Modified Constraint-Induced Movement Therapy for Children with Perinatal Brachial Plexus Palsy

(1)

Modi ﬁ ed constraint-induced movement therapy during

hospitalization in children with perinatal brachial plexus palsy:

A randomized controlled trial

Beyhan Eren MD

^a^,^*

, Evrim Karada g Sayg ı MD

^b

, Duygu Tokgöz PhD

^b

, Merve Akdeniz Leblebicier MD

^c

aPhysical Medicine and Rehabilitation Department, Marmaris State Hospital, Mugla, Turkey

bPhysical Medicine and Rehabilitation Department, School of Medicine, Marmara University, Istanbul, Turkey

cPhysical Medicine and Rehabilitation Department, School of Medicine, Dumlupınar University, Kütahya, Turkey

a r t i c l e i n f o

Article history:

Received 17 December 2018 Received in revised form 29 September 2019 Accepted 31 December 2019 Available online xxx

Keywords:

Constraint-induced movement therapy Upper extremity function

Perinatal brachial plexus palsy

a b s t r a c t

Study Design:Prospective single-blind, randomized controlled study.

Introduction:Children with perinatal brachial plexus palsy (PBPP) have motion limitations in the affected upper extremity. Modiﬁed constraint-induced movement therapy (mCIMT) is one of the treatment op- tions used for the improvement of the function of the affected limb.

Purpose of the Study:The purpose of this study was to compare the effect of mCIMT and conventional therapy in improving active range of motion (ROM) and functional use of the affected upper extremity in children with PBPP with injuries to upper and middle trunks in the hospital environment.

Materials:26 patients received conventional rehabilitation program (control group) and 13 patients participated in a mCIMT program (study group). Children had a mean age 56.3 months (range 4-10 years). The mCIMT included 1 hour therapy sessions emphasizing the affected arm use for 14 consecutive days during hospitalization. Their normal arm was also constrained for 6 hour per day. All the patients were assessed at the baseline, one day, one month, and three months after completion of therapy using active ROM, active movement scale, hand dynamometer, box and blocks test.

Results:The mCIMT group improved more than the control group in shoulder internal rotation, forearm supination, elbowﬂexion active ROMs, hand grip strength, and in upper extremity function.

Conclusion:mCIMT has a potential to promote functional gains for children with PBPP; this approach should be widely applied within routine clinical practice.

Introduction

Perinatal brachial plexus palsy (PBPP) is partial or total paralysis of the upper extremity related to an injury on the brachial plexus, C5-T1, that occurs before, during, or just after birth.¹ The rate of PBPP is approximately 1.5 of 1000 live births.²The functional limitation at upper extremity can restrict participation of child in daily living and social activities.^3,4 To improve joint range of motion

(ROM), muscle strength and function of the affected extremity conservative or surgical treatment should be applied.⁵ The constraint-induced movement therapy (CIMT) may be one of these conservative treatments.

The CIMT takes place among promising rehabilitation in- terventions for improving upper extremity function in patients with neurological dysfunction.^6-8The restriction of the unaffected upper limb more than 90% of waking hours by using a sling or mitt, along with application of shaping techniques and/or intensive motor activities more than 3 hours per day during at least two consecutive weeks are the two main features of the CIMT.⁹

Modiﬁed CIMT (mCIMT) is different from the CIMT protocol.^10-

13These differences include the use of restriction methods alter- native to casting, decreasing the duration of restriction and intensive exercise program. Besides, the therapy can be applied in the environment child is familiar with and as part of a play activity.¹²

Shepherd observed that although muscle function of some children was restored, they persisted in not using their arms.

This study was presented at 2013 25th Annual European Academy of Childhood Disability Conference, October 10 to 12, 2013, Newcastle, UK.

Conﬂict of interest: All named authors hereby declare that they have no conﬂicts of interest to disclose.

Funding: This project was supported by the Scientiﬁc Research Project Coordi- nation Unit (BAPKO) of Marmara University, Istanbul, Turkey. Project number: SAG- C-TUP-130612 to 0203.

*Corresponding author. Physical Medicine and Rehabilitation Department, Marmaris State Hospital, Yeniçevre Yolu 320 Street, Marmaris, Mugla, Turkey. Tel.:

þ905053888014; fax:þ90 252 413 44 46.

E-mail address:[email protected](Beyhan Eren).

Contents lists available atScienceDirect

Journal of Hand Therapy

j o u r n a l h o m e p a g e : w w w . j h a n d t h e r a p y . o r g

https://doi.org/10.1016/j.jht.2019.12.008

(2)

Consequently, the treatment of children with PBPP is suggested to be based on CIMT principles.¹⁴There is increasing evidence that peripheral plasticity, spinal cord plasticity, and cortical plasticity play an important role in the recovery process of PBPP.^15-17 In infants with PBPP, the “learned non-use”also named as developmental disregard may be conductive to a failure to develop functional use of the affected extremity. In the light of these facts, use of the affected limb in daily activities can overcome developmental disregard.^11,17

CIMT has been shown to improve upper limb function in conditions affecting central nervous system such as cerebral palsy (CP) and stroke-induced hemiparesis in children.^9,18,19 There have been no randomized control trials investigating CIMT for PBPP, but there are some recent case reports that are suggestive of a potential beneﬁcial effect.^20-24To what extent CIMT may be effective in conditions arising from injury to the peripheral nerves is not understood.

Purpose of the study

The primary objective of this exploratory study was to compare the effect of mCIMT and conventional therapy on the affected extremity active ROM and function of children with PBPP in the hospital environment. We propose that mCIMT will beneﬁt children with upper and middle trunkeinvolved PBPP who are at risk for developmental disregard and subsequent motion dysfunction.

Also, treating and observing children in a hospital environment would increase study participation.

Methods Trial design

This single-blind randomized study included 39 patients who were being followed up in the pediatric rehabilitation clinic in Marmara University Hospital and diagnosed with an upper and middle trunk PBPP (Narakas classiﬁcation type II). Patients and their parents were notiﬁed about the study both verbally and in written form. Children were included in the study after approval and consentment of the parents. Outcome measures were performed before the intervention and 1 day, 1 month, and 3 months after the intervention. This study was approved by the ethical committee of Marmara University with the approval number of B.30.2.MAR.0.01.02/AEK/117.

Subjects

Children were eligible for inclusion if they were clinically diagnosed as PBPP with involvement of upper and middle trunks; had the capacity to understand and apply the directives;

had full passive ROMs in all motions at the shoulder, elbow, and wrist joints; had no restriction at activeﬁnger ROM and if they were aged between 4 and 10 years. Subjects were excluded if they had contracture in their affected upper extremity, cognitive impairment, visual problems, not enough postural stability, previous orthopedic or neurological surgery, or if they previously had CIMT.

Randomization and blinding

Randomization was performed by randomly assigning a number from one to six to patients who fulﬁlled the inclusion criteria. Pa- tients receiving numbers between one and four were part of the control group and patients receiving the numbersﬁve and six were part of the mCIMT intervention. Different physicians who were

unaware of the group allocation applied assessments of before and after treatments.

Intervention

The control group received standard conventional therapy to their affected extremity 1 hour a day for 14 consecutive days. An expert child physiotherapist performed the treatment in an outpatient clinic. Therapy involved shoulder, elbow, wrist active assistive and active ROM exercises with twenty repetitions, neuromuscular electrical nerve stimulation (20 minutes), and strengthening exercises to deltoid, triceps, biceps, supinator, and wrist extensor muscles. After children's parents have been trained, parents continued with same exercise program at home. Differ- ently, the mCIMT group has been hospitalized in a group of 2 or 3 people (they stayed in the hospital over 14 days). An intensive motor exercise program was applied to the affected limb of the mCIMT group during hospitalization 1 hour per day for 14 consecutive days. Their unaffected limbs were restricted with a custom-made static upper limb orthosis. Orthosis started from humerus distal 1/3 part down tofinger tips; elbow was positioned in 20offlexion, wrist was positioned in 10of extension, thumb was positioned in abduction, andfingers were positioned in sem- iflexion (Fig. 1). Children wore this orthosis 6 hour a day. Consid- ering that the child might get bored or frustrated, the daily restriction time was divided into two with an hour interspace.

Jebsen Taylor Hand Function Test (JTT) is being used for assessment of upper limb function such as grasp, release speed, and dexterity.²⁵The test involves activities similar to daily living activities. On this basis, JTT was used for the exercise play set. While children wore orthosis to their affected limb, they were asked to practice the seven activities of JTT andfinger step exercises with ten repetitions. Training program also involvedfine motor, gross motor, active shoulder, forearm and elbow active assistive and active ROM, and strengthening exercises with 20 repetitions each. If the patient was unable to perform the exercises individually, they were actively assisted; nevertheless the difficulty of the tasks was not increased.

Fig. 1.Custom-made static upper extremity orthosis that was used for healthy upper extremity constriction.

(3)

After completing the therapy, parents of the mCIMT group continued home exercises similar to those prescribed to the control group.

Assessments and outcome measures

The gender, age, and the affected side were determined. Natal history, when the pathology have been noticed, presence of clavicle fracture, age and program through which the child started to receive rehabilitation were also questioned.

All patients were evaluated at the baseline, 1 day after intervention, and then 1 and 3 months later. Assessments were carried out with upper limb active ROMs, Active Movement Scale, hand dynamometer, and Box and Blocks Test (BBT).

Shoulder, elbow, and wrist passive and active ROMs were measured with a Standard Baseline 12-inch plastic goniometer. In cases when patients were not able to understand simple orders, data were graded with toy stimulation or with the parent's help.

Shoulder (glenohumeral joint) is evaluated with the scapula sta- bilized during passive and activeﬂexion, abduction, internal, and external rotation. Shoulderﬂexion was assessed with trunk upright, arm actively elevated in sagittal plane, with the palm down.

Shoulder abduction ROM was measured with trunk upright, arm elevated actively in coronal plane, with thumb pointed the ceiling.

The angle between the lateral border of the scapula and the humerus was measured when the humerus is in maximum elevation with firm stabilization of the scapula onto the thorax. Shoulder external rotation was tested in supine position, hips and knees flexed to 45; arm was supported on the table in 90abduction and the elbowflexed to 90with wrist in neutral position. Then towel was placed under humerus to ensure neutral horizontal positioning. Shoulder internal rotation was measured in the prone position with the tested arm supported on the table in 90 abduction, the elbowflexed to 90with the wrist in neutral position. A towel roll was placed directly under the arm to ensure neutral horizontal positioning and to provide stabilization. Shoul- der external and internal rotation were also measured with the humerus in maximal adduction against the body and elbow at 90 offlexion. The arm is moved into external and internal rotation in the transverse plane. Elbowflexion and extension were defined by the angle between the humerus and the ulna on a profile view.

Forearm supination and pronation were deﬁned by angle between the dorsal side of the hand and the humerus line on a face view.

Dorsal and volar alignment technique was used for wristﬂexion and extension measurements.

Active Movement Scale is a rating scale for the quantiﬁcation of movement and evaluation of upper limb motor function in children with PBPP. It evaluates 15 joint movements (range of movement and muscle strength) from shoulder to hand on an 8-point scale (0 ¼ no muscle tone or contraction when gravity eliminated, 7¼full range against gravity). This measurement tool has moderate to excellent intra- and inter-rater reliability when used by experi- enced clinicians on children with PBPP between 1 month and 15

years of age. It has an excellent inter-observer reliability for elbow ﬂexion/extension, but moderate/poor for forearm supination and pronation.²⁶

BBT is a functional performance test of gross motor dexterity, which measures the number of blocks transferred from one side to other side of the box in 60 s. Subjects were seated so that the board and pegs were directly in front of the subject's centerline and within comfortable reach. Its validity at assessing upper extremity functional loss was tested in neurologic diseases such as stroke, multiple sclerosis, and traumatic brain injury. It has been stated that this highly reliable test could be used for assessing functional loss and treatment effect.²⁷

The hand grip strength was measured using a pneumatic hand dynamometer (Riester Dynatest) in kilogram-force per square centimeter (kgf∕cm²). Participants were seated with the shoulder at 0 abduction andﬂexion, the elbow at 90 ﬂexion, and were then instructed to grip and squeeze the device with a maximal effort for 3 s.

Patients completed two trials of BBT and hand grip strength with the affected hand. Trials were separated by a 1-minute rest period. Second of the two trials was used for analysis.

Size of study population

Because no study of this nature has been reported previously, the necessary sample was estimated from a power calculation based on functional improvement rate observed in connection with a previous study in children with CP.²⁸ The difference in the number of patients exposed to each modality (n¼13,n¼26) is because of two reasons. Within same period, the control group enables inclusion of a greater number of patients than the mCIMT group. In addition, methodological requirement to administer both modalities in parallel reduced the number of patients exposed to mCIMT mode because of the physical and human source available.

Statistical analysis

The data were analyzed using SPSS version 16.0 software for Windows. The main characteristics of patients were evaluated with descriptive studies. Analysis within groups was conducted by using Wilcoxon Ranks and Friedman tests. Continuous variables between the two groups were compared with ManneWhitney U test because the data did not follow a normal distribution. Bonferroni was used to correct multiple comparisons. AP-value <.05 was considered as statistically signiﬁcant.

Results

Characteristics of study population

Adherence to the study protocol was excellent. Thirty-nine patients completed the study, there were no dropouts. There was no signiﬁcant difference in demographic and clinical characteristics between the two groups (Table 1).

Table 1

Demographic and clinical characteristics of the study population

Demographic and clinical features mCIMT groupn¼13 Control groupn¼26 Signiﬁcance

Age (mo) (meanstandard deviation) 57.615 55.717 0.464

Gender (frequency, percentage) 0.113

Girl 9 (69.2%) 11 (42.3%)

Boy 4 (30.8%) 15 (57.7%)

Pvalue .166 .433

Affected side (frequency, percentage) 0.163

Right 10 (76.9%) 14 (53.8%)

Left 3 (23.1%) 12 (46.2%)

Pvalue .052 .695

(4)

Changes in active range of motion

There were no statistically signiﬁcant differences in active ROMs in all motions at the shoulder, elbow, forearm, and wrist between the two groups in the beginning. After three months of therapy, the

mCIMT group exhibited greater beneﬁts in shoulder internal rotation, elbow ﬂexion, and forearm supination active ROMs as compared with the control group (Tables 2 and 3).

Within each group comparisons showed signiﬁcant improvement only in the mCIMT group at shoulder external rotation, shoulder, elbow, and wrist extension angles at three months after treatment (Tables 2-4). All shoulder ROMs, forearm supination, and elbow and wristﬂexion ROMs increased in both groups compared with the baseline as shown inTables 2-4.

Changes in active movement scale

As joint movement changes were compared between the two groups, signiﬁcant improvement was noticed only within the Table 2

Results of between groups and within groups of the active range of shoulder (glenohumeral) joint motions

Range of joint motion (degree) mCIMT groupn¼13 Control groupn¼26 Signiﬁcance

Shoulderﬂexion

Pretreatment 12631.8 13923.5 0.213

1 d post-treatment 14424.3^a 14621.4^a 1

1 mo post-treatment 14826^a 15119.7^a 0.952

3 mo post-treatment 14829^a 14929^a 0.580

Shoulder abduction

Pretreatment 12233.9 13924.8 0.94

1 d post-treatment 13629.6^a 14824.5^a 0.181

1 mo post-treatment 14231.5^a 14827.1^b 0.396

3 mo post-treatment 14533.9^a 14826.7^b 0.786

Shoulder Internal rotation (Arm in adduction with the elbowﬂexed to 90)

Pretreatment 5317 5320 0.928

1 d post-treatment 6816^a 6017^a 0.290

3 mo post-treatment 838.5^a 7116^a 0.025

Shoulder Internal rotation (Arm abducted to 90, elbowﬂexed to 90)

1 d post-treatment 6525 7314^b 0.483

1 mo post-treatment 6826^b 7414^b 0.807

3 mo post-treatment 7322^b 7414^b 0.735

Shoulder External rotation (arm in adduction with the elbowﬂexed to 90)

1 d post-treatment 5519^a 4325^a 0.130

1 mo post-treatment 5821^a 4526^b 0.141

3 mo post-treatment 6121^a 4528^b 0.065

Shoulder External rotation (arm abducted to 90, elbowﬂexed to 90)

1 d post-treatment 7314^b 6717^b 0.288

3 mo post-treatment 7413^b 6819 0.401

Pvalues were for within-group changes from the baseline.

Bold values indicate statistical signiﬁcance<0.05.

aP<.01.

bP<.05.

Table 3

Results of between groups and within groups of the active range of elbow joint motions

Range of joint motion (degree)

mCIMT group n¼13 meanstandard deviation

Control group n¼26 meanstandard deviation

Signiﬁcance

Elbowﬂexion

1 d post-treatment 1306.6â 1237.9 0.011 1 mo post-treatment 1307.2â 1239.5 0.022 3 mo post-treatment 1295.9â 1229.3 0.010 Elbow extension limitation

Pretreatment 1014 89.7 0.826

1 d post-treatment 55.7^b 57.4^b 0.446

1 mo post-treatment 55^b 69.2 0.871

3 mo post-treatment 35 79.8 0.397

Forearm pronation

1 d post-treatment 866.3 8116 0.380

1 mo post-treatment 875.9 866.1^b 0.612 3 mo post-treatment 885.5 866.1^b 0.489 Forearm supination

1 d post-treatment 5717^a 4725^b 0.267

3 mo post-treatment 7113^a 5421^a 0.019 Pvalues were for within-group changes from the baseline.

aP<.01.

bP<.05.

Table 4

Results of between groups and within groups of the active range of wrist joint motions

Range of joint motion (degree)

mCIMT group n¼13 meanstandard deviation

Control group n¼26 meanstandard deviation

Signiﬁcance

Wristﬂexion

1 d post-treatment 769.8^a 787.8 0.687

1 mo post-treatment 808.3^a 816.7^b 0.899 3 mo post-treatment 827.2^b 816.9^a 0.858 Wrist extension

1 d post-treatment 5917^a 6619 0.108

1 mo post-treatment 6519^a 6912 0.666

3 mo post-treatment 6919^a 7112 0.861

Pvalues were for within-group changes from the baseline.

aP<.01.

b P<.05.

(5)

mCIMT group in supination movement. This improvement has initiated at post-treatment day 1 and was maintained during the other controls (Table 5). A signiﬁcant improvement was observed in most of the measured variables of the mCIMT group as their pretreatment and 3 months post-treatment mean values were compared, except for the active shoulderﬂexion, external rotation, and elbow extension movement.

Changes in box and blocks test

Although signiﬁcant increase was observed in both groups in all controls compared with pretreatment, transferred block numbers increased signiﬁcantly only in the mCIMT group. The increase was observed at 1 month control and continued until 3 months (Fig. 2).

Changes in hand grip strength

As changes in hand grip strength have been compared between the groups, grip strength increased signiﬁcantly only in the mCIMT group. The gain in strength started to increase immediately after the intervention and continued along 1st and 3rd months (Table 6).

Discussion

This study demonstrates that mCIMT provides additional benefit beyond conventional rehabilitation in shoulder internal rotation, elbowflexion, and forearm supination ROMs, supination function, and in the ability to use the affected upper extremity. At the end of the study, in the mCIMT group, elbowflexion and forearm supination active ROMs increased by 12.1% (2.5% in the control group) and 97.2% (31.7% in the control group), respectively.

It was thought that knowing deﬁciency at grip strength increased participants' motivation. Signiﬁcant increase in grip strength occurred only in the mCIMT group, beginning from the end of the treatment and kept on: 11% in the control group and 87.5% in the mCIMT group compared with the baseline.

Most children with PBPP will achieve full functional recovery;

the rest of the patients will need specialist multidisciplinary team approach for residual deﬁcits.²⁹Early physiotherapy aims to pre- vent contractures, bone deformities, and compensatory movement patterns, maintain adequate joint ROM and function, and improve muscle strength and joint suppleness. The most desired joint motion change in upper and middle trunkeinvolved PBPP are shoulder abduction and external rotation, elbowﬂexion, and wrist extension. At older age, the aim of rehabilitation is to improve bimanual function, school and work participation, and independence with daily living activities.

Although strong evidence for CIMT is currently limited to adults after stroke and children with hemiplegic CP, this technique may have a role in PBPP by minimizing neglect as well as improving quality and symmetry of movements in the involved upper extremity. CIMT is based on“learned non-use”that underuse of the affected arm leads to a progressive suppression of movements of the affected arm.^17,30Mechanism of action is neuroplasticity, based on the fact that neuronal circuits can be activated to relieve damaged neurons by several injuries creating new functioning synapses. The efﬁcacy of this type of rehabilitation and plastic brain changes have been demonstrated in several studies also with transcranial magnetic stimulation and functional magnetic resonance imaging.^31,32

In a two-year-old child with PBPP, application of CIMT was found to improve function.²¹CIMT after botulinum toxin injections led to improved scores on Mallet and Gilbert shoulder assessments in two children beyond age 6 and 7 years with Erb-Duchenne palsy.²²Electrical stimulation and CIMT provided improvement in a child aged 2 years with global PBPP.²⁴ However, in two older children aged 12 years, observation-based functional gains were modest.²⁰The results of our study are consistent with these case reports that mCIMT principles had a potential to promote functional gains for children with PBPP. On the other hand, it was found that improvements recorded as a result of mCIMT might not be considered substantial.

At 4 years old, children could perform bimanual activities that improve with age.³³It was observed that standard neurological examination could be applied and that children achieve postural stability by the age of 4 years.³⁴In the light of this information, children of age 4 to 10 years were admissible for eligibility.

Table 5

Between-group variation of the supination movement according to Active Movement Scale Active movement scale Control groupn¼26 median

(minimum-maximum)

mCIMT groupn¼13 median (minimum-maximum)

Signiﬁcance

Forearm supination <0.05

Pretreatment 3 (2-7) 2 (2-7)

1 d post-treatment 3 (2-7) 3 (2-7)

1 mo post-treatment 3 (2-7) 3 (3-7)

3 mo post-treatment 3 (2-7) 7 (3-7)

Modified CIMT group

Control group

0 5 10 15 20 25 30 35 40 45 50

Pretreatment 1 day posttreatment

30 day posttreatment

90 day posttreatment

Box block number/60 second

Fig. 2.Comparison of Box and Blocks Test results between the two groups.

Table 6

Results of between groups and within groups of the hand grip strength Hand grip strength (kilogram-force∕square centimeter)

mCIMT group n¼13

Control group n¼26

Signiﬁcance

Pretreatment 0.0480.04 0.0360.05 0.435

1 d post-treatment 0.0660.04 0.0370.05 0.043 1 mo post-treatment 0.0730.04 0.0400.06 0.021 3 mo post-treatment 0.0900.06 0.0400.06 0.009

Pvalue .022 .676

(6)

With CIMT an increase in the child's communication, social re- lations, entertainment in performing daily activities, and self-con- ﬁdence was observed in literature.²⁸In this study, it was observed that use of the hand in everyday life increased in the mCIMT group.

Furthermore, although some children were unaware and put aside their arm before treatment, it was noticed that their awareness of limb was increased with treatment.

There is no exact information about the duration of restriction;

it varies from 1 hour to 24 hour a day and continues for 10 days up to 2 months.^9,35 Eliasson et al applied CIMT to patients with hemiplegic CP at home with glove restriction 2 hour a day for 2 months. They reported that their target restriction time was 120 hours in total but duration has varied between 16 and 120 hours (mean¼59 hours).³⁶In some studies, effective orthosis wearing time was stated subjectively; in this study, the restriction time was set exactly by children's hospitalization. During the study, children in the mCIMT group wore orthosis 84 hours for 14 days.

Methods for restriction are closing the opening of sleeve and forearm orthosis in case reports in PBPP.^20-24In the literature, it was stated that restriction method in CIMT was not an important factor.

It was, however, declared that casting was the most difﬁcult restraint technique as it might irritate skin and increase risk of fall, together with reducing stabilization and leading the child to get frustrated during play.⁹ Neither any falls nor contractures of el- bows, wrists, or ﬁngers due to restraining with orthosis were observed during this study.

After treatment, no decrease at joint ROMs was observed and usage of unaffected limb or participation to bimanual games was not declined. Only one mother reported that her child's hand- writing with unaffected limb worsened, yet the issue ameliorated at the end of study. Twelve children from the mCIMT group were adapted to orthosis by the 2nd day of treatment. One child with 54 months of age started getting bored from orthosis use at the 2nd day; continuity of orthosis use was provided by verbal feedback.

Mothers and children stated that orthosis use is a grinding work and it requires effort. Hospitalization of children in a group of 2 or 3 people increased their adaptation to treatment and their concen- tration during the study.

Better results with more intensive motor therapy were not reported. Charles et al applied CIMT 6 hours per day throughout 2 or 3 weeks, whereas Eliasson et al applied it 2 hours per day for 2 months.^12,34Some argue that periods of intense structured practice may be important than the duration of restraint wear.¹² The treatment protocol was applied patient-based by some physicians and group-based by others, as it was the case in the present study.10,18,37,38

Studies concerning stroke patients demonstrated more improvement in the CIMT group compared with control group.^6,39 Some reported that CIMT did not provide additional benefit.⁴⁰ Brogardh et al applied CIMT to adult poststroke patients and they noticed significant increase in hand function at the 3rd-month follow-up. In contrast to ourfindings, they reported that there was no difference between the groups at 12th month and restraining of the unaffected limb was not providing additional benefit to upper extremity function.⁴¹

mCIMT was applied at different environments such as home, kindergarten, daily camp, and clinic. Eliasson et al emphasized that CIMT must be applied at natural environment as home or kindergarten, whereas Gordon et al imported that it must be applied at the clinic in order not to affect family life. In this study, the combination of summer time camp model of Eliasson et al and group model of Gordon et al was administered.^11,36It was thought that via this method, children and relatives would take treatment seriously, adapt well to the process, and increase their social interaction and courage. Applying the treatment at home would particularly be

challenging, as some children might not follow their parents' instructions.

In previous studies, occupational therapists, physiotherapists, mothers, caregivers, or teachers applied the exercise. In this study, a specialist pediatric physiotherapist applied the exercise therapy and she encouraged children by verbal feedback to join exercises.

The improvement in upper extremity function in both groups may be due to children's high motivation and competition between them to use the affected limb in daily activities. Similar to the studies in children with CP, they became more cheerful by using their affected hands and joined exercises with more qualiﬁed movement ability.^9-11It was difﬁcult to determine whether that was due to central changes, to improved endurance, improved joint ROMs, or due to combination of these three factors. This functional improvement may result from the treatment of developmental disregard or from learning repeated outcome tests in progressive controls.

Factors such as age, severity of sensorimotor impairment, cognition, behavior, side of hemiparesis, presence of additional problems, site of lesion, and symmetry at bimanual movements all have potential to impact on CIMT outcomes.⁵ It is thought that irrespective of environment where CIMT is applied, education and cognitive levels of parents also affect the success of treatment.

Which patient group will beneﬁt from CIMT the most and inclusion criteria of this treatment are not yet known.

In this present study, the effects of mCIMT to the affected limb was assessed; the changes occurred in bimanual activities were not assessed. So new studies assessing bimanual effects of mCIMT in patients with PBPP should be conducted.

Strengths and limitations

Limitations of this study were the relatively small sample size, unequal numbers of children in the study groups, and short follow- up period. There were limitations in the ability to estimate the sample size because there were no available studies for similar ages in PBPP. Because of absence of sensitive tests that could show shoulder functional change after rehabilitation, minimal changes could not be shown statistically. Also, we did not examine if the healthy hand was the dominant side; so we did not know if this situation affected grip strength and dexterity improvements.

On the other hand, being conducted as a randomized controlled trial and application of mCIMT via hospitalization were strengths of this study. The ones applying the treatment were different from those who assessed the treatment, contributing to objective results.

Conclusions

Results of the present study suggest that mCIMT has a potential to promote functional gains for children with PBPP; this approach should be widely applied within routine clinical practice.

mCIMT during 2 weeks in PBPP seems to be an effective treatment method by providing increase in affected extremity joint active ROMs especially forearm supination and elbow ﬂexion, forearm supination function, gross motor skills, and hand grip strength. mCIMT was also considered to be feasible by the parents and no adverse effects were found. Consequently, it was thought that application of mCIMT in patients with PBPP may be beneﬁcial.

To determine parameters for training and to elucidate which components of mCIMT are the most effective, toﬁnd out the most appropriate age for this modality, to observe long-term results of mCIMT, and to analyze its reproducibility, other randomized controlled studies with larger sample size are needed. To investi- gate peripheral effects, to obtain electrophysiologicﬁndings, and to visualize changes at spinal and cortical presentation, studies with

(7)

transcranial magnetic stimulation and functional magnetic resonance imaging are needed.

Acknowledgments

The authors thank Yas¸ar Tatar in Marmara University Prosthetics and Orthotics Centre for designing and making custom-made static upper extremity orthoses for patients.

References

1. Gonik B. Neonatal brachial plexus palsy: antecedent obstetrical factors. In:

Chung KC, Yang LJ-S, McGillicuddy JE, eds.Practical Management of Pediatric and Adult Brachial Plexus Palsies. New York, NY: Elservier Saunders; 2012:46e 53.

2. Foad SL, Mehlman CT, Ying J. The epidemiology of neonatal brachial plexus palsy in the United States.J Bone Joint Surg Am. 2008;90(6):1258e1264.

3. Ho ES, Curtis CG, Clarke HM. Pediatric evaluation of disability inventory: its application to children with obstetric brachial plexus palsy.J Hand Surg Am.

2006;31(2):197e202.

4. Strömbeck C, Remahl S, Krumlinde-Sundholm L, Sejersen T. Long-term follow- up of children with obstetric brachial plexus palsy II: neurophysiological as- pects.Dev Med Child Neurol. 2007;49:204e209.

5. Dodds SD, Wolfe SW. Perinatal brachial plexus palsy. Curr Opin Pediatr.

2000;12:40e47.

6. Yadav RK, Sharma R, Borah D, KotharıSY. Efﬁcacy of modiﬁed constraint movement therapy in the treatment of hemiparetic upper limb in stroke patients: a randomized controlled trial.J Clin Diagn Res. 2016;10(11):1e5.

7. Boyd RN, Morris ME, Graham HK. Management of upper limb dysfunction in children with cerebral palsy: a systematic review.Eur J Neurol. 2001;8(5):150e 166.

8. Sterr A, Elbert T, Berthold I, Kölbel S, Rockstroh B, Taub E. Longer versus shorter daily constraint-induced movement therapy of chronic hemiparesis: an exploratory study.Arch Phys Med Rehabil. 2002;83:1374e1377.

9. Brady K, Garcia T. Constraint-induced movement therapy (CIMT): pediatric applications.Dev Disabil Res Rev. 2009;15:102e111.

10. Naylor CE, Bower E. Modiﬁed constraint induced movement therapy for young children with hemiplegic cerebral palsy: a pilot study.Dev Med Child Neurol.

2005;47:365e369.

11. Gordon AM, Charles J, Wolf SL. Methods of constraint-induced movement therapy for children with hemiplegic cerebral palsy: development of a child- friendly intervention for improving upper-extremity function.Arch Phys Med Rehabil. 2005;86(4):837e844.

12. Charles J, Wolf SL, Schneider JA, Gordon A. Efﬁcacy of a child-friendly form of constraint induced movement therapy in hemiplegic cerebral palsy: a randomized control trial.Dev Med Child Neurol. 2006;48:635e642.

13. Gordon AM, Charles J, Wolf SL. Efﬁcacy of constraint-induced movement therapy on involved upper-extremity use in children with hemiplegic cerebral palsy in not age dependent.Pediatrics. 2006;117:363e373.

14. Shepherd RB. Brachial plexus injury. In: Campbell SK, ed.Decision-Making in Neurologic Physical Therapy. Philadelphia, PA: Churchill Livingstone; 1999:

235e259.

15. Korak KJ, Tam SL, Gordon T, Frey M, Aszmann OC. Changes in spinal cord ar- chitecture after brachial plexus injury in the newborn.Brain. 2004;127:1488e 1497.

16. Colon AJ, Vredeveld JW, Blaauw G. Motor evoked potentials after transcranial magnetic stimulation support hypothesis of coexisting central mechanism in obstetric brachial palsy.J Clin Neurophysiol. 2007;24:48e51.

17. Simon NG, Franz CK, Gupta N, Alden T, Kliot M. Central adaptation following brachial plexus injury.World Neurosurg. 2016;85:325e332.

18. Komar A, Ashley K, Hanna K, et al. Retrospective analysis of an ongoing group- based modiﬁed constraint-induced movement therapy program for children with acquired brain injury.Phys Occup Ther Pediatr. 2016;36(2):186e203.

19. Taub E, Grifﬁn A, Uswatte G, Gammons K, Nick J, Law CR. Treatment of congenital hemiparesis with pediatric constraint-induced movement therapy.

J Child Neurol. 2011;26(9):1163e1173.

20.Buesch FE, Schlaepfer B, de Bruin ED, Wohlrab G, Ammann-Reiffer C, Meyer- Heim A. Constraint-induced movement therapy for children with obstetric brachial plexus palsy: two single-case series.Int J Rehabil Res. 2010;33(2):187e 192.

21.Vaz DV, Mancini MC, do Amaral MF, de Brito Brandao M, de Franca Drummond A, Fonseca ST. Clinical changes during an intervention based on constraint induced movement therapy principles on use of the affected arm of a child with obstetric brachial plexus injury: a case report.Occup Ther Int.

2010;17(4):159e167.

22.Santamato A, Panza F, Ranieri M, Fiore P. Effect of botulinum toxin type A and modiﬁed constraint-induced movement therapy on motor function of upper limb in children with obstetrical brachial plexus palsy. Childs Nerv Syst.

2011;27(12):2187e2192.

23.Brown SH, Napier R, Nelson VS, Yang LJ. Home-based movement therapy in neonatal brachial plexus palsy: a case study.J Hand Ther. 2015;28(3):

307e312.

24.Berggren J, Baker LL. Therapeutic application of electrical stimulation and constraint induced movement therapy in perinatal brachial plexus injury: a case report.J Hand Ther. 2015;28(2):217e220. quiz 221.

25.Sears ED, Chung KC. Validity and responsiveness of the Jebsen-Taylor hand function test.J Hand Surg Am. 2010;35(1):30e37.

26.Curtis C, Stephens D, Clarke HM, Andrews D. The active movement scale: an evaluative tool for infants with obstetrical brachial plexus palsy.J Hand Surg Am. 2002;27(3):470e478.

27.Connell LA, Tyson SF. Clinical reality of measuring upper limb ability in neurological conditions: a systemic review.Arch Phys Med Rehabil. 2012;93:

221e228.

28.Nascimento LR, Gloria AE, Habib ES. Effects of constraint-induced movement therapy as a rehabilitation strategy for the affected upper limb of children with hemiparesis: systemic review of the literature.Rev Bras Fisioter. 2009;13(2):

97e102.

29.Smith K, Patel V. Congenital brachial plexus palsy.Paediatr Child Health. 2016:

1e5.

30.Taub E, Uswatte G, Mark VW, Morris DM. The learned nonuse phenomenon:

implications for rehabilitation.Eura Medicophys. 2006;42:241e256.

31.Malcolm MP, Triggs WJ, Light KE, et al. Repetitive transcranial magnetic stimulation as an adjunct to constraint-induced therapy: an exploratory randomized controlled trial.Am J Phys Med Rehabil. 2007;86:707e715.

32.Wu CY, Hsieh YW, Lin KC, et al. Brain reorganization after bilateral arm training and distributed constraint-induced therapy in stroke patients: a preliminary functional magnetic resonance imaging study.Chang Gung Med J. 2010;33:

628e638.

33.Otte E, Van Mier HI. Bimanuel interference in children performing a dual motor task.Hum Mov Sci. 2006;25:678e693.

34.Alexander MA, Molnar GE. History and examination. In: Alexander MA, Matthews DJ, eds.Pediatric Rehabilitation. New York: Demos Medical Pub- lishing; 2010:7e8.

35.Hoare BJ, Wasiak J, Imms C, Carey L. Constraint-induced movement therapy in the treatment of the upper limb in children with hemiplejic cerebral palsy.

Cochrane Database Syst Rev. 2007;18(2):CD004149.

36.Eliasson AC, Krumlinde-Sundholm L, Shaw K, et al. Effects of constraint- induced movement therapy in young children with hemiplegic cerebral palsy: an adapted model.Dev Med Child Neurol. 2005;47:266e275.

37.DeLuca SC, Echols K, Ramey SL, et al. Pediatric constraint-induced movement therapy for a young child with cerebral palsy: two episodes of care.Phys Ther.

2003;83:1003e1013.

38.Eliasson AC, Bonnier B, Krumlinde-Sundholm L. Clinical experience of constraint induced movement therapy in adolescents with hemiplegic cerebral palsy-a day camp model.Dev Med Child Neurol. 2003;45:357e 360.

39.Kwakkel G, Veerbeek JM, van Wegen EE, Wolf SL. Constraint induced movement therapy after stroke.Lancet Neurol. 2015;14(2):224e234.

40.Etoom M, Hawamdeh M, Hawamdeh Z, et al. Constraint-induced movement therapy as a rehabilitation intervention for upper extremity for stroke patients: systemic review and meta-analysis. Int J Rehabil Res.

2016;39(3):197e210.

41.Brogardh C, Lexell J. A 1- year follow up after shortened constraint induced movement therapy with and without mitt poststroke.Arch Phys Med Rehabil.

2010;91:460e464.