parents
parents decided to discontinue physiotherapy or were unable to continue
to participate in administering the therapy, religious
full physiotherapy
convictions, and reluctance by the parent to accept the
Group Cases Months of age Months of age when parents diagnosis.
motor development decided to discontinue Four of the five children in the FT group could stand for 5
acquired physiotherapy or were unable
s or walk at the time of outcome evaluation. None of the five
to continue full physiotherapy
and the reason children in the IT group could stand. This was a statistically
significant difference (P ¼ 0:0278) (Table 6).
Full training 1 n:5, r:7, s:8, u:9
2 n:5, r:8, s:7, u:33 3.1. Case report 1 – full training
3 n:5, r:6, s:6, u:16
4 n:5, r:10, s: 7, u:17
5 n:5, r:7, s:12, u:22 The child shown in Fig. 2 was born at 30 weeks
Insufficient 6 n: 6, r:15, s:9, u:26 8, asthma gestational age weighing 1680 g. The brain MRI scan (Axial
training SE 2,200/120 image) at 2 years and 4 months of age (Fig. 1b)
7 n: 5, r:7, s: 4, u:16 4, advice of neonatal doctor showed irregular ventricular dilatation at bilateral posterior
8 n:5, r:9, s:18, u:24 9, religion
horns and also showed end stage of periventricular
9 n:7, r:11, s:11, u:24 8, religion
10 n:5, r:9, s:11, u:28 6, busyness of parents leucomalacia around anterior, body and posterior horns.
There was loss of myelinated white matter along the
n, neck control; r, rolling over; s, supported sitting with upper
bilateral body and posterior horns of the lateral ventricles.
extremities; u, unsupported sitting. Months of age motor development
acquired between two groups was not significant by Mann –Whitney U-test At a corrected age of 1 month, the child was unstable when
in relation to rolling over, supported sitting with upper extremities and supine, and exhibited a frequent Moro reflex (Fig. 2(1)).
unsupported sitting. Ventral suspension reaction at 1 month of corrected age

5. 122 T. Kanda et al. / Brain & Development 26 (2004) 118–126
Fig. 1. (a) Case 1: the Axial SE 2,500/120 image at 3 years and 4 months of age shows irregular ventricular dilatation at the anterior horn, the body and posterior
horns where the left side is more dilated than the right and also shows end stage of periventricular leucomalacia around anterior, body and posterior horns. (b)
Case 2: the Axial SE 2,200/120 image at 2 years and 4 months of age shows irregular ventricular dilatation at bilateral posterior horns and also shows end stage
of periventricular leucomalacia around anterior, body and posterior horns. There was loss of myelinated white matter along the bilateral body and posterior
horns of the lateral ventricles. (c) Case 3: the Axial SE 4,535/128 image at 1 year and 10 months of age shows ventricular dilatation at the body and posterior
horns where the left side is more dilated than the right and also shows porencephaly at the posterior of the right posterior horn. (d) Case 4: the Axial FSE
3,500/125 image at 1 year and 2 months of age shows irregular ventricular dilatation at the right posterior horn and also shows end stage of periventricular
leucomalacia around body and posterior horns. There was loss of myelinated white matter at the right posterior horn of the periventricle. It also shows small
cavities at lateral and dorsal sides of the body of lateral ventricles. (e) Case 5: the Axial SE 2,500/120 image at 2 years and 8 months of age shows irregular
ventricular dilatation at the left side and also shows end stage of periventricular leucomalacia around anterior, body and posterior horns. There was loss of
myelinated white matter at the bilateral body and posterior horns of the periventricle. It also shows small cavities along lateral sides of the body of lateral
ventricles. (f) Case 6: the Axial SE 4,535/128 image at 3 years and 6 months of age shows irregular ventricular dilatation at anterior, body and posterior horns
and also shows end stage of periventricular leucomalacia around anterior, body and posterior horns. (g) Case 7: the Axial SE 2,000/120 image at 4 years and 5
months of age shows irregular ventricular dilatation at bilateral posterior horns and also shows end stage of periventricular leucomalacia at body and posterior
horns. There was loss of myelinated white matter along the bilateral body and posterior horns of the lateral ventricles. (h) Case 8: the Axial SE 2,500/120 image
at 7 months of age contains a lot of water, shows irregularity of the ventricular figure at the body and also shows periventricular leucomalacia around the body.
It also shows subdural effusion. (i) Case 9: the Axial SE 2,200/120 image at 8 months of age contains a lot of water, shows irregularity of the ventricular figure
at bilateral anterior horns, body and posterior horns and also shows periventricular leucomalacia around bilateral anterior horns, body and posterior horns. (j)
Case 10: the Axial SE 2,200/120 image at 1 year and 10 months of age shows irregular ventricular figure and also shows end stage of periventricular
leucomalacia around the body and posterior horns. There was loss of myelinated white matter along the right body and posterior horn of the lateral ventricle.
revealed asymmetric head position, flexed arms, bilateral 7 months of age, although there was still slight flexion at the
fisting and extended legs with ankle plantar flexion hips and knees and foot weakness (Fig. 2(8)). She started
(Fig. 2(2)). This child was diagnosed to be at risk of walking at 5 years 10 months of age, still with tiptoeing. At
motor impairment, and physiotherapy was recommended. In 6 years 6 months of age she could walk, even outside, with
the supine position at a corrected age of 2 months, the child normal shoes although she was still tiptoeing. At this time,
exhibited a fencing position (extended left extremities and the mother was still administering physiotherapy with the
flexed right extremities). The head was inclined to the right Vojta Method three times daily, and the child attended a
with bilateral fisting and a left side ankle equinus position normal school (Fig. 2(9,10)).
(Fig. 2(3)). In the prone position (corrected age 2 months),
the child exhibited neck stiffness without head rolling or 3.2. Case report 2 – insufficient training
head extension, and flexing of the elbows with fists and
extended legs with tiptoeing (Fig. 2(4)). At 1.5 years of age The child shown in Fig. 3 was one of twins born at 31
the child could pull herself up on her mother’s knees, at weeks gestation and weighed 1562 g. The brain MRI scan
which time she had a round and hypotonic trunk and (Axial SE 2,000/120 image) at 4 years and 5 months of age
extended legs with ankle plantar flexion (Fig. 2(5)). She is shown in Fig. 1g. It shows irregular ventricular dilatation
started crawling at 1 year 5 months of age. The child was at bilateral posterior horns and also shows end stage of
pulling herself up at 2 years and 9 months of age with severe periventricular leucomalacia at body and posterior horns.
tiptoeing on both legs (Fig. 2(6)). She started walking There was loss of myelinated white matter along the
sideways with support at 2 years 5 months of age. At that bilateral body and posterior horns of the lateral ventricles.
time she could touch the floor with both heels upon At a corrected age of 2 months, the child showed no
command (Fig. 2(7)). She could stand still for 5 s at 5 years asymmetry of the neck and extremities in the supine

6. T. Kanda et al. / Brain & Development 26 (2004) 118–126 123
Table 6
Motor developmental level in the diplegic group at the time of outcome
evaluation
Item Group Statistics
Full Insufficient
training training
Number of cases 5 5
The highest motor development
at outcome evaluation
Unable to stand
Can’t move or only roll over 0 0 *P ¼ 0:0278
W sitting with no hand support 1 0
Crawl 0 1
Pull up 0 2
Walk with canes 0 2
Total 1* 5*
Able to stand or walk
Stand still# 2 0
Walk 2 0
Total 4* 0*
*P ¼ 0:0278, cases who could stand still or walk were significantly
more in the full training group than in the insufficient training group. #Able
to stand for 5 s with or without short braces.
position (Fig. 3(1)). He was regarding toys at that stage,
indicating good cognitive function. He also had extended
upper extremities with fisting. In the prone position at 2
months corrected age, he exhibited an opisthotonic posture
with excessive head extension (Fig. 3(2)). The family did
not wish to start physiotherapy at that time, but preferred
only to be followed up. At 1 year and 3 months of age, he
was able to pull himself up, but exhibited a round back and
tiptoeing (Fig. 3(3)). Physiotherapy was started at that time
because the family noticed the abnormal posture, and
Fig. 2. Case report 1 – full training (the brain MRI scan: Fig. 1b). The
neither twin had started walking. He started crawling at 1 child shown in this figure was born at 30 weeks gestational age
year 4 months of age. He was pulling himself up after 1 weighing 1680 g. At a corrected age of 1 month, the child was unstable
month of intensive training, even without tiptoeing. His when supine, and exhibited a frequent Moro reflex (1). Ventral
posture revealed bilateral valgus (Fig. 3(4)). The child could suspension reaction at 1 month of corrected age revealed asymmetric
stand still for only 1 s at 2 years 6 months of age (Fig. 3(5)). head position, flexed arms, bilateral fisting and extended legs with ankle
plantar flexion (2). This child was diagnosed to be at risk of motor
He started walking sideways with support at this time. He impairment, and physiotherapy was recommended. In the supine
underwent surgery for internal strabismus at 2 years 3 position at a corrected age of 2 months, the child exhibited a fencing
months of age. Until this time the mother had continued full position (extended left extremities and flexed right extremities). The
training for both twins. When he had reached this motor head was inclined to the right with bilateral fisting and a left side ankle
equinus position (3). In the prone position (corrected age 2 months), the
level, the family wanted to stop training as the other twin
child exhibited neck stiffness without head rolling or head extension,
had started walking and the family expected him to walk and flexing of the elbows with fists and extended legs with tiptoeing
without further training. One and a half years later, at 4 (4). At 1.5 years of age the child could pull herself up on her mother’s
years of age, the family wanted to start training again. At knees, at which time she had a round and hypotonic trunk and extended
that time, his pulling up pattern was hip flexion, internal legs with ankle plantar flexion (5). She started crawling at 1 year 5
months of age. The child was pulling herself up at 2 years and 9
rotation, and adduction of both legs with tiptoeing
months of age with severe tiptoeing on both legs (6). She started
(Fig. 3(6)). He wanted to walk sideways relying on support walking sideways with support at 2 years 5 months of age. At that time
but it was not very effective. At 4 years 5 months of age, she could touch the floor with both heels when told to (7). She could
after 5 months of training, he could pull himself up without stand still for 5 s at 5 years 7 months of age, although there was still
tiptoeing (Fig. 3(7)). At 4 years 7 months of age, he could slight flexion at the hips and knees and foot weakness (8). She started
walking at 5 years 10 months of age, still with tiptoeing. At 6 years 6
walk sideways without tiptoeing and with decreased months of age she could walk, even outside, with normal shoes
adduction of bilateral legs (Fig. 3(8)), but he still relied on although she was still tiptoeing. At this time, the mother was still
furniture to walk sideways. We noticed that the pace of administering physiotherapy with the Vojta Method three times daily,
change in this child, namely disappearing of tiptoeing, was and the child attended a normal school (9 and 10).

7. 124 T. Kanda et al. / Brain & Development 26 (2004) 118–126
much slower when physiotherapy was restarted at 4 years of
age than when physiotherapy was first started at 1 year and 3
months of age. At 5 years 2 months of age, he could stand
still for only 1 s, and his family discontinued training at this
point. The total training time was 2 years 5 months. He
never walked unassisted or stood for 5 s, but did later walk
with canes and attended a special school. No cognitive
deficits were diagnosed in this child.
4. Discussion
This was a non-randomized study in which the motor
development of two groups of children with abnormal
neurologic examinations in infancy and abnormal findings
on brain MRI were compared. We were able to assess
outcomes in children whose families did not choose to
receive therapies or who received insufficient therapy
because our hospital is the regional training and medical
center for children with cerebral palsy and other motor
disorders in the Kyoto Prefecture. The hospital operates a
kindergarten for disabled children and physicians provide
ongoing evaluations of health, neurological and develop-
mental status. This allowed us to follow the development of
these children for approximately 5 years after assessment as
infants and initiation of therapy.
All of the patients in the study group exhibited similar
Fig. 3. Case report 2 – insufficient training (the brain MRI scan: Fig. 1g).
types and severity of abnormal findings on MRI regardless
The child shown in this figure was one of twins born at 31 weeks gestation
and weighed 1562 g. At a corrected age of 2 months, he showed no of whether they received full or partial physiotherapy.
asymmetry of the neck and extremities in the supine position (1). He was Clinically, they demonstrated signs of spastic diplegic
regarding toys at that stage, indicating good cognitive function. He also had cerebral palsy consistent with premature birth of less than
extended upper extremities with fisting. In the prone position at 2 months 33 weeks gestation and a birth weight of less than 2000 g.
corrected age, he exhibited an opisthotonic posture with excessive head
The division of subjects into two groups was based on
extension (2). The family did not wish to start physiotherapy at that time,
but preferred only to be followed up. At 1 year and 3 months of age, he was parental participation in therapy. Since this occurred before
able to pull himself up, but exhibited a round back and tiptoeing (3). the acquisition of ‘rolling over’ in the study subjects,
Physiotherapy was started because the family noticed the abnormal posture, subsequent motor milestone development was not a factor in
and neither twin had started walking. He started crawling at 1 year 4 months assigning subjects to treatment groups.
of age. He was pulling himself up after 1 month of intensive training, even
In the group which received physiotherapy from the first
without tiptoeing. His posture revealed bilateral valgus (4). The child could
stand still for only 1 s at 2 years 6 months of age (5). He started walking few months of life until the end of the study period (FT),
sideways with support at this time. He underwent surgery for internal four of five cases acquired the ability to stand or walk. The
strabismus at 2 years 3 months of age. Until this time the mother had difference in the ability to achieve these motor milestones
continued full training for both twins. When he had reached this motor between this group and the group that received partial
level, the family wanted to stop training as the other twin had started
therapy (IT) was statistically significant. This difference
walking and the family expected him to walk without further training. One
and a half years later, at 4 years of age, the family wanted to start training persisted 2.5 years after the study period, suggesting that a
again. At that time, his pulling up pattern was hip flexion, internal rotation, follow-up evaluation after 5 years of therapy was appro-
and adduction of both legs with tiptoeing (6). He wanted to walk sideways priate for determining the effects of this treatment on motor
relying on support but it was not very effective. At 4 years 5 months of age, development.
after 5 months of training, he could pull himself up without tiptoeing (7). At
The Vojta Method of treatment which was used for this
4 years 7 months of age, he could walk sideways without tiptoeing and with
decreased adduction of bilateral legs (8), but he still relied on furniture to study is a program which employs isometric strengthening
walk sideways. We noticed that the pace of change in this child, namely techniques [4] through tactile stimulation [5] to encourage
disappearing of tiptoeing, was much slower when physiotherapy was the development of normal movement patterns in children
restarted at 4 years of age than when physiotherapy was first started at 1 with brain damage who are at risk for developing cerebral
year and 3 months of age. At 5 years 2 months of age, he could stand still for
palsy [3]. Extensive parent involvement in therapy is an
only 1 s, and his family discontinued training at this point. The total training
time was 2 years 5 months. He never walked unassisted or stood for 5 s, but integral part of this treatment due to the demands for
did later walk with canes and attended a special school. No cognitive multiple daily therapy sessions. Although parental partici-
deficits were diagnosed in this child. pation is known to be a characteristic of the most effective

8. T. Kanda et al. / Brain & Development 26 (2004) 118–126 125
programs [6], it is difficult to measure especially in the long- or per month [12,14,16]. The amount of therapy provided in
term implementation of therapy. The ability of parents to the home by the parents or other trained individuals is not
follow through was documented in this study by the usually reported because of the difficulty with confirming
individual therapist who was responsible for the care of compliance with the treatment regimen. The quality,
each child. amount, and consistency of home therapy administered by
Very early initiation of therapy within the first 3 months trained caregivers is rarely documented or even mentioned
of age corrected for prematurity was possible due to the role in studies that look at outcomes from therapy. Methods for
of our hospital in the early infancy screening program in encouraging compliance with home based treatments are
Kyoto Prefecture. This is a unique feature of this report. In needed in future studies of therapeutic effectiveness. In this
an evidence based summary of research on the neurodeve- study an attempt was made to closely monitor parental
lopmental treatment (NDT) method, no studies were found follow through with therapies over a prolonged time.
that reported results of treatment started under 5 months of The ‘dose-response’ relationship between therapy fre-
age and only four out of the 21 reported studies initiated quency and location with outcomes is just starting to be
treatment under 12 months of age [7]. critically evaluated. The review by Butler and Darrah [7]
We think that consistent sensory stimulation during the did not find that more intensive therapies conferred greater
period of postnatal cerebellar myelination that occurs benefit. These authors concluded that more studies are
between 0 and 12 – 18 months [8,9] helps to establish required (i.e. second generation studies) that specifically
normal anti-gravity motor patterns before abnormal patterns evaluate the time of onset of therapy and the intensity of
can develop. An initial goal of this therapy method is to treatments.
concentrate on spinal alignment to improve truncal This is a report of a case series with prolonged follow-up
hypotonia in children with cerebral palsy. of a group of children with abnormal neurological exams
The duration of treatment is another important factor for and brain MRI abnormalities associated with spastic
assessing the efficacy of therapy programs. No literature diplegic cerebral palsy. They had similar abnormal move-
exists as to how long therapy needs to be given to achieve the ment patterns of legs and similar delay of acquisition of
optimal outcome. Randomized trials conducted over a period motor development from rolling over to unsupported sitting
of about 1 year may be insufficient to prove treatment effects in infancy. Therapy was initiated before 6 months of age.
[10,11]. Piper et al. reported that more intensive and longer It is an attempt to begin to answer some of the questions
trials of early intervention would be necessary to demonstrate raised about the intensity, duration, and content of
efficacy of physiotherapy [10]. The longest duration of therapeutic programs for improving outcomes in children
treatment in the review of NDT by Butler and Darrah was with spastic diplegic cerebral palsy. Early intervention
only 21 months [7]. Five of the 21 studies in this report had consisting of long-term intensive physiotherapy may result
treatment durations from 6 to 12 months and the remaining in statistically better motor development of prematurely
15 had durations of treatment of under 6 months. In our study, born children with spastic diplegia than less intensive
a treatment duration of 5 years was possible mainly due to the shorter term therapy.
unique setting in which these programs were conducted that We speculate that the difference in the highest motor
included a stable community and a developmental resources development level achieved was related to the early onset of
center that provided ongoing services to these families. The therapy and its effects on cerebellar myelination. As with
prolonged duration of treatment in this study may be one of many studies that evaluate the effectiveness of therapy,
the reasons for the better motor outcomes observed in the FT collecting and following a group of similar children for a
group as suggested by Piper et al. [10]. long enough period of time to observe differences related to
A third factor that may influence the effectiveness of therapy is extremely difficult. Additional studies are needed
therapy is the intensity of treatment. Some studies have that identify comparable children at risk for developing
demonstrated that programs with a higher frequency of spastic diplegic cerebral palsy at early ages and follow them
treatments result in better outcomes [12,13] while other for prolonged periods of time while enrolled in a consistent
studies have shown no differences [14]. Concern has been therapy program.
raised that too much therapy is stressful and tiring for the
providers and may lead to low compliance and ineffective
treatment [14]. Trahan and Malouin investigated the effects References
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