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 2003Abstract The objective of this study is to determine the clinical effectiveness of early onset long-term intensive physiotherapy on motordevelopment 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 gestationalage of less than 33 weeks and a birth weight of less than 2000 g. Brain magnetic resonance imaging revealed periventricular white matterinjury in nine subjects and moderate grade bilateral porencephaly in one. Five completed a full course of training of 52 months (mean), twodid not receive therapy, and three received an insufﬁcient course of therapy. The study was conducted at the Regional Center for Childrenwith Disabilities including outpatient clinics and a school for children with special needs. The Vojta Method was used, which is an extensivefamily oriented physiotherapy program which uses isometric strengthening of muscles with tactile stimulation. Subjects were evaluated forthe highest motor developmental level at the outcome evaluation 59 months (mean) after initiation of therapy. Four of the ﬁve who completedtraining 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 therapyinitiation. This was a statistically signiﬁcant difference (P ¼ 0:0278). A consistently applied physiotherapy program resulted in better motoroutcomes 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 study1. 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 forprograms for premature infants at risk for motor problems cerebral palsy. Infants who received therapy were comparedis a difﬁcult task. Although a randomized clinical trial to those who either received inadequate therapy or nowould provide the most deﬁnitive proof, there are many therapy.obstacles to successfully completing studies of this kindover a prolonged period of time . This report describes a 1.1. Backgroundgroup of children who were offered therapy starting at a veryyoung age and then consistently followed for 5 years. Motoroutcomes 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: email@example.com (T. Kanda). government or private medical insurance. Aid is given if0387-7604/$ - see front matter q 2003 Elsevier B.V. All rights reserved.doi:10.1016/S0387-7604(03)00111-6
T. Kanda et al. / Brain & Development 26 (2004) 118–126 119the 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 ageat 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 orPrefecture, which includes a population of 2.56 million older. Physiotherapy was discontinued and assessmentspeople. Referrals come from hospitals and regional health were conducted once a year until the age of 6 if a childcare centers that have government contracts to screen started walking in a stable way, without evidence of toehealthy infants and toddlers. walking or valgus. 2.4. Follow-up study2. Method The total number of patients who were ﬁrst assessed in2.1. 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 theyPhysical therapy was recommended based on Vojta’s motor reached a corrected age of 3 months. Assessment of therisk criteria or central coordination disturbance criteria, study parameter items from patients’ charts was done inwhich includes spontaneous movements, postural reactions October 1998, and additional information was obtained byand primitive reﬂexes . The decision to recommend calling families or making assessments at the hospital iftherapy was based on either the initial consultation or a necessary. The decision as to whether or not patients hadrepeat examination within 1 month of the ﬁrst consultation. received continuing full physiotherapy depended on information provided by the therapist in charge. Not2.2. 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 therapeuticVojta . Therapists who had completed an 8 week training exercises during return visits to the hospital. To providecourse and who had at least 5 years of clinical experience appropriate interventions at the correct locations on thewith this method participated in this study. The same child for a sufﬁcient duration requires continuing practicetherapist was responsible for therapy for each child in this much like practicing playing the piano. It is difﬁcult toreport throughout the course of treatment. Movement and maintain the proper skills if sufﬁcient intervention is notreﬂex patterns were taught to the parents over a 3 month being done at home.period which included 1 month of inpatient teaching duringwhich parents received daily training about the Vojta 2.5. Selection of the study groupMethod, 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 initiallyperiod and after the patterns were learned. Parents evaluated before a corrected age of 3 months, 34 weresubsequently visited the hospital for instruction from followed-up until at least 3 years of age. Two childrentherapists 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 dividedtreat their children three to four times daily. Fully treated into three groups by the neurological ﬁndings within 1 yearchildren received 90– 120 sessions per month. Therapy after the ﬁrst evaluation. The most severe group includedsessions took 30 min or more to complete, which included three children who showed very abnormal muscle tone withtime for displays of affection and afﬁrmation to the children paucity of spontaneous movements and seven abnormalat 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 inwere provided after 1 year of age. They included this group would not be expected to roll over or crawl. Theseoccupational therapy and feeding therapies. children had greater than eight abnormal primitive reﬂexes as described by Vojta . Twenty-one children had a slight2.3. 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 , abnormal primitive reﬂexes . These children werewhich included voluntary movements, postural reactions classiﬁed as the ‘mild group’. Six of these children wereand 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 onpostural reactions. Each patient was assessed once monthly their toes or other motor disturbances during the study
120 T. Kanda et al. / Brain & Development 26 (2004) 118–126period. The remaining ten children had patterns of Table 1spontaneous movement, postural reactions, primitive Characteristics of the study groupreﬂexes, and muscle tone abnormalities which were Item Group Statisticsintermediate between the severe and mild groups. Eight ofthese children were diagnosed as at risk for cerebral palsy Full training Insufﬁcientbased on these neurological ﬁndings. Two of these children trainingshowed increased asymmetry of spontaneous movements, Number of cases 5 5postural 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 . 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 developmentmonths), 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 0disturbance. All ten cases were diagnosed with spastic 70 ^ DQ . 50 0 0 50 ^ DQ . 30 1 2diplegia at the time of the outcome evaluation at a mean age 30 ^ DQ 0 0of 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 coordinationthree children with a birth weight , 1500 g in the FT group disturbanceand two in the IT group. Mental development in the two Diagnosis at the time ofgroups 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 visualdisorder. One child in the FT group had microcephaly. Allcases showed distinct neurological signs at the ﬁrst clinical Table 2examination (Table 2). All except one case showed paucity Neurological signs before corrected 3 months of age at the ﬁrst clinicalof spontaneous movements of legs (Table 3). check Rolling over, supported sitting, and unsupported sittingdid not occur at signiﬁcantly different ages between the Group Cases Corrected Corrected Corrected 0 months 1 months 2 monthsgroups that received full therapy or partial therapy (Mann–Whitney U-test; Table 4). Rolling over developed after the Full training 1 b, d, f, iage when parents decided to discontinue physiotherapy or 2 a, b, c, h, iwere 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, ileucomalacia in four children in the FT group and ﬁve 5 a, b, h, i Insufﬁcient 6 b, d, hchildren in the IT group. One child in the FT group had trainingmoderate grade bilateral porencephaly. 7 a, b, d, e, i The socioeconomic background was similar for both 8 a, b, c, hgroups. 9 c, d, h, I 10 a, b, g, h, i (a) Hypertone; (b) all the seven postural reactions by Vojta are3. 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 muchThe mean age at outcome evaluation was 56 months for the extended elbows; (h) reﬂex of foot grasp is inhibited or delayed either onFT group and 67 months for the IT group. The mean both or one side; (i) other pathologic primitive reﬂexes are found.
T. Kanda et al. / Brain & Development 26 (2004) 118–126 121Table 3 Table 5Patterns of spontaneous movements of legs before corrected 3 months of Findings of magnetic resonance in the study groupage at the ﬁrst clinical check Findings of magnetic resonance Group StatisticsGroup Cases Corrected Corrected Corrected 0 months 1 months 2 months Full training Insufﬁcient trainingFull 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 casesseen; (c) plantar ﬂexion is seen at the extended legs; (d) hyperabduction and þP 0 cases 0 caseshyperrotation outward of legs are seen; (e) only bilateral total ﬂexion or þ AP 4 cases 2 casestotal extension of legs are seen; (f) stepping like movement is seen in both alegs; (g) plantar ﬂexion is seen while legs are extended or ﬂexed. Complicated. b Peritrigonal white matter reduced in both hemispheres is evaluated byduration of training was longer in the FT group (52 months) comparing the normal control in the same age which appears in thethan 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 lateralpalsy and one as at risk for central coordination disturbance ventricles; þA means that the high intensity area was also seen at thein 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 physiotherapyextremity movement patterns and sought early attention. occurred was in a family where one twin survived andThis also occurred in two other families where twins or the other died. This mother dedicated herself to providingtriplets 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 physiotherapyTable 4 in the IT group varied. They included severe asthma inMonths of age motor development acquired and months of age when one child, insufﬁcient time or commitment by the parentsparents decided to discontinue physiotherapy or were unable to continue to participate in administering the therapy, religiousfull physiotherapy convictions, and reluctance by the parent to accept theGroup 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 weeksInsufﬁcient 6 n: 6, r:15, s:9, u:26 8, asthma gestational age weighing 1680 g. The brain MRI scan (Axialtraining 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 developmentacquired between two groups was not signiﬁcant by Mann –Whitney U-test At a corrected age of 1 month, the child was unstable whenin 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
122 T. Kanda et al. / Brain & Development 26 (2004) 118–126Fig. 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 posteriorhorns 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 stageof periventricular leucomalacia around anterior, body and posterior horns. There was loss of myelinated white matter along the bilateral body and posteriorhorns 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 posteriorhorns 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 FSE3,500/125 image at 1 year and 2 months of age shows irregular ventricular dilatation at the right posterior horn and also shows end stage of periventricularleucomalacia around body and posterior horns. There was loss of myelinated white matter at the right posterior horn of the periventricle. It also shows smallcavities 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 irregularventricular dilatation at the left side and also shows end stage of periventricular leucomalacia around anterior, body and posterior horns. There was loss ofmyelinated white matter at the bilateral body and posterior horns of the periventricle. It also shows small cavities along lateral sides of the body of lateralventricles. (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 hornsand 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 5months of age shows irregular ventricular dilatation at bilateral posterior horns and also shows end stage of periventricular leucomalacia at body and posteriorhorns. 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 imageat 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 ﬁgureat 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 periventricularleucomalacia 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 theﬁsting 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. Atmotor impairment, and physiotherapy was recommended. In 6 years 6 months of age she could walk, even outside, withthe 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 theﬂexed right extremities). The head was inclined to the right Vojta Method three times daily, and the child attended awith 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 traininghead extension, and ﬂexing of the elbows with ﬁsts andextended legs with tiptoeing (Fig. 2(4)). At 1.5 years of age The child shown in Fig. 3 was one of twins born at 31the child could pull herself up on her mother’s knees, at weeks gestation and weighed 1562 g. The brain MRI scanwhich time she had a round and hypotonic trunk and (Axial SE 2,000/120 image) at 4 years and 5 months of ageextended legs with ankle plantar ﬂexion (Fig. 2(5)). She is shown in Fig. 1g. It shows irregular ventricular dilatationstarted crawling at 1 year 5 months of age. The child was at bilateral posterior horns and also shows end stage ofpulling 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 thesideways 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 nocommand (Fig. 2(7)). She could stand still for 5 s at 5 years asymmetry of the neck and extremities in the supine
T. Kanda et al. / Brain & Development 26 (2004) 118–126 123Table 6Motor developmental level in the diplegic group at the time of outcomeevaluationItem Group Statistics Full Insufﬁcient training trainingNumber of cases 5 5The highest motor developmentat 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 signiﬁcantlymore in the full training group than in the insufﬁcient training group. #Ableto 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 extendedupper extremities with ﬁsting. In the prone position at 2months corrected age, he exhibited an opisthotonic posturewith excessive head extension (Fig. 3(2)). The family didnot wish to start physiotherapy at that time, but preferredonly to be followed up. At 1 year and 3 months of age, hewas able to pull himself up, but exhibited a round back andtiptoeing (Fig. 3(3)). Physiotherapy was started at that timebecause the family noticed the abnormal posture, and Fig. 2. Case report 1 – full training (the brain MRI scan: Fig. 1b). Theneither twin had started walking. He started crawling at 1 child shown in this ﬁgure was born at 30 weeks gestational ageyear 4 months of age. He was pulling himself up after 1 weighing 1680 g. At a corrected age of 1 month, the child was unstablemonth of intensive training, even without tiptoeing. His when supine, and exhibited a frequent Moro reﬂex (1). Ventralposture revealed bilateral valgus (Fig. 3(4)). The child could suspension reaction at 1 month of corrected age revealed asymmetricstand 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 motorHe started walking sideways with support at this time. He impairment, and physiotherapy was recommended. In the supineunderwent surgery for internal strabismus at 2 years 3 position at a corrected age of 2 months, the child exhibited a fencingmonths of age. Until this time the mother had continued full position (extended left extremities and ﬂexed right extremities). Thetraining 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), thelevel, 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 tiptoeingwithout 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’syears of age, the family wanted to start training again. At knees, at which time she had a round and hypotonic trunk and extendedthat 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 9rotation, 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 timebut 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 couldafter 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 stilltiptoeing (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 6walk sideways without tiptoeing and with decreased months of age she could walk, even outside, with normal shoesadduction of bilateral legs (Fig. 3(8)), but he still relied on although she was still tiptoeing. At this time, the mother was stillfurniture 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).
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 similarFig. 3. Case report 2 – insufﬁcient training (the brain MRI scan: Fig. 1g). types and severity of abnormal ﬁndings on MRI regardlessThe child shown in this ﬁgure was one of twins born at 31 weeks gestationand 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 diplegicregarding toys at that stage, indicating good cognitive function. He also had cerebral palsy consistent with premature birth of less thanextended 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 onextension (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 beforeable 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 inand 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 ﬁrstwithout tiptoeing. His posture revealed bilateral valgus (4). The child couldstand 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. Thestrabismus at 2 years 3 months of age. Until this time the mother had difference in the ability to achieve these motor milestonescontinued full training for both twins. When he had reached this motor between this group and the group that received partiallevel, the family wanted to stop training as the other twin had started therapy (IT) was statistically signiﬁcant. This differencewalking and the family expected him to walk without further training. Oneand 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 aagain. 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 motorrelying 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 this4 years 7 months of age, he could walk sideways without tiptoeing and withdecreased adduction of bilateral legs (8), but he still relied on furniture to study is a program which employs isometric strengtheningwalk sideways. We noticed that the pace of change in this child, namely techniques  through tactile stimulation  to encouragedisappearing of tiptoeing, was much slower when physiotherapy was the development of normal movement patterns in childrenrestarted at 4 years of age than when physiotherapy was ﬁrst started at 1 with brain damage who are at risk for developing cerebralyear and 3 months of age. At 5 years 2 months of age, he could stand still for palsy . Extensive parent involvement in therapy is anonly 1 s, and his family discontinued training at this point. The total trainingtime was 2 years 5 months. He never walked unassisted or stood for 5 s, but integral part of this treatment due to the demands fordid 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
T. Kanda et al. / Brain & Development 26 (2004) 118–126 125programs , it is difﬁcult to measure especially in the long- or per month [12,14,16]. The amount of therapy provided interm implementation of therapy. The ability of parents to the home by the parents or other trained individuals is notfollow through was documented in this study by the usually reported because of the difﬁculty with conﬁrmingindividual 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 mentionedof age corrected for prematurity was possible due to the role in studies that look at outcomes from therapy. Methods forof our hospital in the early infancy screening program in encouraging compliance with home based treatments areKyoto Prefecture. This is a unique feature of this report. In needed in future studies of therapeutic effectiveness. In thisan evidence based summary of research on the neurodeve- study an attempt was made to closely monitor parentallopmental 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 betreatment under 12 months of age . critically evaluated. The review by Butler and Darrah  We think that consistent sensory stimulation during the did not ﬁnd that more intensive therapies conferred greaterperiod of postnatal cerebellar myelination that occurs beneﬁt. These authors concluded that more studies arebetween 0 and 12 – 18 months [8,9] helps to establish required (i.e. second generation studies) that speciﬁcallynormal anti-gravity motor patterns before abnormal patterns evaluate the time of onset of therapy and the intensity ofcan 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-uphypotonia 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 spasticassessing 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 ofoptimal outcome. Randomized trials conducted over a period motor development from rolling over to unsupported sittingof 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 questionstrials of early intervention would be necessary to demonstrate raised about the intensity, duration, and content ofefﬁcacy of physiotherapy . The longest duration of therapeutic programs for improving outcomes in childrentreatment in the review of NDT by Butler and Darrah was with spastic diplegic cerebral palsy. Early interventiononly 21 months . Five of the 21 studies in this report had consisting of long-term intensive physiotherapy may resulttreatment durations from 6 to 12 months and the remaining in statistically better motor development of prematurely15 had durations of treatment of under 6 months. In our study, born children with spastic diplegia than less intensivea 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 motorincluded a stable community and a developmental resources development level achieved was related to the early onset ofcenter that provided ongoing services to these families. The therapy and its effects on cerebellar myelination. As withprolonged 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 agroup as suggested by Piper et al. . 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 neededtherapy is the intensity of treatment. Some studies have that identify comparable children at risk for developingdemonstrated that programs with a higher frequency of spastic diplegic cerebral palsy at early ages and follow themtreatments result in better outcomes [12,13] while other for prolonged periods of time while enrolled in a consistentstudies have shown no differences . Concern has been therapy program.raised that too much therapy is stressful and tiring for theproviders and may lead to low compliance and ineffectivetreatment . Trahan and Malouin investigated the effects Referencesof periods of intensive therapy (four times/week for 4weeks) combined with periods of no therapy (8 weeks) over  Butler C, Chambers H, Goldstein M, Harris S, Leach J, Campbell S.a 6 month period in a convenience sample of ﬁve children Evaluating research in developmental disabilities: a conceptualwith severe cerebral palsy . This program resulted in framework for reviewing treatment outcomes. Dev Med Child Neurol 1999;41:55 –9.improvements of motor function that were maintained over ¨ ¨  Vojta V. Die zerebralen Bewegungsstorungen im Sauglingsalter.the ‘rest’ periods. ¨ ¨ Frudiagnose und Fruhtherapie. Stuttgart: Enke; 1988. Most studies that look at the intensity of therapies have  Vojta V, Peters A. Das Vojta-Prinzip. Muskelspiele in Reﬂexbewe-compared the number of center based treatments per week gungen und motorischer Ontogenese. Berlin: Springer; 1996.
126 T. Kanda et al. / Brain & Development 26 (2004) 118–126  Damiano DL, Abel MF. Functional outcomes of strength training in  Wright T, Nicholson J. Physiotherapy for the spastic child: an spastic cerebral palsy. Arch Phys Med Rehabil 1998;79:119–25. evaluation. Dev Med Child Neurol 1973;15:146–63.  Feldman R, Eidelman AI. Intervention programs for premature  Mayo NE. The effect of physical therapy for children with motor infants. How and do they affect development? Clin Perinatol 1998;25: delay and cerebral palsy. A randomized clinical trial. Am J Phys Med 613–26. Rehabil 1991;5:258–67.  Shonkoff JP, Hauser-Cram P. Early intervention for disabled infants  Bower E, McLellan DL. Effect of increased exposure to physiother- and their families: a quantitative analysis. Pediatrics 1987;80: apy on skill acquisition of children with cerebral palsy. Dev Med 650–8. Child Neurol 1992;34:25 –39.  Butler C, Darrah J. Effects of neurodevelopmental treatment (NDT)  Bower E, Michell D, Burnett M, Campbell MJ, McLellan DL. for cerebral palsy: an AACPDM evidence report. Dev Med Child Randomized controlled trial of physiotherapy in 56 children with Neurol 2001;43:778 –90. cerebral palsy followed for 18 months. Dev Med Child Neurol 2001;  Yakoviev PI, Lecours AR. The myelogenic cycles of regional 43:4–15. maturation of the brain. Regional development of the brain in early  Trahan J, Malouin F. Intermittent intensive physiotherapy in children life. Oxford: Blackwell; 1967. p. 3 –7. with cerebral palsy: a pilot study. Dev Med Child Neurol 2002;44:  Barcovich AJ. Pediatric neuroimaging. New York: Raven Press; 1990. 233 –9. p. 20.  Reddihough D, Bach T, Burgess G, Oke L, Hudson I. Comparison Piper MC, Kunos VI, Willis DM, Mazer BL, Ramsay M, Silver KM. of subjective and objective measurements of movement perform- Early physical therapy effects on the high-risk infant: a randomized ance of children with cerebral palsy. Dev Med Child Neurol 1991; controlled trial. Pediatrics 1986;78:216–24. 33:578–84.