Deep Brain Stimulation for Pediatric Movement Disorders

Marks, Warren A.; Honeycutt, John; Acosta, Fernando; Reed, MaryAnn

doi:10.1016/j.spen.2009.04.001

Cited by 69 publications

(57 citation statements)

References 44 publications

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“…However, DBS for secondary dystonia is still controversial, as there are only limited data from this varied patient population. 25 Our study, although limited by a small patient population, shows that there is some statistically significant functional improvement with DBS. However, the BADS and BFMDRS fail to capture the effect on patient comfort that we see after DBS.…”

Section: Discussionmentioning

confidence: 57%

“…They also relied on patient and caretaker reports and serial video assessments to monitor treatment response, since scores alone do not detect subtle changes that may represent significant functional improvements in individual patients. 25 In their initial series of 8 patients younger than 16 years with cerebral palsy-related dystonia who were treated with DBS, they saw a 37.84% improvement in BFMDRS motor scores, a 14.44% improvement in BFMDRS disability scores, and a 19.48% improvement in BADS scores at 6-month followup. However, the 6 patients who were older than 16 years only had an improvement of 8.96%, 1.63%, and 1.39% in BFMDRS motor, BFMDRS disability, and BADS scores, respectively.…”

Section: Discussionmentioning

confidence: 99%

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Deep brain stimulation in children and young adults with secondary dystonia: the Children's Hospital Los Angeles experience

Olaya

Christian

Ferman

et al. 2013

FOC

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Background Dystonia is a movement disorder in which involuntary sustained or intermittent muscle contractions cause twisting and repetitive movements, abnormal postures, or both. It can be classified as primary or secondary. There is no cure for dystonia and the goal of treatment is to provide a better quality of life for the patient. Surgical intervention is considered for patients in whom an adequate trial of medical treatment has failed. Deep brain stimulation (DBS), specifically of the globus pallidus interna (GPi), has been shown to be extremely effective in primary generalized dystonia. There is much less evidence for the use of DBS in patients with secondary dystonia. However, given the large number of patients with secondary dystonia, the significant burden on the patients and their families, and the potential for DBS to improve their functional status and comfort level, it is important to continue to investigate the use of DBS in the realm of secondary dystonia. Object The objective of this study is to review a series of cases involving patients with secondary dystonia who have been treated with pallidal DBS. Methods A retrospective review of 9 patients with secondary dystonia who received treatment with DBS between February 2011 and February 2013 was performed. Preoperative and postoperative videos were scored using the Barry-Albright Dystonia Scale (BADS) and Burke-Fahn-Marsden Dystonia Rating Scale (BFMDRS) by a neurologist specializing in movement disorders. In addition, the patients' families completed a subjective questionnaire to assess the perceived benefit of DBS. Results The average age at DBS unit implantation was 15.1 years (range 6–20 years). The average time to follow-up for the BADS evaluation from battery implantation was 3.8 months (median 3 months). The average time to follow-up for the subjective benefit evaluation was 10.6 months (median 9.5 months). The mean BADS scores improved by 9% from 26.5 to 24 (p = 0.04), and the mean BFMDRS scores improved by 9.3% (p = 0.055). Of note, even in patients with minimal functional improvement, there seemed to be decreased contractures and spasms leading to improved comfort. There were no complications such as infections or hematoma in this case series. In the subjective benefit evaluation, 3 patients' families reported “good” benefit, 4 reported “minimal” benefit, and 1 reported no benefit. Conclusions These early results of GPi stimulation in a series of 9 patients suggest that DBS is useful in the treatment of secondary generalized dystonia in children and young adults. Objective improvements in BADS and BFMDRS scores are demonstrated in some patients with generalized secondary dystonia but not in others. Larger follow-up studies of DBS for secondary dystonia, focusing on patient age, history, etiology, and patterns of dystonia, are needed to learn which patients will respond best to DBS.

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Section: Discussionmentioning

confidence: 57%

Section: Discussionmentioning

confidence: 99%

Deep brain stimulation in children and young adults with secondary dystonia: the Children's Hospital Los Angeles experience

Olaya

Christian

Ferman

et al. 2013

FOC

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“…23 Children with dyskinetic CP, especially those with dystonia, tend to have more severe motor impairment and an increased likelihood of learning disability, epilepsy, and visual and hearing impairments. 15,20 Well-known etiological factors are hypoxic-ischemic encephalopathy (HIE), infection, and hyperbilirubinemia, 15 which preferentially affect the basal ganglia and thalamus.…”

mentioning

confidence: 99%

“…23,35 While primary dystonia has been shown to respond robustly to pallidal stimulation in both adults and children, 1,2,6,12,21,29,39 secondary dystonia, especially that related to CP, appears to be less responsive. 1,2,9,21,23,27,30 The inherent difficulty in evaluating the effect of DBS within this heterogeneous population is that the literature is sparse and fraught with case series that include incomplete data, involve the inconsistent use of dystonia rating scales that may not be sensitive to the subtleties of secondary dystonia, and consist of patients of different age groups suffering from various forms of secondary dystonia. 1,18,23,24 Recently, however, better controlled and more homogeneous studies have been published.…”

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confidence: 99%

Deep brain stimulation for the treatment of childhood dystonic cerebral palsy

Keen¹,

Przekop

Olaya

et al. 2014

PED

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Object Deep brain stimulation (DBS) for dystonic cerebral palsy (CP) has rarely been reported, and its efficacy, though modest when compared with that for primary dystonia, remains unclear, especially in the pediatric population. The authors present a small series of children with dystonic CP who underwent bilateral pallidal DBS, to evaluate the treatment's efficacy and safety in the pediatric dystonic CP population. Methods The authors conducted a retrospective review of patients (under the age of 18 years) with dystonic CP who had undergone DBS of the bilateral globus pallidus internus between 2010 and 2012. Two of the authors independently assessed outcomes using the Barry-Albright Dystonia Scale (BADS) and the Burke-Fahn-Marsden Dystonia Rating Scale–movement (BFMDRS-M). Results Five children were diagnosed with dystonic CP due to insults occurring before the age of 1 year. Mean age at surgery was 11 years (range 8–17 years), and the mean follow-up was 26.6 months (range 2–42 months). The mean target position was 20.6 mm lateral to the midcommissural point. The mean preoperative and postoperative BADS scores were 23.8 ± 4.9 (range 18.5–29.0) and 20.0 ± 5.5 (range 14.5–28.0), respectively, with a mean overall percent improvement of 16.0% (p = 0.14). The mean preoperative and postoperative BFMDRS-M scores were 73.3 ± 26.6 (range 38.5–102.0) and 52.4 ± 21.5 (range 34.0–80.0), respectively, with a mean overall percent improvement of 28.5% (p = 0.10). Those stimulated at least 23 months (4 patients) improved 18.3% (p = 0.14) on the BADS and 30.5% (p = 0.07) on the BFMDRS-M. The percentage improvement per body region yielded conflicting results between rating scales; however, BFMDRS-M scores for speech showed some of the greatest improvements. Two patients required hardware removal (1 complete system, 1 unilateral electrode) within 4 months after implantation because of infections that resolved with antibiotics. Conclusions All postoperative dystonia rating scale scores improved with pallidal stimulation, and the greatest improvements occurred in those stimulated the longest. The results were modest but comparable to findings in other similar series. Deep brain stimulation remains a viable treatment option for childhood dystonic CP, although young children may have an increased risk of infection. Of particular note, improvements in the BFMDRS-M subscores for speech were comparable to those for other muscle groups, a finding not previously reported.

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“…General anesthesia and sedation have each been successfully utilized for DBS in children. 3,4 That being said, there are advantages and disadvantages to using general anesthesia and sedation in children undergoing DBS from both an anesthesia and a neurosurgical perspective. We report a case of VAE in an awake and spontaneously breathing child undergoing DBS.…”

Section: To the Editormentioning

confidence: 99%

Venous air embolism during deep brain stimulation surgery in an awake child

Gooden

Osborn

2009

Can J Anesth/J Can Anesth

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To the Editor,Venous air embolism (VAE) has been reported as a rare occurrence during deep brain stimulation (DBS) for movement disorders. 1 As might be expected, the incidence of VAE is much less with DBS than with other neurosurgical procedures. 1,2 The occurrence of VAE in children undergoing DBS has not been described. General anesthesia and sedation have each been successfully utilized for DBS in children. 3,4 That being said, there are advantages and disadvantages to using general anesthesia and sedation in children undergoing DBS from both an anesthesia and a neurosurgical perspective. We report a case of VAE in an awake and spontaneously breathing child undergoing DBS.A 9-yr-old, 31 kg child with primary dystonia presented to our institution for globus pallidus internus mapping and DBS. Prior to the start of DBS, the patient was sedated with propofol for stereotactic head-frame placement and magnetic resonance imaging to map for electrode placement. Following completion of the study, the patient was transferred awake to the operating room where standard monitors were placed (i.e., bispectral index processed electroencephalogram [BIS monitor] and supplemental oxygen administered via nasal cannula with an end-tidal carbon dioxide [ETCO 2 ] sampling line). The patient was positioned supine on the operating table with a 30°head-up tilt. An infusion of dexmedetomidine was initiated. A 1 lg Á kg -1 bolus iv was given over 10 min, followed by a maintenance infusion of 0.2-0.7 lg Á kg -1 Á hr -1 . The procedure was well underway and uneventful until burr hole drilling. Soon thereafter, the patient began to cough and complain of chest pain. These intraoperative signs and symptoms were accompanied by a decrease in SpO 2 from 100 to 88% and a decrease in ETCO 2 from 39 to 16 mmHg. At this point in time VAE was suspected. The patient was immediately placed in the Trendelenburg position; a facemask was applied, and the surgical field was flooded with saline. The patient remained without evidence of hemodynamic instability or deviations in the electrocardiogram. The decision was made to close the operative field, and within 10 min the patient returned to baseline. Afterwards, a computed tomography scan was performed and the patient remained in the hospital overnight for observation. No further sequelae were observed, and the patient returned a few weeks later for DBS without complications.Venous air embolism in an awake and spontaneously breathing child undergoing DBS has not been reported previously. This case underscores the importance of a high index of suspicion for VAE in the presence of coughing during DBS. A similar case of coughing with VAE was described in an adult patient undergoing DBS. 5 In addition, since this child was awake and complained of chest pain, this finding availed us with useful information. Ultimately, the combination of our patient's cough and complaint of chest pain as well as the information we derived from the monitors confirmed our diagnosis of VAE and led to successful treatment.Competi...

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Deep Brain Stimulation for Pediatric Movement Disorders

Cited by 69 publications

References 44 publications

Deep brain stimulation in children and young adults with secondary dystonia: the Children's Hospital Los Angeles experience

Deep brain stimulation in children and young adults with secondary dystonia: the Children's Hospital Los Angeles experience

Deep brain stimulation for the treatment of childhood dystonic cerebral palsy

Venous air embolism during deep brain stimulation surgery in an awake child

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