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Case Reports |
S de Bode, PhD, is Senior Researcher (Assistant Professor), Sector of Child Neuropsychology, University Medical Center Utrecht, Wilhelmina Children's Hospital, KG 01.327.1, PO Box 85090, 3508 AB Utrecht, the Netherlands.
SL Fritz, PT, PhD, is Clinical Assistant Professor, Department of Physical Therapy, Arnold School of Public Health, University of South Carolina, Columbia, South Carolina.
K Weir-Haynes, PT, is Professor, Department of Physical Therapy, Arnold School of Public Health, University of South Carolina.
GW Mathern, MD, is Professor, Department of Neurosurgery, Mental Retardation Research Center and Brain Research Institute, David Geffen School of Medicine, University of California, Los Angeles.
Address all correspondence Dr de Bode at: s.debode{at}umcutrecht.nl
Submitted August 20, 2007;
Accepted January 12, 2009
 | Abstract |
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Case Description: The patients received a shortened version of CIMT for 3 hours each day for a period of 10 days. In addition, a standard resting splint was used for the unimpaired hand for an 11-day period. Each patient was encouraged to wear the splint for 90% of his or her waking hours. The following outcome measures were used: the Actual Amount of Use Test (AAUT), the Box and Block Test (BBT), and the upper-extremity grasping and motor portions of the Fugl-Meyer Assessment of Motor Recovery (FM).
Outcomes: Immediately after therapy, improvements were found in AAUT and BBT scores, but no improvements were found in FM scores. Three patients underwent brain imaging before and after therapy and showed qualitative changes consistent with reorganization of sensorimotor cortical representations of both paretic and nonparetic hands in one isolated hemisphere.
Discussion: The findings suggest that CIMT may be a feasible method of rehabilitation in individuals with chronic hemiparesis, possibly leading to neuroplastic therapy–related changes in the brain.
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Because the neural basis of brain reorganization following both developmental and acquired insults to sensorimotor cortices and resulting hemiparalysis is not fully understood, hemispherectomy research is important to unequivocally address the consequences of lesions resulting in the complete unilateral loss of original pathways subserving voluntary movements. This research may help to develop specific therapies targeting the use and unmasking of the ipsilateral motor network and to elucidate both possibilities and limitations of rehabilitation in populations with the unavailable contralateral corticospinal tract.
Individuals who have had a hemispherectomy exhibit different degrees of hemiplegia; however, the distal muscles of the upper extremity (UE) often are more severely involved.4 We conducted a case series to investigate the effects of constraint-induced movement therapy (CIMT) in this population. Constraint-induced movement therapy has been demonstrated to be an effective UE intervention for some individuals with hemiplegia.5–7 It is used mainly in people after a stroke to increase functional use of the neurologically weaker UE through massed practice of hand and arm tasks, while restraining the less-affected UE.8 The goals of CIMT are to overcome a learned non-use behavior and to improve functional use of the affected UE by "forcing" use of the affected UE. The focus of CIMT is on the continual plasticity of the cortex; thus, functional improvements should be able to occur at any time.9
In our experience, hemiplegia in children and young adults after hemispherectomy has a clinical presentation similar to those of individuals with a variety of conditions, such as cerebral palsy, stroke, and hydrocephalus. Although there are no formal comparison studies, we have observed similar motor and sensory impairments and similar functional limitations in our patients after hemispherectomy. We suggest that, similar to these other patient groups, the impairments seen in children after cerebral hemispherectomy have a negative effect on their quality of life and restrict their participation in mainstream activities, but the effects of novel therapeutic approaches in this population have not been addressed. Constraint-induced movement therapy has been introduced in pediatric populations relatively recently but not in individuals after hemispherectomy.10
The methodology behind CIMT has been successfully implemented in a pediatric population, and child-friendly guidelines have recently been formulated.11 The etiologies of children who participated in published CIMT studies included cerebral palsy, stroke, trauma, and unknown etiologies. The specifics of traditional CIMT require the restraint of the unaffected UE for 90% of the individual's waking hours for 14 consecutive days. For 10 of the 14 days, the individual engages in 6 hours of intensive therapy involving repetitive tasks with the affected UE.12 Several studies have experimented with the therapeutic approach by changing duration, intensity, or dosage13–16 and indicated positive changes in UE function; however, only portions of the entire CIMT guidelines were utilized, and duration of both treatment and use of limb restraint varied. In our case series, data collection was completed prior to publication of the pediatric guidelines; however, the only main change from traditional CIMT was treatment for 3 hours per day versus the traditional 6 hours per day.
The neurobiological substrate of motor learning has become an area of extensive research since the discovery that experience-driven motor learning causes reorganization of cortical maps.17 However, the neural substrate mediating partial sparing of function in individuals after hemispherectomy remains a poorly researched area and presents a challenge, given the frequency of depressed mental age in this population. In our case series, we applied functional imaging to examine partial recovery of paretic limb function in order to determine whether cortical reorganization can be manipulated through physical therapy to improve the recovery.
After considering the status (effects of seizure medications, low fitness level), depressed mental ages, and decreased emotional maturity of our patients, we chose to use the "shortened" 3-hour method of CIMT to make its use more feasible in this population. The aims of this case series were: (1) to evaluate the feasibility of this therapeutic approach involving a shortened version of the CIMT within a small case series of individuals who had undergone hemispherectomy, (2) to examine improvements that occurred within the upper extremity of the hemiparetic side, (3) to investigate feasibility of conducting brain imaging in individuals with depressed mental ages, and (4) to examine changes in the sensorimotor cortex following intervention.
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All patients had a clinical presentation of hemiplegia after hemispherectomy. None of the patients reported currently receiving rehabilitation. The primary functional inclusion criterion to participate in the therapy was the ability to release a massed flexion grasp.19 Additional inclusion criteria were: (1) ability to lift the hand from the lap to a standard-height table, indicating approximately 30 degrees of active shoulder flexion; (2) ability to follow simple instructions; (3) ability to sit independently without back or arm support for 5 minutes; (4) ability to stand for 2 minutes with assistance, if needed; and (5) at least half of the normal passive range of motion of all UE motions.
Individuals were excluded if they: (1) had metal in their bodies, such as an implanted pacemaker or medication pump, metal plate in the skull, or metal objects in the eye or skull (due to functional magnetic resonance imaging [fMRI] safety requirements); (2) had a mental age lower than 4
years as evaluated by PPVT (to be able to follow commands, pay attention to trainers, and cooperate in a scanner during imaging sessions), or (3) lacked seizure control.
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During the therapy sessions, the intervention was provided on a one-on-one basis for individualized task development for each patient. The activities chosen for the therapy sessions consisted of sets of tasks and games that were performed using the impaired UE, such as grasping pick-up sticks, playing checkers, gripping and throwing tennis balls, and working puzzles. An activity log was used to document the tasks performed and progression of patients throughout the therapy sessions. With improvement in performance and knowledge of the tasks, the complexity and difficulty of the tasks were increased to continue to challenge the patients. The changes in the tasks included timing of tasks, increasing height or distance to perform the task, and increasing pattern complexity within the task. Functional tasks were used; however, they were modified to allow for success based on each patient's degree of finger and hand control. There was no set amount of rest time, but the patients were encouraged to stay active throughout the 3 hours of CIMT, with no more than 10 minutes of rest per hour. All trainers were senior-level Doctor of Physical Therapy students and were overseen by a physical therapist. The same 4 trainers rotated working with the patients (1 or 2 trainers per day).
Outcome Measures
Prior to and following the completion of the therapy sessions, 2 evaluators (a physical therapist and a senior DPT student) obtained scores with the following outcome measures: the Fugl-Meyer Assessment of Motor Recovery (FM),20 the Box and Block Test (BBT),21 and the Actual Amount of Use Test (AAUT),22 consisting of the Amount of Use (AOU) scale and the Quality of Movement (QOM) scale (Tab. 1). Both evaluators were trained in the evaluations and used identical instructions and recording forms. In addition, 3 patients underwent an fMRI session prior to and following therapy, as described below.
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fMRI Methods and Analysis
Three patients who were able to cooperate with fMRI procedures (ie, were comfortable in a scanner room, could remain still, tolerated noise levels, and were capable of performing a functional paradigm) participated in pretherapy and posttherapy imaging sessions and were asked to squeeze a rubber ball during an fMRI scan. We used a rubber ball with loops for individual fingers (GripSaverPlus*). The fMRI studies were performed for both the paretic and nonparetic hands, paying particular attention to avoiding moving both hands synergistically ("mirror movements"). Attempts to squeeze with a weaker hand sometimes result in mirror movements. In order to control for mirror movements, the patients practiced ball squeezing outside the scanner room and then in the scanner after they had been positioned supine. A therapist (SdB) remained with each patient during scanning, along with a physical therapist who monitored for the absence of mirror movements, monitored paradigm adherence, and counted the number of hand flexion and extension movements during each block. Sessions were repeated if mirror movements were noticed.
The patients were cued to start squeezing the ball when they saw a Sesame Street character (Elmo) appear on a screen and to stop when Elmo disappeared. All patients practiced the Elmo paradigm outside of the scanner room and had no problems understanding instructions. During each 3-minute fMRI scan, the patients performed 5 trials of squeezing (20 seconds each) with five 20-second rest periods. The same procedure was repeated for their nonparetic side. The pressure of squeezing may affect the intensity and extent of a signal observed as the brain activates. Ideally, the total pressure generated by each individual should be the same before and after therapy to conclude that observed changes are not due to greater strength (force-generating capacity) but are due to neuroplastic changes. To match a weaker squeeze by the paretic hand, all patients were instructed to squeeze with their nonparetic hand lightly as if they were squeezing a juice box, making sure juice does not spill out. The frequency of squeezing was kept constant between sessions.
Functional MRI scans were obtained using a 3T Philips Intera scanner
at the Medical University of South Carolina, Charleston. A 3-dimensional T1-weighed anatomical scan was obtained in a rapid FLASH acquisition (voxel size=1 x 1 x 1 mm, matrix size=256 x 256 x 160, 15° flip angle, echo time=5.7 milliseconds, repetition time=9.5 milliseconds per FLASH line, effective inversion time=800 milliseconds, frequency encoding head to foot with SENSE r=2 applied in the left-right direction). Functional imaging used a gradient echo planar imaging (EPI) sequence (EPI: repetition time=2,400 milliseconds, flip angle=80°, 36 slices with a slice thickness of 3.25 mm and with no gap oriented parallel to the anterior-posterior commissure line). The fMRI data were processed and displayed using FSL24 and MRIcro(n)25 software.
Data Analysis
Functional scans were co-registered onto anatomical scans and spatially smoothed according to a standard protocol,26 and a cutoff value of .30 (Pearson r coefficient, corresponding to a P value of less than .01 uncorrected) was used to calculate the number of activated voxels above threshold in an anatomically defined region of interest: S1M1. The missing hemisphere was masked prior to analysis, and for each individual, the analysis phase correlated the signal intensity within each voxel over time using a boxcar model with predicted signal increase during the ball squeezing and predicted decrease during rest.
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Therapy Duration: 3 Versus 6 Hours per Day
In this case series, we found that a shortened version of the CIMT is a feasible and tolerable intervention for individuals with longstanding UE dysfunction due to hemispherectomy. A shortened intervention scheme also was particularly appropriate for our patients, who had decreased mental ages and limited ability to sustain motivation.
Most of the current literature on CIMT has focused specifically on the adult population. Two groups of authors, however, used a more "traditional" CIMT delivery for pediatric clients. DeLuca et al5 reported on their use of CIMT in children with an average age of 3.25 years. Karman et al14 investigated the use of CIMT for hemiplegic children with acquired brain injuries whose average age was 12.5 years. Similar to our case series, they also had a small sample size (N=7) and reported significant improvements in all patients. After careful consideration of the specifics of our patients, we decided to follow the suggestions of a report of 3 case studies of adults, which indicated that 3 hours of CIMT significantly improved motor function in individuals with chronic stroke.27 These authors concluded that 6 hours a day of CIMT may have caused frustration, boredom, or undue fatigue for their patients, translating to a decreased capacity for motor learning.28,29 In retrospect, we believe that a 3-hour intervention was an appropriate duration for our patients because of their mental and psychological disabilities.
Adherence to CIMT
During therapy, we were able to maximize adherence to wearing the restraint using strategies such as patient decision making about therapeutic activities and subsequent structured rewards.11 As with all reports of the use of CIMT, these accommodations were achieved at the expense of standardization of the intervention but were crucial for all patients, especially for the patient with the lowest mental age. Although the time frame of 3 hours of intervention daily was a standard, there was no standard protocol to follow to ensure consistency of the intervention across patients. We found that conducting CIMT in these patients was a challenging task. However, by creating individual routines that were congruent with each patient's mental age, physical fitness level, and personal interests, we were able to ensure on-site adherence in all participants.
Outcome Measures
In this case series, our intervention was associated with improved motor function and increased use of the hemiparetic extremity, as measured by the AAUT and BBT. Despite chronic, and often severe, impairment of hand function, all patients made improvements. The degree of improvement varied significantly among the patients (ie, from 10% to 360% for the AOU scale of the AAUT, from 8% to 190% for quality of movements as measured by the QOM scale of the AAUT, and from 5% to 150% for the BBT). Relatively larger improvements in these measures were made by the 2 patients with the lowest initial scores (patients 1 and 3). We propose that the FM may not be an appropriate instrument for measuring changes associated with this kind of therapy. In order to demonstrate improvement on the FM UE motor test, motor movements must advance from in synergy to out of synergy. However, the primary goal of CIMT is increased use of the affected limb. The training used in CIMT does not focus on facilitation or correction of synergistic patterns that may occur with increased tone (resistance to movement) and spasticity (velocity-dependent hypertonicity). As we did not have a control group, we cannot comment on whether documented gains were due to the intervention or to other factors affecting the results. In general, with a case report such as ours, the lack of treatment effect remains a distinct possibility.
Our findings suggest that the time elapsed since hemispherectomy does not appear to be a factor limiting the efficacy of therapy in patients with chronic impairment. Improvements were seen even 18 years after hemispherectomy. However, the effect of this intervention may be different for children immediately after hemispherectomy. In addition, individual differences due to medical variables such as etiology of disease, seizure control, age at surgery, and cognitive level were problems associated with this case series and with prior research in individuals after hemispherectomy. These combined factors could affect outcomes following therapy and should be investigated as possible covariates.
Neurobiological Mechanism of CIMT and fMRI Testing
Rehabilitation in adults is believed to result in the experience-induced expansion of motor maps and related improvements in motor performance.30 Therapy-related improvements in hand function are shown to correlate with increases in fMRI activity in adults,31 and 2 distinct mechanisms of increased synaptic efficiency or reorganization involving extension and recruitment of additional cortical areas were proposed to explain these effects.32
The specific effects of CIMT on cortical reorganization following insult have only recently begun to be addressed. Constraint-induced movement therapy is believed to alter the representation of the UE within the primary motor cortex in adults.33 Only one study has investigated the effects of CIMT in younger patients with congenital hemiparesis and demonstrated increases in activation in the lesioned hemisphere following therapy.34 In contrast to those individuals with both hemispheres in place, we showed that the paretic side in our patients was controlled by the remaining, "healthy" hemisphere, suggesting that residual paretic hand motility is the result of either a partially spared ipsiateral corticospinal tract35 or axonal sprouting of the contalateral corticospinal tract.36 Cortical motor representations of paretic and nonparetic hands were overlapping or closely represented in all 3 patients who underwent fMRI testing.
We conclude that functional imaging is a useful tool in this population and may help to detect reorganization in the remaining hemisphere following intervention. It remains to be seen whether CIMT is associated with the increase in the activation extent or intensity. We did not see an increase in activations, although we did observe subtle shifts in the location of activations that still remained within primary sensorimotor and supplementary motor areas. Due to the preliminary nature of this case report and the small number of patients, we did not investigate correlations between brain activations and motor improvements. We suggest that next step of similar studies should involve studying correlations between changes in fMRI activity and behavioral gains.
Limitations of the Therapeutic Approach
There are a few major limitations of our therapeutic approach. We were unable to determine to what extent cognitive limitations influenced the efficiency or interest of the patients. Many more individuals are needed to adequately control for all clinical variables associated with this group, such as age at surgery, age at seizure onset, and seizure medications. The additional limitations are: (1) the lack of pre-established interrater and intrarater reliability of outcome measurements for the evaluators and the fact that the testers were not masked to test sessions, (2) the lack of quantitative measures of squeezing pressure while measuring brain activation and the unknown effects of task practice, and (3) the lack of information on history of past rehabilitation. Although our case report suggests the feasibility of using CIMT in individuals after hemispherectomy, caution should be exercised to avoid the limitations of our treatment approach.
| Footnotes |
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The authors thank all of the participants and their families for their time, effort, and courage. They also are grateful to all students in the Department of the Exercise Science, University of South Carolina, who took part in this project for their devotion, creativity, and loving professionalism.
This work was supported by National Institute of Diabetes and Digestive and Kidney Diseases/National Institutes of Health grant R21 HD050707.
* Metolius, 63189 Nels Anderson Rd, Bend, OR 97701. 
Philips Healthcare, 3000 Minuteman Rd, Andover, MA 01810-1099.
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