Journal of Hand Therapy xxx (2015) 1e4
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Special Issue
Therapeutic application of electrical stimulation and constraint induced movement therapy in perinatal brachial plexus injury: A case report Jamie Berggren OTR/L a, *, Lucinda L. Baker PhD, PT b a b
Division of Pediatric Rehabilitation Medicine, Children’s Hospital Los Angeles, 4650 Sunset Blvd., #56, Los Angeles, CA 90027, USA Division of Biokinesiology and Physical Therapy, University of Southern California, Los Angeles, CA, USA
a r t i c l e i n f o
a b s t r a c t
Article history: Received 9 December 2014 Accepted 9 December 2014 Available online xxx
Infants and children with perinatal brachial plexus injury (PBPI) have motion limitations in the shoulder, elbow, forearm and hand that are dependent on the level of injury and degree of recovery. The injury and subsequent recovery period occur during critical periods of central and spinal neural development placing infants and children at-risk for developmental disregard and disuse of the affected arm and hand. A case report outlines the therapy and surgical interventions provided in the first 2 years of life for a child with global PBPI and a positive Horner’s sign. Electrical stimulation and constraint induced movement therapy provided sequentially were effective therapy interventions. Neurosurgery to repair the brachial plexus was performed at an optimal time period.2 The Assisting Hand Assessment,12 Modified Mallet13 and Active Movement Scale14 are effective outcome measures in PBPI and served as valuable guides for therapy intervention. Oxford Level of Evidence: 3b; Individual Case Control Study. Ó 2015 Hanley & Belfus, an imprint of Elsevier Inc. All rights reserved.
Keywords: Perinatal brachial plexus injury Developmental disregard Electrical stimulation Constraint induced movement therapy Assisting hand assessment Modified Mallet Active movement scale
Introduction This retrospective case report outlines the therapy and surgical interventions provided in the first 2 years of life for a child with global (C5-T1) right perinatal brachial plexus injury (PBPI) and a positive Horner’s sign. The incidence of PBPI ranges from 0.38 to 4.6 per 1000 live births.1 Most infants (92%) show partial or full recovery in the first 3 months. Infants with global injury (flail arm) and a positive Horner’s sign generally show no recovery by 3 months and carry a worse prognosis.2 Our primary objective in this case was to determine if the application of electrical stimulation (ES) and constraint induced movement therapy (CIMT) would augment muscle activation and active movement in the affected arm and promote functional recovery. We propose that these targeted interventions along with appropriate surgical intervention, will benefit young infants and children with global PBPI who are at-risk for developmental disregard and subsequent motion and prehensile dysfunction.3 In animal models of PBPI the onset of fibrosis and muscle shortening can begin as early as 4 weeks in the denervated muscles.4 Gentle stretching of the shoulder girdle muscles and the inferior
* Corresponding author. Tel.: þ1 323 361 2118; fax: þ1 323 361 8032. E-mail address:
[email protected] (J. Berggren).
scapula-humeral angle (ISHA) are important interventions to begin early and continue in order to prevent contracture. The effect of electrical stimulation (ES) on nerve regrowth is unclear.5 Direct motor ES has been shown to increase muscle bulk. However, long pulsed (ms) durations can be uncomfortable and is controversial when used during nerve regeneration.6 Some studies using short pulsed (ms) sensory ES after peripheral nerve injury have shown comparatively earlier functional recovery times.7,8 We begin sensory ES on infants with PBPI as a home program as early as 6 weeks to optimize sensory activation of spared nerves in order to enhance cortical awareness. We stimulate the muscles that show loss of bulk. Infants with PBPI may not be able to move their affected arm in a typical pattern or display isolated joint movement even when muscle recovery or reinnervation occurs. They often display muscle co-contraction and stiff movement. Reciprocal ES applied to opposing muscle groups primes the muscles and recreates the natural reciprocal pattern that occurs in early development. This form of motor ES has been shown to be effective in promoting reciprocal muscle activation and recovery of isolated movement in children with cerebral palsy (CP).9 We have also found this approach to be clinically effective in children with PBPI (Fig. 1). Constraint induced movement therapy (CIMT) has been shown to increase use of the affected arm in children with hemiplegic CP.10 Case reports suggest it may also be useful to treat monoplegia and developmental disregard in PBPI.11,12 The use of restraints on the
0894-1130/$ e see front matter Ó 2015 Hanley & Belfus, an imprint of Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.jht.2014.12.006
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initiated. When motor recovery became apparent, sensory ES was stopped. Reciprocal ES (150 ms pulse duration with trace (1/5) level of contraction) was initiated at 11 months to promote efficient motor recruitment and isolated movement.16 CIMT casting of the arm and hand was implemented 4 times to increase the use of the affected arm and focus on specific skill development. Results
Fig. 1. Reciprocal ES for finger flexion and extension.
unaffected arm of infants with PBPI in the form of socks or soft elbow extension splints promotes use of the affected arm when the infant decreases the integration of both hands. Following early restraining with CIMT casting or splinting at strategic intervals promotes visual attention and skill when combined with shaping activities for the affected arm. The Assisting Hand Assessment (AHA),13 Modified Mallet14 and Active Movement Scale (AMS)15 are frequently used outcome measures in PBPI. The AHA measures how well the affected hand is used in bimanual activity.13 The Modified Mallet14 and AMS15 are measures of active motion. In this retrospective case, the AMS was used at periodic intervals starting 2 weeks after birth until age 2 (Fig. 2). The AHA and Modified Mallet were used every 2 months starting at 18 months (Table 1). These outcome measures were used to guide episodes of reciprocal ES, CIMT and bimanual training in order to determine their effectiveness and the necessity for different interventions.
AMS Graph (Fig. 2) depicts changes in active movement over time. Scores below 4 indicate movement with gravity eliminated. Scores above 5 indicate movement against gravity. The initial AMS score at 2 weeks indicated no movement throughout the arm and hand. At 6 weeks there was a palpable contraction in the triceps. Neurosurgery occurred at 3 months. At 7 months (4 months post op) there were palpable contractions in the muscles of the shoulder, elbow and finger flexors. Movement against gravity emerged at 12 months in the elbow, 13 months in the fingers, 14 months in the shoulder, and 20 months in the wrist and thumb. AHA and Modified Mallet (Table 1) At 18 months, the child met the valid age criteria for the AHA and was able to perform the Modified Mallet. These outcome measures were used prior to CIMT casting and 4 weeks after CIMT casting. The AHA scores are reported as logit based 0 to 100 AHA units. An increase of 5 AHA units is significant.17 There were significant increases in the AHA after CIMT casting episodes. Modified Mallet scores were unchanged after the 19 day episode of CIMT casting but increased after the 5 week episode of CIMT casting. Discussion
Intervention The timeline, sequence of interventions and outcomes in this retrospective case report of an infant with global right PBPI between 2 weeks and 2 years of age is depicted in Table 1. At 2 weeks PROM was initiated. At 6 weeks, a home program of sensory ES (100 ms pulse duration for optimal sub-motor threshold) was
This case describes the therapy and surgical interventions in the first 2 years of life for a child with right global PBPI. Targeted interventions provided at specific time points had a positive effect. Early on, to augment muscle activation and prevent joint contracture in the right upper extremity (RUE), daily PROM, gentle stretching of shoulder girdle muscles and sensory ES were used.
Fig. 2. Active movement scale.
J. Berggren, L.L. Baker / Journal of Hand Therapy xxx (2015) 1e4
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Table 1 Timeline of interventions and outcomes Age
Intervention
Measures/protocol
Outcome
2 Weeks
Post-op
PROM/stretching Handout: environmental redesign for your infant with PBPIa Prone positioning on parent’s chest
PROM ISHA AMS
WNL Abduction: 150/160 AMS: 0/7 all Areas
6 Weeks
Sensory ES Posterior/middle Deltoid Biceps Wrist/finger ext. Prone positioning/no support
20 pps 100 ms PD 15 min 2/day AMS
Triceps contraction AMS: 1/7 elbow Extension 0/7 all Other areas
3 Mos
4 Mos
PROM to 90 shoulder until sutures heal/then full PROM AROM/unrestricted
6 Weeks
Resume prone Positioning Sensory ES Infraspinatus, Posterior/middle Deltoid, biceps
8 Weeks
Tolerated prone 10/mins Resumed home program/ muscles determined by OR report Duration/8 weeks Co-contraction at elbow
Soft cast LUE 1 week Post-cast-sock 6 þ times/day
[ visual attention to R side
Approach and reach for objects with R hand
Reciprocal ES Elbow flex/ext.
25 pps 150 ms PD Cycle 10 s ON 10 s OFF 15 min 2/day AMS
Isolated elbow flex and extension AMS results Fig. 2
Able to bring R hand to mouth Duration/7 weeks
Reciprocal ES Finger flex/ext.
25 pps 150 ms PD Cycle 4 s ON 4 s OFF 15 min 2/day
Active opening and closing of fingers
Passive grasp and release
25 pps 150 ms PD Cycle 4 s ON 4 s OFF 15 min 2/day Soft cast on LUE/21 days
Inconsistent pinch with thumb and IF
Duration/12 weeks
Able to reach for objects at shoulder level Active grasp and release
Self-feeding with R hand
AMS
8e9 Mos
5e6 Mos
Motor learning approach used to encourage environmental exploration
Discouraged sitting on floor
10 Mos
7 Mos
CIMT Initially soft cast Later sock
11 Mos
8 Mos
13 Mos
10 Mos
Reciprocal ES Thumb & index finger flex/ext.
CIMT Task specific practice: reaching grasping self-feeding 15e17 Mos
Rolling supine to prone Passive grasp
AMS results Fig. 2 Return of muscle activation in shoulder, elbow and finger flexors Crawling to retrieve toys out of reach
KinesiotapeÔ Y elbow extension [ elbow flex
18e20 Mos
Neck collar and ace bandage used to secure the right arm to trunk for 4 weeks. Circulation checks to elbow/hand 3/day
20 pps 100 ms PD 15 min 2/day
2e4 Mos
11 Mos
a) Neurolysis: C8, T1, inferior trunk; b) Nerve transfer: branch of spinal accessory nerve to suprascapular nerve; c) 8 cable Sural nerve grafts to reconstruct C5 & C6 spinal nerves and upper trunk. Post op protocola
5e7 Mos
14 Mos
Home program/Muscles determined by loss of bulk Duration/6 weeks Tolerated prone 7/min
Nerve exploration and microsurgical repair
4 Weeks
Miscellaneous
CIMT task specific practice: stabilizing holding
Soft cast on LUE/19days: Measures: AMS, Modified Mallet & AHA
Sitting at 8 Mos Crawling at 9 Mos
Duration/5 weeks
Pre-cast: AMS results Fig. 2 Modified Mallet e 11/25 AHA e 49 4 weeks Post cast: AMS results Fig. 2 Modified Mallet e 11/25 AHA e 57 Post-cast bimanual ability: Spontaneously transferred objects between hands
Walking
Moving upper arm and fingers in greater AROM R Hand weak, unable to hold 1 lb. weight (continued on next page)
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J. Berggren, L.L. Baker / Journal of Hand Therapy xxx (2015) 1e4
Table 1 (continued ) Age
Post-op
Intervention
Measures/protocol
Outcome
Miscellaneous
21e23 Mos
18e20 Mos
CIMT task specific practice: orientation manipulation supination
Soft cast LUE/5 weeks per parent request and MD approval Measures: AMS, Modified Mallet, & AHA
With KinesiotapeÔ or a supinator strap [ active supination Pre-cast: AMS results Fig. 2 Modified Mallet e 13/25 AHA e 59
Moving forearm in greater AROM
Measures: AMS, Modified Mallet & AHA
4 weeks Post cast: AMS results Fig. 2 Modified Mallet e 16/25 AHA e 74 Faster shoulder and elbow movement
Able to stabilize/grip objects effectively/no slipping emerging pincer grasp, yet index finger not always engaged
[ Strength
[ RUE use
24 Mos
23 Mos
Trumpeter sign not observed
mos ¼ months, mins ¼ minutes, sec ¼ seconds, ms ¼ microseconds, PROM ¼ Passive Range of Motion, ISHA ¼ Inferior Scapula-Humeral Angle, AMS ¼ Active Movement Scale, ES ¼ Electrical Stimulation, CIMT ¼ Constraint Induced Movement Therapy, AHA ¼ Assisting Hand Assessment, PD ¼ Pulse Duration. a Handout; “Environmental Redesign” and Post Op Protocol are available from the corresponding author on request.
Later on, to promote active movement in the RUE, reciprocal ES, CIMT casting and bimanual training were used. We hypothesized that these interventions influenced central and spinal neural development given the emphasis on arm/hand engagement in task specific practice in the clinic and home environment.16,18 Neurosurgery to explore and repair the brachial plexus occurred at an optimal age (3 months) to allow for reinnervation and muscle recovery. The AHA, Modified Mallet, and AMS scores changed after intervention with significant increases in the AHA scores after episodes of CIMT. The ability of parents to engage in treatment and implement home-based strategies can be pivotal to the success of intervention. The parents in this case were exemplary in their active participation during treatment sessions and their ability to integrate the habitual use of ES and task specific practice during CIMT into their daily routines. They creatively engaged their child in playful practice sessions. They felt strongly that longer periods of CIMT were more effective than shorter bouts. At the time this case was written, the child was 2 years old and had full PROM in the RUE, almost full AROM in shoulder, elbow and wrist with functional AROM in wrist and thumb. He used the affected hand collaboratively with the unaffected hand with no differences in limb length or girth of his arms. The therapy and surgical interventions contributed to these results. Despite the improvement, the child continues to have difficulty using the index finger and thumb in palmar pinch tasks. Although he has full elbow flexion he does not always use it during activities. Over the next 6 months we plan to work on increasing hand strength, function and frequency of use in the affected arm and hand. We will continue periodic use of ES and CIMT if the frequency of use in the affected arm and hand decreases. The following questions remain: Would infants with all levels of PBPI benefit from these targeted interventions? If intervention was strategically timed during the first 2 years of life, could developmental disregard be prevented? The child in this case moved comfortably and easily from sensory to reciprocal ES. Would other infants and children tolerate this stimulation? Repeated CIMT sock restraint and casting was effective in this case. How do we determine the best doseeresponse for this mode of treatment? We look forward to answering these questions in collaboration with others dedicated to the provision of successful treatment for infants with PBPI.
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