Current and future indications for deep brain stimulation in pediatric populations

Lipsman, Nir; Ellis, Michael J.; Lozano, Andrés M.

doi:10.3171/2010.5.focus1095

Cited by 55 publications

(40 citation statements)

References 52 publications

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“…1,3,13,23,24,26,42 Cerebral palsy is the most common cause of secondary dystonia. 33 The incidence of cerebral palsy is approximately 2 per 1000 births.…”

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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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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“…1,3,13,23,24,26,42 Cerebral palsy is the most common cause of secondary dystonia. 33 The incidence of cerebral palsy is approximately 2 per 1000 births.…”

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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“…23,35 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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“…208 Lipsman and colleagues reported 1 patient with secondary dystonia who was treated with DBS in their series, but they did not have any follow-up available for this patient. 118 A recent metaanalysis of 20 articles comprising 68 pediatric and adult patients with cerebral palsy showed a 23.6% improvement in the BFMDRS motor score and a 9.2% improvement in the BFMDRS disability score after DBS. 97 …”

Section: Treatment Of Dystoniamentioning

confidence: 99%

Deep brain stimulation: a mechanistic and clinical update

Karas

Mikell

Christian

et al. 2013

FOC

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Deep brain stimulation (DBS), the practice of placing electrodes deep into the brain to stimulate subcortical structures with electrical current, has been increasing as a neurosurgical procedure over the past 15 years. Originally a treatment for essential tremor, DBS is now used and under investigation across a wide spectrum of neurological and psychiatric disorders. In addition to applying electrical stimulation for clinical symptomatic relief, the electrodes implanted can also be used to record local electrical activity in the brain, making DBS a useful research tool. Human single-neuron recordings and local field potentials are now often recorded intraoperatively as electrodes are implanted. Thus, the increasing scope of DBS clinical applications is being matched by an increase in investigational use, leading to a rapidly evolving understanding of cortical and subcortical neurocircuitry. In this review, the authors discuss recent innovations in the clinical use of DBS, both in approved indications as well as in indications under investigation. Deep brain stimulation as an investigational tool is also reviewed, paying special attention to evolving models of basal ganglia and cortical function in health and disease. Finally, the authors look to the future across several indications, highlighting gaps in knowledge and possible future directions of DBS treatment.

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Current and future indications for deep brain stimulation in pediatric populations

Cited by 55 publications

References 52 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

Deep brain stimulation: a mechanistic and clinical update

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