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Constraint-induced movement therapy for children with obstetricbrachial plexus palsy

Buesch, F E; Schlaepfer, B; de Bruin, E D; Wohlrab, G; Ammann-Reiffer, C;Meyer-Heim, A

Buesch, F E; Schlaepfer, B; de Bruin, E D; Wohlrab, G; Ammann-Reiffer, C; Meyer-Heim, A (2010).Constraint-induced movement therapy for children with obstetric brachial plexus palsy. International Journal ofRehabilitation Research. Internationale Zeitschrift für Rehabilitationsforschung. Revue Internationale deRecherches de Réadaptation, 33(2):187-192.Postprint available at:http://www.zora.uzh.ch

Posted at the Zurich Open Repository and Archive, University of Zurich.http://www.zora.uzh.ch

Originally published at:International Journal of Rehabilitation Research. Internationale Zeitschrift für Rehabilitationsforschung. RevueInternationale de Recherches de Réadaptation 2010, 33(2):187-192.

Buesch, F E; Schlaepfer, B; de Bruin, E D; Wohlrab, G; Ammann-Reiffer, C; Meyer-Heim, A (2010).Constraint-induced movement therapy for children with obstetric brachial plexus palsy. International Journal ofRehabilitation Research. Internationale Zeitschrift für Rehabilitationsforschung. Revue Internationale deRecherches de Réadaptation, 33(2):187-192.Postprint available at:http://www.zora.uzh.ch

Posted at the Zurich Open Repository and Archive, University of Zurich.http://www.zora.uzh.ch

Originally published at:International Journal of Rehabilitation Research. Internationale Zeitschrift für Rehabilitationsforschung. RevueInternationale de Recherches de Réadaptation 2010, 33(2):187-192.

CIMT for children with brachial plexus palsy 1

Constraint induced movement therapy (CIMT) for children with obstetric brachial

plexus palsy – two single case series

Running head: CIMT for children with brachial plexus palsy

Francisca Eugster Buescha,b, Barbara Schlaepferb, Eling D. de Bruinb, Gabriela Wohlraba,

Corinne Ammann-Reiffera, Andreas Meyer-Heima.

a Rehabilitation Centre Affoltern am Albis, University Children’s Hospital Zurich

b Institute of Human Movement Sciences and Sport, ETH, Zurich

Corresponding author

Andreas Meyer-Heim MD

Rehabilitation Centre, University Children’s Hospital Zurich

Mühlebergstr. 104, 8910 Affoltern am Albis, Switzerland

Andreas.Meyer-Heim@kispi.uzh.ch

Tel: +41 44 762 51 11; Fax +41 11 762 52 11

Grant: Stiftung Cerebral, Switzerland

Brief research report

Word count 3428

CIMT for children with brachial plexus palsy 2

Abstract

Objective of this pilot study was to investigate the feasibility of Constraint induced movement

therapy (CIMT) in children with obstetric brachial plexus palsy (OBPP) and receive

preliminary information about functional improvements. Two patients (12 years) with OBPP

were included for a 126 hours home-based CIMT intervention (3 and 4.5 weeks) and weekly

monitored with the Melbourne Assessment of Unilateral Upper Limb function (MA) and the

nine hole peg test (NHPT). The Assisted Hand Assessment (AHA) was performed 3 weeks

prior to the beginning of intervention, pre-, post and at 3 weeks follow-up (ABA single case

design). Tests were analyzed descriptively, visually, and where possible, using a times series

analysis with C statistic. Results revealed changes of arm function in both patients between

pre intervention and follow-up test as follows: AHA from 85 to 92% in patient A and 80 to

89% in patient B, in the MA from 87 to 90% (C = .578, z = 1.876, p < .05) in patient A and 72

to 80% (C = .827, z = 2.68, p < .005) in patient B respectively. There was no improvement in

the NHPT in both patients. Improvements due to CIMT found in this pilot study may not be

considered substantial, however, justify for further research since some findings indicate to

be related to the implementation of intervention in the “B”-phase. In order to enhance

motivation and to plan a controlled randomized clinical trial, child-friendly applications of the

CIMT concept and adequate measures to monitor compliance are being discussed.

Keywords

Obstetric brachial plexus palsy, child, constraint induced movement therapy, forced use

CIMT for children with brachial plexus palsy 3

Introduction

Obstetrical brachial plexus palsy (OBPP) is defined as flaccid pareses of an upper extremity

and is caused from stretch injury at birth to the cervical roots C5-C8 and thoracic root T1

(Evans-Jones et al., 2003, Pollack et al., 2000).

The incidence of OBPP has a wide range from 0.38 to 3 per 1000 live births and high birth

weight is known to be the most significant risk factor for shoulder dystocia and OBPP

(Pondaag et al., 2004; Zafeiriou and Psychogiou, 2008). The burden of disability of

permanent sequelae of OBPP in those patients not recovering spontaneously (5-50%)

remains high (Strömbeck et al., 2007; O Brian et al., 2006). A multidisciplinary team

approach including physical and occupational therapy is necessary to maintain passive

range of motion and supple joints as well as muscle strengthening (Dodds and Wolfe, 2000).

Some patients may benefit from recent advantages in micro-neurosurgical procedures like

early plexus reconstruction and “palliative” surgery such as muscle releases (Terzis and

Kokkalis, 2008).

In the nineteen eighties, Taub described the concept of Constraint induced movement

therapy (CIMT) (Taub 1980). This therapy aims to overcome the “learned non-use” of an

extremity in hemiplegic patients by forcing the affected arm into use, immobilizing the non-

affected arm with a splint or sling (Taub 1993, Taub 2006). Recent meta-analyses showed

evidence for CIMT to be an effective treatment option in adult stroke patients, whereas in

children with hemiplegic cerebral palsy it is still considered experimental (Bonaiuti et al.,

2007; Hoare et al., 2007).

So far, this therapeutic approach has only been used for patients with palsies due to cortical

lesions like stroke. Korak et al. (2004) proposed that not only peripheral reconstruction but

also the role of spinal cord plasticity should be included in treatment strategies of OBPP

(Korak et al., 2004). Assuming that a chronic non- or diminished use of an arm due to an

incomplete peripheral palsy induces smaller representations of motor areas and fibers of the

spinal cord and cortex, some functional gain from CIMT could be expected.

CIMT for children with brachial plexus palsy 4

To our knowledge this is the first trial investigating CIMT as a treatment option for children

with OBPP. The aim of this single case study design pilot study is to examine if CIMT is a

feasible treatment option in a community based setting and leads to an improvement of arm

and hand function in children with chronic impairment of OBPP.

Patients and Methods

Participants

Two patients diagnosed postnatally with left upper (C5-C7) OBPP were included in this

single-case series (both male, age 12 years, attending regular school). Patient A still had one

weekly session of physiotherapy and occupational therapy. The therapy frequency of both

therapies was left unchanged for ethical reasons and CIMT was applied additionally.

Functional impairment in the activity of daily living described by the patient involved handling

a knife for cutting meat, using a computer keyboard and longer time span needed for self

care. Muscular hypotrophy was present in the shoulder girdle, upper- and forearm with

reduced strength in the manual muscle testing: Biceps Brachii M4, Supraspinatus M3+,

medial Deltoid M3+ and grip strength M4+ (MMT: M5 normal strength to M0 no muscle

contraction) (Kendall et al. 1993). The passive range of motion (pROM) measures were: arm

abduction/adduction 130-0-10°, ante/retroversion 150-0-30°, internal/external rotation when

arm adducted 95-10-0°elbow flexion/extension 135-15-0°, supination/pronation 60-0-30.

Epicritic and proprioceptive sensibility was intact.

Patient B had undergone a tendon transfer (Supraspinatus from Levator scapulae) at the age

of 10 years. In this case regular therapies had been discontinued several months prior to

study onset. The patient described no considerable limitations in the activities of daily living

except for the apparatus gymnastics. There was a hypotrophic muscular relief of the

shoulder girdle, forearm and hypothenar. Muscle strength for elbow flexion and extension

was reduced in the manual muscle testing: Biceps Brachii M4+, Supraspinatus M4 medial

CIMT for children with brachial plexus palsy 5

Deltoid M4 and grip strength M4+ ,. Passive, active range of motion (pROM/aROM)

respectively were: arm abduction 90° and 50° resp., elbow flexion/extension 130/20/0° and

120/20/0° resp., internal/external rotation when arm abducted 70-0-70° and 60-0-5° resp,

supination/pronation active and passive 70-0-30°. Finger movements were unrestricted.

Epicritic and proprioceptive sensibility was intact.

No micro-reconstruction has been done in either of the two patients.

For inclusion in the study patients had to be able to lift their arm against gravity (M3) and to

achieve a minimal grasp. The CIMT concept had to be understood by both the children and

their parents. Ethical approval was obtained from the local ethic’s committee and written

consent had been given by the parents and the participants.

Intervention

CIMT using a forearm splint on the non-affected arm (fig 1) was conducted during 6

hours/daily for 3 weeks (Patient A) and 4 hours/daily for 4.5 weeks (Patient B) respectively.

Total wearing time of the splints was 126 hours in each case. As the intervention was carried

out in the community setting, both patients were given a checklist with one-handed activities

of daily living. The ongoing therapy sessions of patient A had been left unchanged.

Procedure and measures

The Melbourne Assessment of Unilateral Upper Limb Function (MA) (Johnson et al., 1994),

the Assisting Hand Assessment (AHA) (Krumlinde-Sundholm et al., 2007) and the Nine hole

Peg Test (NHPT) (Mathiowetz et al., 1985) were used as outcome measures and timed as

shown in Fig. 2.

The MA is a unilateral hand function test validated for children with neurological deficits aged

5-15 years. The test battery consists of 16 items and evaluates the quality of hand function

by a scoring system (0-4 points) including a video analysis of strictly defined camera

positions. Randall (2001) found a very high internal consistency of test items (∝=0.96) and

high intrarater reliability (0.97), as well as high interrater reliability (0.95) for total test scores

CIMT for children with brachial plexus palsy 6

in children with CP (Randall et al., 2001). These findings have been confirmed by Cusick

even for novice raters (Cusik et al., 2004). The MA has been used in several intervention

studies, even though to our knowledge of literature sensitivity to change has not been

studied systematically yet (Klingels et al. 2008).

The AHA measures the spontaneous bimanual use of hands during activities of daily living

and is validated for children aged 2-12 years with OBPP, CP and traumatic brain injuries.

The performance of 22 items is filmed and scored with 1 to 4 points. The AHA has been

found to be sensitive to change and a reliable instrument (ICC: 0.98 two-rater design, 0.97

20-rater design, 0.99 intrarater) to assess hand function in children with CP and OBPP

(Krumlinde-Sundholm et al. 2007).

The NHPT provides accurate information about finger dexterity and hand-eye coordination in

measuring the time to insert and remove nine pegs in the dominant and the non-dominant

hand. Normative data studies have proven the NHPT to be a reliable (interrater rs>0.99, test-

retest (rs=0.81 and 0.79) and valid tool for dexterity assessment of children and sensitive to

the developmental age, whereas sensitivity to change has only been shown in adult patients

(Smith et al. 2000, Poole et al. 2005).

The complete study period for both individuals consisted of an ABA single case study design

(Payton, 1994): a BASELINE period of 3 weeks, an INTERVENTION period of3 and 4.5

weeks respectively (consisting of 126 h constraint) and a POST-CIMT period of 3 weeks.

As suggested in Taub’s protocol (1993), a diary including a feedback form had to be filled out

by patients in order to support and monitor the daily CIMT training. Neurological and clinical

assessments were performed by the consultant and the therapists. The MA, AHA and NHPT

were conducted by a especially trained rater (Bachelor of Human Movement Science,

second co-author) not involved in the clinical and therapeutical follow ups of the patients.

Data Analysis

CIMT for children with brachial plexus palsy 7

Data were analyzed through visual inspection of the graphed measurements of the

dependent variables. In the visual interpretation the stability of the baseline, the mean

(average) shift across phases, the change in level between phases, the variability of the

measures, and the change in slope across phases were assessed. Visual inspection of the

data was, where possible, complemented with a times series analysis with the C statistic

(Arnau & Bono, 1998) in order to identify any systematic departure from random variation in

the set of data.

Results

Melbourne Assessment (MA)

Patient A started with a mean percentage score of 87 ± 1.3% (mean baseline, n = 4), then

reached a mean of 89 ± 1% in the CIMT intervention period (n = 4) and showed 90 ± 1% in

the follow up phase (n = 4). There was a reversal in slope observable between the baseline

data slope (first A phase) and the intervention phase (B). The follow-up phase (second A

phase) shows light continued improvement in the MA score. Changes were seen in the

items: pronation/supination, reach to the opposite shoulder and hand to mouth and down

movement. Analysis with the C statistic revealed the following values: Baseline: C = .368, z =

1.008, p > .05; Treatment: C = 0.333, z = 0.912, p > .05; Follow-up: C = -0.09, z = -0.248, p

> .05; Baseline + Treatment: C = 0.54, z = 1.75, p < .05; Baseline + Follow-up: C = .686, z =

2.223, p < .05; Treatment + Follow-up: C = .578, z = 1.876, p < .05. Within the separate

phases no trend was evident. The addition of the treatment created change greater than

would be expected from random variation and this change continued in the follow-up phase.

Patient B started with a mean percentage score of 72 ± 2.8% in the baseline; he then

continuously improved to a mean of 74 ± 4.3% in the intervention phase. In the follow-up

phase he reached a mean score of 80 ± 1.4%. There was a reversal in slope observable

between the baseline data (first A phase) and the intervention phase (B). The follow-up

phase shows a light decrease in the MA score, however, with a higher level of performance

CIMT for children with brachial plexus palsy 8

compared to the baseline phase. Improvements were observed in the items: reaching

forward, reaching forward to an elevated position, drawing grasp and pronation/supination.

Analysis with the C statistic revealed the following values: Baseline: C = -0.076, z = -0.21, p

> .05; Treatment: C = 0.689, z = 1.888, p < .05; Follow-up: C = -0.166, z = -0.456, p > .05;

Baseline + Treatment: C = 0.514, z = 1.667, p < .05; Baseline + Follow-up: C = .505, z =

1.638, p = .05; Treatment + Follow-up: C = .827, z = 2.68, p < .005. Within the separate

phases no trend was evident in the baseline and follow-up phase. The treatment phase

shows a significant change toward improvement. The addition of the treatment created

change greater than would be expected from random variation and this change discontinued

in the follow-up phase.

Assisted Hand Assessment (AHA)

Patient A showed in the baseline test a mean percentile rank of 83% and a continuous slope

pattern across all three phases. During intervention a change from 85 % to 88 % was

observed; the highest percentile rank of 92% was found in the follow up assessment.

Improvements were mainly due to higher scores in the items: initiates use, chooses assisting

hand, moves forearm and moves upper arm.

After a stable baseline period with a percentile rank of 80% Patient B showed a change in

slope after intervention was initiated with improvement during the CIMT intervention period

up to 88%. In the follow-up phase patient B maintained the score (89%) in the follow up test

on a higher level as compared to baseline. Improvements were found in the items: reaches,

moves upper arm, grasps and manipulates.

Nine hole peg test (NHPT)

Patient A showed a larger distribution of results in the affected left arm (range 18–23 sec),

which was slower as the non-affected right arm (range 15-17.8 sec). The mean of Baseline

was 20.8 ± 1.7 sec for the affected arm, 21.3 ± 1.6 sec in the intervention phase, and 20.9 ±

2.1 sec in the three week follow-up phase. Analysis with the C statistic revealed the following

CIMT for children with brachial plexus palsy 9

values for the affected hand: Baseline: C = 0.199, z = 0.547, p > .05; Treatment: C = 0.487,

z = 1.335, p > .05; Follow-up: C = -0.348, z = -0.953, p > .05; Baseline + Treatment: C =

0.356, z = 1.155, p > .05; Baseline + Follow-up: C = -0.333, z = -1.079, p > .05; Treatment +

Follow-up: C = -0.013, z = -0.042, p > .05. Within the separate phases no trend was evident

in either phase. Initiation of the treatment caused a reversal in slope with worse scores

during this B-phase. Withdrawal of the intervention resulted in a similar slope pattern as pre-

intervention. No significant changes were revealed with the C statistic analysis between

baseline and the follow-up phase in the non-affected hand (data not shown).

Also in patient B the affected arm has been found to have a larger distribution of results

(baseline mean 25 ± 3.5 sec; intervention 25.4 ± 3.4 sec; follow-up 27.4 ± .7 sec; range 21-

29 sec) and has been found to be slower than the non-affected arm (baseline mean 18.1 ± .8

sec; intervention 17.9 ± .8 sec; follow-up 17.7 ± 2 sec; range 16-19 sec).

Analysis with the C statistic revealed the following values for the affected hand: Baseline: C

= 0.681, z = 1.86721, p < .05; Treatment: C = 0.632, z = 1.731, p < .05; Follow-up: C = -

0.127, z = -0.361, p > .05; Baseline + Treatment: C = 0.659, z = 2.137, p < .05; Baseline +

Follow-up: C = .204, z = 0.632, p > .05; Treatment + Follow-up: C = .673, z = 2.085, p < .05.

Within the separate phases a reverse trend was evident in the baseline and intervention.

There was no improvement in the affected hand after the intervention and follow up period

respectively. Initiation of the treatment caused a reversal in slope with worse scores during

this B-phase. Withdrawal of the intervention resulted in a similar slope pattern as pre-

intervention. No significant changes were revealed with the C statistic Analysis between

baseline and the follow-up phase in the non-affected hand (data not shown).

Fig. 2 a-f.

Diaries and feedback from parents, patients and therapists

Taken from the comments in the diaries it has been revealed that the inclusion of CIMT in the

daily routine is a very difficult task. No clear inference could be drawn from the diaries in

regards to the compliance of wearing the splints but both patients indicated that the daily

CIMT for children with brachial plexus palsy 10

wearing time was occasionally shorter than asked by the study protocol. Parents of patient A

reported considerable improvements in hand dexterity and in the activity of daily living after

the intervention period. No observations indicating change have been made by the parents of

patient B.

The neurological examination and therapist’s observation after intervention of patient A

revealed improvements in the symmetry and dexterity of the left arm and hand function,

increase of active and passive ROM of 10° in the external rotation and hand supination, but

a deterioration of 5° in passive elbow extension. A strengthening of hand (especially thenar)

muscles and an improved hand position (less ulnar abduction) but more tension in the

shoulder girdle was observed. However, the initial MMT values did not change during the

CIMT period. In patient B no substantial changes were revealed by the therapist and in the

neurological assessment.

Discussion

The aim of this single case study design pilot study was to examine whether CIMT is a

feasible treatment option in a community based setting that leads to an improvement of arm

and hand function in children with chronic impairment of OBPP. The feedbacks of the

participants yielded that consistent wearing of the splint had been a very demanding and

exhausting task, both for the patients and their parents. In our opinion, therefore, the current

CIMT concept is only suited for highly motivated patients. This was also observed in previous

trials, in which the CIMT application had to be modified especially for children (Wallen et al.,

2008). Other means to augment motivation like video diaries or virtual reality based

telerehabilitation techniques could be considered for inclusion in home based study designs.

A close cooperation with the schools would eventually help to prevent that the CIMT has to

be carried out in leisure time only, which may further jeopardize the motivation to take part in

a CIMT program.

CIMT for children with brachial plexus palsy 11

The results of this pilot study suggest a certain degree of improvements for both patients, as

visually observed in the dependent variables AHA and MA. The two subjects in this study

showed similar patterns of improvement in the MA and AHA respectively as well the slower

performance in the NHPT that seemed closely related to the initiation of the CIMT

intervention. A functional improvement observed by the parents and the participant was

reported on patient A, which was in accordance to improvements of motor functions

observed in neurological assessment and by the therapists of this patient. This patient was

even motivated to repeat a CIMT cycle on an individual basis.

Functional improvements after CIMT in a child with cerebral palsy have been found to be

associated with permanent changes in cortical reorganisation measured by fMRI (Sutcliffe

2007). There is increasing evidence that not only restoration of peripheral nerve continuity

but also plasticity of the corresponding spinal cord plays an important role in the recovery

process of the OBPP (Korak et al., 2004). From their results of preserved motor evoked

potentials in the deltoid and biceps muscle in four to seven months old children with

complete avulsion of the roots C5 and C6 Colon et al. (2007) derived an insufficient cortical

representation at that age and emphasised the role of cortical plasticity also in OBPP. We

hypothesised in this study that the learned non-use might possibly be held responsible – at

least partially - for persistent motor malfunction in OBPP patients through affection of the

cortical representation of the impaired arm.

We are aware of several limitations of this pilot study. First, a small sample size had to be

chosen because of ethical reasons as CIMT is considered to be experimental for this group

of patients. Visual data inspection alone may lead to erroneous conclusions if the data are

too dependent. We therefore tried to strengthen our conclusions by doing a C statistics

analysis of our data. A positive motivational bias is likely as patients had to agree to a

substantial study protocol. The observed effects cannot definitely be attributed to the CIMT

only, as learning effects could also play a role. Contrariwise, we cannot rule out that the high

frequency of hand assessment in our study protocol negatively influenced the motivation of

the patients, as additional expenditure of time was inevitable. The diaries can only reflect an

CIMT for children with brachial plexus palsy 12

incomplete monitoring of the effective time wearing the splints, there remains an uncertainty

that the 126 hrs have thoroughly been accomplished. Only the thenar muscle of the Patient A

has been observed to improve, however this could not be reproduced in the MMT of grip

strength. On one hand we do not expect distinct changes in muscle strength after a two-

week CIMT period which focuses on functions of daily living, on the other hand the MMT

seems not to be sensitive enough to document fine improvements after such a short period

of therapy (Bohannon 2005). A sensitive muscle testing like dynamometry should be

included into a future research protocol. The observation of clinical changes observed by

ROM has to be considered cautiously given the limited sensitivity and reliability of these

measurements in children (Escolar et al. 2001, Bialocerkowski and Galea 2006). In our

opinion, one can not differentiate, if the functional improvements occurred were due to

central changes, improved fitness or ROM; it may be a combination of all.

We conclude that our findings justify conducting a controlled and randomized clinical trial. As

there was no functional deterioration in either, the affected or the non-affected arm ethical

concerns can be met. However, conditions of CIMT must be adapted to be motivational and

feasible for this group of young patients. Further research should consist of functional

measurements including a long-term follow up after 6 and 12 months and use of goal

attainment scaling in order to measure intra-individual changes in activity and participation.

Additional functional or anatomical MRI or DTI techniques to investigate changes of spinal

and cortical representation in combination with electrophysiological measures for

investigation of peripheral effects enhanced by CIMT would be of particular interest.

Acknowledgements

We are thankful to the participants and their families for taking part in this study and to the

therapists for their support. We thank Beth Padden for carefully reviewing the manuscript.

This study was supported by a grant of the Stiftung Cerebral, Switzerland.

CIMT for children with brachial plexus palsy 13

Competing interest

none

CIMT for children with brachial plexus palsy 14

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Legends

Fig 1: Patient wearing the CIMT splint made from soft/scotch cast material on the non-

affected right arm involved in activities of daily living.

Fig 2 a-f: Graphs of the Melbourn Assessement (MA), Assisted Hand Assessment (AHA) and

Nine Hole Peg Test (NHPT) of Patient A (a,c,e) and B (b,d,f). X-axis: Intervention periods in

patient A lasted 3, in patient B 4.5 weeks, baseline and follow up were 3 weeks in both

patients. The assessments have been distributed regularly over all periods.

CIMT for children with brachial plexus palsy 19