Stimulation of the globus pallidus internus for childhood‐onset dystonia

Zorzi, Giovanna; Marras, Carlo Efisio; Nardocci, Nardo; Franzini, Angelo; Chiapparini, Luisa; Maccagnano, Elio; Angelini, Lucia; Caldiroli, Dario; Broggi, Giovanni

doi:10.1002/mds.20510

Cited by 138 publications

(115 citation statements)

References 21 publications

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“…dystonia treated with DBS. 42 The 2 patients who were not in status dystonicus had overall improvement in BFM-DRS scores of 31% and 71%. The 1 patient with secondary dystonia who was in status dystonicus prior to surgery had resolution of status dystonicus 1 week after surgery.…”

Section: Discussionmentioning

confidence: 92%

“…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%

“…The 1 patient with secondary dystonia who was in status dystonicus prior to surgery had resolution of status dystonicus 1 week after surgery. 42 Lipsman et al reported on a patient with secondary dystonia who was treated with DBS in their series. However, they did not have any follow-up data for this patient.…”

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: 92%

“…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%

See 1 more Smart Citation

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

View full text Add to dashboard Cite

show abstract

“…The 1 patient with secondary dystonia who was in status dystonicus prior to surgery experienced resolution of status dystonicus 1 week after surgery. 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.…”

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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“…Some patients had favorable outcomes but others experienced no or only minimal improvement (Alterman and Tagliati, 2007;Pretto et al, 2008;Zorzi et al, 2005). This variability in response is most likely due to the heterogeneity of this condition.…”

Section: Cerebral Palsymentioning

confidence: 99%