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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...

Article in Occupational Therapy International · December 2010

DOI: 10.1002/oti.295 · Source: PubMed

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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

Daniela Virgínia Vaz¹, Marisa Cotta Mancini²*^†, Maíra Ferreira do Amaral³,

Marina de Brito Brandão³, Adriana de França Drummond² & Sérgio Teixeira da Fonseca¹

1Physical Therapy Department, Universidade Federal de Minas Gerais, Brazil

2Occupational Therapy Department, Universidade Federal de Minas Gerais, Brazil

3Graduate Program in Rehabilitation Sciences, Universidade Federal de Minas Gerais, Brazil

Abstract

The objective of this study was to test the feasibility of a treatment programme based on the elements of constraint- induced movement therapy (CIMT) to encourage use of the affected arm of a child with obstetric brachial plexus injury (OBP), as well as to document clinical changes observed with this intervention. A 2-year-old female child with Erb’s palsy had 14 weeks of daily home-based treatment with 30-minute sessions planned according to the principles of CIMT. The child was assessed every 2 weeks with the Toddler Arm Use Test. Test scores throughout the intervention period demonstrated improvements in quality of movement, amount of use and willingness to use the affected extremity. The child’s mother reported improved ability to perform bimanual activities at home. The results suggest that treatment based on CIMT principles has potential to promote functional gains for children with OBP. Experimental studies should test the effects of this kind of intervention for children with OBP. Copyright © 2010 John Wiley & Sons, Ltd.

Received 25 February 2010; Revised 21 April 2010; Accepted 22 April 2010

Keywords

obstetric brachial palsy; constraint induced movement therapy; upper extremity function; toddler; pediatric occupational therapy

*Correspondence

Marisa Cotta Mancini, Departamento de Terapia Ocupacional, Programa de Pós-Graduação em Ciências da Reabilitação, Universidade Federal de Minas Gerais, Av. Antônio Carlos, 6627, Campus Universitário – UFMG, Pampulha, Belo Horizonte – MG, Brasil. CEP 31270-010.

†Email: [email protected]

Published online 19 July 2010 in Wiley Online Library (wileyonlinelibrary.com) DOI: 10.1002/oti.295

(Heise and Gherpelli, 2006). OBP incidence is compa- rable with that of other important health conditions in infancy, such as Down syndrome and cerebral palsy (Bialocerkowiski et al., 2006). The literature points to several risk factors for OBP, including increased weight at birth, prolonged labour, perinatal asphyxia and forceps delivery. Although the relation between these factors and OBP is well established in the literature,

Introduction

Obstetric brachial plexus injury (OBP) causes partial or total paralysis of the upper limb because of trauma to the brachial plexus during delivery (Evans-Jones et al., 2003). Despite the signifi cant improvements in obstetric techniques, the incidence of this condition may reach 1.5 cases per 1,000 births in developed countries

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CIMT-Based Intervention for a Child with OBP Vaz et al.

factors related to the functional prognosis of the condition are still under investigation (Evans-Jones et al., 2003) and include injury site and severity (Strömbeck et al., 2007).

OBP has important repercussion for the child as structural defi cits that interfere with the use of the upper extremity can result in limitations to the performance of activities of daily living and restrict participation of the child at school and community (Strömbeck et al., 2007). In addition to conservative treatments, several surgical techniques are used with the aim of improving the child’s clinical status. Nevertheless, the literature regarding the functional rehabilitation of children with OBP is remarkably scarce.

Among the most promising rehabilitation treatments aimed at improving upper extremity function in individuals with neurological dysfunctions is the technique named constraint-induced movement therapy (CIMT) (Liepert et al., 2000; Boyd et al., 2001; Sterr et al., 2002). This technique consists of constraint of the non-affected extremity associated with intensive task- related training of the affected extremity. Development of CIMT was based on studies with monkeys that exhibited a behaviour called learned non-use after deaf- ferentiation of one upper limb. The animals learned not to use the affected limb because they could perform satisfactorily with the other limbs. This habitual non-use persisted so that even after recovery of the affected limb, the animals did not seem aware of the new functional possibilities (Taub et al., 1999). Experi- ments with animals demonstrated that CIMT could minimize this behaviour. Subsequently, CIMT has been shown to increase the use of the affected extremity of adults with cerebrovascular accidents (Taub et al., 1994, 1999; Kunkel et al., 1999; Miltner et al., 1999; Liepert et al., 2000; Eliasson et al., 2003) and children with hemiparesis because of cerebral palsy (Eliasson et al., 2003, 2005; Taub et al., 2004, 2007; Gordon et al., 2005;

Naylor and Bower 2005; Charles et al., 2006; Deluca et al., 2006; Cope et al., 2008; Brady and Garcia 2009;

Coker et al., 2009). The proposed mechanism of improvement after CIMT is attributed to the overcoming of learned non-use and the effects of task training on neural plasticity (Kim et al., 2004; Taub et al., 2004).

A process analogous to learned non-use, termed developmental disregard by Gordon et al. (2005), is likely to occur in infants with OBP and may contribute to the failure to develop functional use of the affected

extremity. Repeated failure after initial attempts to use the affected extremity may lead the infant to stop trying.

Developmental disregard may also prevent recovery of function that could have occurred as a result of nerve regeneration (Shepherd, 1999). Clinical observation indicates that some infants with good return of muscle function nevertheless ignore the arm and refuse to use it. Because of such observations, Shepherd suggests the treatment of children with OBP should be based on CIMT principles (Shepherd, 1999).

The objective of this study was to test the feasibility of using elements of CIMT in the treatment of a child with OBP as well as to document clinical changes on the use of the affected arm after this intervention.

Results from this case report may suggest alternative mechanisms as possible explanations for the effects underlying this intervention.

Methods

The child

AC, a female child, was diagnosed with Erb’s palsy of the right arm after a forceps delivery. She received physical therapy sessions twice a week until the age of 2 years, when she no longer demonstrated functional improvements. Although she demonstrated ability to use the affected upper extremity to reach and hold objects during treatment sessions, her mother com- plained she demonstrated little spontaneous use of the arm at home. The family was then invited to take part in a course of treatment planned according some of the principles of CIMT. According to the specifi c complaints related to limitations in activities at home and diffi culty in attending daily sessions of treatment at the outpatient clinic, a home-based intervention was chosen. In addition, the familiar physical and social environments at home could facilitate engagement in the activities and lead to better results. The University Ethics Review Board approved this case study and informed consent was obtained from the child’s mother.

The main concern expressed by the mother in the beginning of the intervention was related to performance of daily life activities. The child had diffi culties at combing, detangling her hair, donning shirts, sweat- ers or dresses without a front opening, and donning trousers without an elastic waistband. The mother’s identifi cation of the functional activities that were not performed by the child according to expectations was

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important to help guide the functional context of intervention.

Physical examination revealed that the child was capable of actively moving the shoulder, as well as wrist and fi ngers, but not throughout the complete range of motion. She did not have active elbow movement and was capable of raising the arm only by associating shoulder abduction with trunk extension and lateral fl exion. She could grasp small objects, but they would easily slip off her hands because of the defi cit in prehension strength.

Intervention protocol

According to the original model proposed by Taub (Taub et al., 1999), CIMT involves the restriction of the non-affected upper extremity for approximately 90% of waking hours, during 2–3 weeks, with intensive and repetitive training of arm and hand function everyday for 6 hours. Nevertheless, some studies conducted with children propose adapted child-friendly models with decreased dosage of daily training compensated by increased protocol duration (Eliasson et al., 2005; Gordon et al., 2005; Naylor and Bower 2005).

Daily training sessions for AC lasted 30 minutes.

Duration of sessions was decided in collaboration with the mother and took into consideration the tolerance and level of frustration of the child, and the time the mother had available in her daily routine to implement the training at home. During all sessions, movement of the non-affected arm was supposed to be restricted. By suggestion of the mother, restriction was attained by dressing the child with a jumper, closing the opening of the left sleeve and tying it on the child’s back with and elastic band. The initial frustration and irritability of child because of the restriction were soon minimized as she received verbal encouragement and rewards from her mother and learned the sessions were short in duration. The constraint was not used any other time of the day.

A schedule of three tasks, each to be practised for 10 minutes every day (summing a total session time of 30 minutes) was defi ned for each 2-week period. The tasks were chosen according to their suitability to the child’s age and interests, and focused on abilities needed for play, self-care and school activities. Mother’s indica- tions of child’s functional limitations guided the choices of activities to be used in the adapted CIMT protocol,

in each occupational area. The abilities trained included reaching, prehension and manipulation with the affected hand. Task diffi culty was increased progres- sively during weekdays and with every schedule change, by manipulations in the demands for velocity, movement amplitude and versatility. Table I lists all activities recommended in the seven schedules used during the fourteen weeks of intervention.

For example, in one of the tasks of the fourth schedule, the child was encouraged to play with her mother’s clown nose, by reaching it and taking it off. The mother received instructions to position her face right in front of her child’s right shoulder at a distance equivalent to arm length in the fi rst day and to progress to higher positions along the days of practice. As another example of progression, in one schedule, the child should reach and grasp foam rubber fi gures stuck to a wet mirror, and in the next schedule, she should remove fi gures stuck with Velcro to a vertical surface, which offered greater resistance and thus demanded greater prehension strength in order to detach the fi gures. The

Table I. Tasks scheduled for each 2-week period

Schedule number

Recommended tasks

1 Finger painting

Tear paper towels (single thickness) Make something with ‘play-dough’

2 Remove foam rubber fi gures stuck to a mirror with water

Throw party balloons to the mother Tear magazine pages

3 Remove foam rubber fi gures stuck with Velcro to a vertical surface

Without looking, fi nd candies inside a box fi lled with shredded paper

Color a drawing with crayons

4 Spread moisturizing cream on mother’s body Pick-up and eat biscuits

Reach and grasps mother’s clown nose 5 Build a tower with toy bricks

Make soap bubbles using a bubble blower Remove big puzzle pieces stuck with Velcro to a

background

6 Throw a ball to the mother

Remove medium puzzle pieces stuck with Velcro to a background

Stick foam rubber fi gures a wet mirror

7 Assemble a foam puzzle

Remove small puzzle pieces stuck with Velcro to a background

Pick-up and eat breakfast cereal

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therapist provided all materials necessary for home activities.

All schedules contained descriptions of the tasks in simple language, as well as instructions on how to increase task diffi culty as the child demonstrated

improvements in performance. At the end of each session, the mother should rate her child’s performance as very good, good or poor by marking the corresponding smiley face in the schedule (Figure 1).

Figure 1 Example of a schedule for task training SCHEDULE NUMBER 4

TASK 1:Spread moisturizing cream on mother’s body TIME: 10 minutes.

DESCRIPTION: The child should spread the cream on a doll or on her mother’s body. In the first day, the doll or body part should be positioned at the height of the child’s hips. Height should be increased every day until child’s shoulder level is reached.

SUNDAY SUNDAY

MONDAY MONDAY

TUESDAY TUESDAY

WEDNESDAY WEDNESDAY

THURSDAY THURSDAY

FRIDAY FRIDAY

SATURDAY SATURDAY

TAKS 2: eat biscuits TIME: 10 minutes.

DESCRIPTION: The child should pick-up and eat biscuits placed in a plate in front of her. Big biscuits should be offered in the first day. Size of biscuits should be decreased every day.

SUNDAY SUNDAY

MONDAY MONDAY

TUESDAY TUESDAY

WEDNESDAY WEDNESDAY

THURSDAY THURSDAY

FRIDAY FRIDAY

SATURDAY SATURDAY

TASK 3: remove clown nose TIME: 10 minutes.

DESCRIPTION: the child should try to reach and remove her mother’s clown nose. In the first day, the nose should be right in front of the child’s right shoulder at an easily reachable distance. Height and distance of the nose should be increased every day.

SUNDAY SUNDAY

MONDAY MONDAY

TUESDAY TUESDAY

WEDNESDAY WEDNESDAY

THURSDAY THURSDAY

FRIDAY FRIDAY

SATURDAY SATURDAY

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Assessments

An adapted version of the Toddler Arm Use Test (TAUT) was used to assess arm function throughout the course of intervention. The TAUT is a standardized observational test in which 21 functional tasks and play activities are scored in respect to the functional use of the affected extremity (Taub et al., 2004). During assessment, the examiner tries to elicit the child’s best effort to perform each task. If the child does not demonstrate any use of the affected extremity in the fi rst attempt, she is requested to perform the task again using the affected arm. Separate scales are used to score performance in relation to amount of participation of the affected arm, how well the arm is used, and willingness to use it. In the participation scale, scores vary from 0 to 2. Score 0 is given if the child does not attempt to use the affected arm, 1 if the child moves the affected arm during the task, but it does not contribute to task completion, and 2 if the child uses the affected arm to carry out the task, regardless of whether the task was performed in an age-typical manner. The how-well scale is scored from 0 to 5, with 0 indicating that the child does not use the arm during the task and 5 indicating that the ability to use the weaker arm for that activity was typical for age. Intermediate scores indicate very poor, poor, fair and almost normal performance.

Willingness scores vary from 0, indicating that the child never attempted activity with the affected arm, to 3, indicating that the child demonstrated no resistance to use the affected arm and attempted activity with it with minimal prompting. Intermediate scores indicate considerable and some resistance to use the affected arm (Taub et al., 2004).

The TAUT has been used to document the effi cacy of interventions aimed at improving function of the less-involved upper extremity in children with hemiplegic cerebral palsy (Taub et al., 2004). In the reported course of intervention, 15 of the 21 tasks were considered appropriate to the age of the child and were therefore used in assessments (Table II).

The fi rst assessment was conducted before initiation of intervention, and subsequent assessments were performed at every 2 weeks, when task schedules were changed and new recommendations were given to the mother. Intervention lasted 14 weeks, and thus included seven schedule changes and eight assessments sessions, carried out in a university paediatric laboratory facility and videotaped to be scored by the therapist after the

end of treatment. After the intervention protocol, the mother was interviewed about her perceptions and opinions regarding treatment.

Results

Figure 2 demonstrates the changes in the scores of the three TAUT scales. In the amount of participation scale, the percentages of each possible score among the 15 tasks were approximately similar in the initial evalu- ations. From the fi fth assessment session on, no 0 scores were observed anymore for any of the 15 tasks, and the percentage of scores 1 decreased gradually while there was a concomitant increase in the percentage of scores 2. In the last assessment, the child received the maximum score of 2 in 93% of the 15 tasks. These results indicate that in the beginning of intervention, the child did not move the affected arm or the movement she demonstrated was not suffi cient to complete most of the tasks. After 14 weeks, however, the arm was used to perform most of the 15 tasks.

The how-well scale scores, presented in Figure 2, demonstrate improvement in movement quality during the course of intervention. From the fi fth assessment on, no 0 scores were attributed to any task, and the frequency of scores 1 and 2 decreased while there was a general increase in the percentage of scores 3 and 4.

In the fi rst assessment, the child did not use the affected arm (score 0) or had very poor (score 1) or poor arm performance (score 2) in most of the tasks (67%) In the

Table II. TAUT tasks used in assessments

TAUT tasks used in assessments 1 Strike a toy with hammer 2 Use markers for scribbling 3 Pick up snack

4 Pull lever 5 Push button

6 Remove puzzle pieces with large knob 7 Remove puzzle pieces with small knob

8 Squeeze horn 9 Remove ball 10 Place or throw ball

11 Remove phone (vertical orientation) 12 Remove phone (horizontal orientation)

13 Unclip clothes pin (weak spring) from the edge of vertical surface

14 Unclip clothes pin (moderate spring) from edge of vertical surface

15 Unclip clothes pin (strong spring) from edge of horizontal surface

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last assessment, performance of the affected arm was considered fair (score 3) or almost normal (score 4) for 58% of the tasks.

The willingness to use scores (Figure 2) also demonstrated improvement. There was a general decrease in the frequency of scores 0 (no attempt to use the affected arm) throughout the assessment sessions and this score was no longer observed from the fi fth assessment on.

There was also a general decrease in the number of tasks for which the child demonstrated considerable resistance to use the arm (score 1), and an increase in the number of tasks for which there was only some resistance (score 2). In the last assessment, no scores 0 or 1 were observed; 92% of the tasks were scored 2 and 8%

of the tasks were scored 3 for the fi rst time, indicating the child demonstrated no resistance to use the affected arm during those tasks.

Figure 2 also illustrates how the child’s mother rated her performance in the tasks practised each day throughout the intervention period (according to the mother, all activities were practised in all scheduled days) In the fi rst assessment, performance of the child was considered poor for 10%, good for 57% and very good for 33% of the days in the 2-week period. From the fourth task schedule on, the child’s daily performance was

never again considered poor. In the last assessment, performance was considered good in 2% and very good in 98% and of the days of the last 2-week period.

The interview at the end of treatment revealed the mother had perceived improvements in manual performance. She reported positive gains in her child’s abilities to perform bimanual activities such as combing the hair, eating and dressing up. The child performed these activities with more consistent and effi cient assistance from the affected arm. As a strong point of treatment, the mother mentioned that the task training moments stimulated stronger mother–child bonds, since she could ‘be closer to’ her daughter, ‘playing with her and witnessing her development’. As a weakness of the protocol, the mother mentioned she initially thought the restrictive elastic band to be somewhat aggressive to the child because her daughter demonstrated irritation when using it. However, both mother and child got used to the restriction as they played and performed the scheduled tasks.

Discussion

Explanations about the mechanisms involved in recovery of function after CIMT are discussed for cases of

Figure 2 Percentage distribution of Amount of Participation, How-well and Willingness scores for the 15 Toddler Arm Use Test tasks tested in each assessment and percentage distribution of the daily ratings given by the child’s mother to her performance during training in each 2-week period

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central nervous system injuries. Different authors suggest that the positive effects of this technique on upper extremity function are related to two possible mechanisms, the use-dependent plasticity of the central nervous system (Liepert et al., 2000; Wittenberg et al., 2003; Kim et al., 2004) and the overcoming of learned non-use, or in the context of child development, developmental disregard (Taub et al., 1994, 2004). Evidence demonstrated increased activation of cortical areas related to movement of the affected extremity after CIMT in patients with central nervous system injuries (Liepert et al., 2000). Long-lasting cortical reorganiza- tion has also been shown to occur after a peripheral nerve injury and there is some evidence of peripheral plasticity involved in early functional recovery after OBP (Lundborg 2000; Vredeveld et al., 2000). Whether training can infl uence mechanisms of peripheral and central plasticity in OBP is not yet known and should be investigated.

Developmental disregard, the decreased functional use of the extremity in relation to its motor potential, may be present in children with OBP. Before treatment, the child’s mother expressed concern on the fact that she thought her daughter had potential to use the affected arm more frequently and effectively than she usually did. The therapist had similar perceptions, and the hypothetical negative role of learned non-use on functional performance was a reason to indicate a treatment schedule based on CIMT principles. In young infants, under the perspective of motor learning, the early implementation of the technique could minimize developmental disregard and prevent the appearance of non-use behaviours, directing the infant’s attention to the affected arm and consequently, promoting the use of the affected extremity. The infant may realize that the incorporation of the affected limb into daily tasks may bring on improvements in functional abilities (Glover et al., 2002; Coker et al., 2009). The perception of the possible use of the affected extremity in daily manual activities would lead to the overcoming of developmental disregard.

The progress demonstrated by the child throughout the 14 weeks of intervention is coherent with these positive expectations and indicates that effects of CIMT protocols for children with OBP should be further investigated. According to scores in 15 TAUT tasks, the child became more willing to move the affected arm and it participated more consistently in the performance of the analysed tasks, with better movement

quality. These results are similar to those reported for children with hemiplegic cerebral palsy submitted to CIMT (Eliasson, et al., 2003, 2005; Taub et al., 2004;

Gordon et al., 2005; Naylor and Bower, 2005). The effects observed in this clinical case may however be not entirely attributed to treatment effects on developmental disregard, but to a learning effect of repeated testing.

This factor should be accounted for in controlled experimental investigations. Nevertheless, even in case learned non-use is not a limiting factor for functional performance of children with OBP, systematic task training during intervention sessions or repeated testing might be able to produce functional gains for these children, as was suggested by the positive perceptions of the mother regarding to her child’s abilities.

Adhesion to the treatment protocol was excellent.

Despite the young age of the child and the initial con- cerns about frustration and irritability, mother and child soon adapted to the routine of training. The fact that the mother administered the training in short sessions at home may have favoured such adaptation.

Also, the functional relevance of the tasks and activities selected for intervention, which were identifi ed by the mother, certainly contributed to the improvement. The greater grades attributed by the mother to the child’s performance during sessions from the fourth task schedule on might also refl ect decreased irritability and decreased resistance to take part in the proposed activities. Nevertheless, this kind of intervention may not be adequate for all children, as some might not tolerate the frustration of having movement of the non-affected arm restricted. Therefore, intervention demands must be fi nely adjusted to the level of performance presented by each child in order to avoid excessive frustration.

Children presenting a very low level of active control over the affected upper extremity may thus not benefi t from this approach. Guidelines to the minimal performance requirements for eligibility to this kind of intervention will need to be determined with more systematic group studies. Additionally, not all parents have the necessary resources to conduct home-based treatment.

Intervention conducted in the clinical environment might be more appropriate to some families.

A limitation of the present study regards the meth- odological design. Although studies such as an A-B-A single case design could offer stronger support to the claim of clinical changes during treatment, a less strong study design based on a case study was used. Consider- ing the specifi c functional complaints and the busy

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family routine, we concluded it would be clinically most appropriate to have 30-minute daily sessions conducted at home by the child’s mother. Given the short duration of sessions, treatment had to be extended in time and was conducted for 14 weeks. The mother was trained and systematically monitored to comply with and meet the intervention’s principles and procedures. We believe that this arrangement maximized the implementation of CIMT principles into the daily routine of the child. Given the recom- mendation of similar lengths for each phase of a single case design (Portney and Watkins, 2009) an A-B-A study would require 11 months of repeated assessments every other week. The feasibility of taking part in such a study would be very limited for any of the families attending our outpatient clinic, because of diffi culties with both fi nancial and time resources. A case study reporting the clinical changes that accompany an intervention based in CIMT principles for a young child with OBP can provide relevant contributions to the fi eld as evidence regarding the effects of CIMT- based interventions is promising for children with upper extremity functional defi cits because of cerebral palsy but very limited for children with OBP (Buesch et al., 2009).

Conclusion

The results from this study suggest that intervention based on CIMT principles has potential to promote functional gains for children with OBP and suggest further investigations regarding the application of CIMT for peripheral nervous system injuries as well as the neurophysiological and behavioural mechanisms related to functional improvement. Controlled experimental studies should test the effects of this kind of intervention specifi cally for children with OBP.

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