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Actigraph-based quantification of sleep in children with dystonia undergoing deep brain stimulation

Frederick Zhang, Karim Mithani, Sara Breitbart, Han Yan, Alfonso Fasano, George M. Ibrahim, and Carolina Gorodetsky

OBJECTIVE

Dystonia is among the most common pediatric movement disorders and can manifest with a range of debilitating symptoms, including sleep disruptions. The duration and quality of sleep are strongly associated with quality of life in these individuals and could serve as biomarkers of dystonia severity and the efficacy of interventions such as deep brain stimulation (DBS). Thus, this study investigated sleep duration and its relationship to disease severity and DBS response in pediatric dystonia.

METHODS

Actigraphs (wearable three-axis accelerometers) were used to record multiday sleep data in 22 children with dystonia, including 6 patients before and after DBS implantation, and age- and sex- matched healthy controls. Data were preprocessed, and metrics of sleep duration and quality were extracted. Repeated-measures statistical analyses were used.

RESULTS

Children with dystonia slept less than typically developing children (p = 0.009), and shorter sleep duration showed trending correlation with worse dystonia severity (r = −0.421, p = 0.073). Of 4 patients who underwent DBS and had good-quality data, 1 demonstrated significantly improved sleep (p < 0.001) postoperatively. Reduction in dystonia severity strongly correlated with increased sleep duration after DBS implantation (r = −0.965, p = 0.035).

CONCLUSIONS

Sleep disturbances are an underrecognized marker of pediatric dystonia severity, as well as the effectiveness of interventions such as DBS. They can serve as objective biomarkers of disease burden and symptom progression after treatment.

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Case-based explanation of standard work tools for selective dorsal rhizotomy for cerebral palsy

Nathan A. Shlobin, Med Jimson D. Jimenez, Maryam N. Shahin, Lindsey Hofflander, Robin Trierweiler, Jennifer Misasi, Joshua M. Rosenow, Ana-Marie Rojas, and Jeffrey S. Raskin

OBJECTIVE

Spasticity is a challenging feature of cerebral palsy (CP) that may be managed with selective dorsal rhizotomy (SDR). Although standard work tools (SWTs) have recently been utilized to inform a standard of care for neurosurgical procedures, no SWTs for SDR have been previously described. The authors present the multidisciplinary approach SWTs for SDR used at their institutions to promote consistency in the field and minimize complication rates.

METHODS

A multidisciplinary approach was used to define all steps in the SDR pathway. Preoperative, intraoperative, and postoperative workflows were synthesized, with specific efforts to improve mobility through inpatient rehabilitation and minimize infection.

RESULTS

The SWTs have been implemented at two institutions for 7 years. An illustrative case of a patient aged 3 years 10 months with a history of premature birth at 29 weeks, spastic-diplegic CP, right-sided periventricular leukomalacia, and developmental delay who underwent L2–S1 SDR is presented.

CONCLUSIONS

The authors detail SWTs for SDR developed by a multidisciplinary team with specific steps at all points in the patient pathway. The illustrative case emphasizes that SWTs may help ensure the safety of SDR while maximizing its long-term efficacy for individuals with CP.

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Editorial. Is percutaneous thermal radiofrequency rhizotomy a viable alternative to selective dorsal rhizotomy or intrathecal baclofen for nonambulatory children with severe spasticity from cerebral palsy?

Kim Burchiel

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Evaluation of the long-term effect of selective tibial neurotomy for the treatment of spastic foot using a personal goal-centered approach

Corentin Dauleac, Jacques Luaute, Gilles Rode, Marc Sindou, and Patrick Mertens

OBJECTIVE

The objective of this study was to evaluate the long-term effectiveness of selective tibial neurotomy (STN) for the treatment of the spastic foot using a goal-centered approach.

METHODS

Between 2011 and 2018, adult patients with a spastic foot (regardless of etiology) who received STN followed by a rehabilitation program were included. The primary outcome was the achievement of individual goals defined preoperatively (T0) and compared at 1-year (T1) and 5-year (T5) follow-up by using the Goal Attainment Scaling methodology (T-score). The secondary outcomes were the presence of spastic deformities (equinus, varus, and claw toes), modified Ashworth scale (MAS) score for the targeted muscles, and modified Rankin Scale (mRS) score at T0, T1, and T5.

RESULTS

Eighty-eight patients were included. At T5, 88.7% of patients had achieved their goals at least "as expected." The mean T-score was significantly higher at T1 (62.5 ± 9.5) and T5 (60.6 ± 11.3) than at T0 (37.9 ± 2.8) (p < 0.0001), and the difference between T1 and T5 was not significant (p = 0.2). Compared to T0, deformities (equinus, varus, and claw toes; all p < 0.0001), MAS score (p < 0.0001), and mRS score (p < 0.0001) were significantly improved at T1 and T5. Compared to T1, MAS score increased slightly only at T5 (p = 0.05) but remained largely below the preoperative value. There was no difference between T1 and T5 regarding other clinical parameters (e.g., deformities, walking abilities, mRS score).

CONCLUSIONS

This study found that STN associated with a postoperative rehabilitation program can enable patients to successfully achieve personal goals that are sustained within a 5-year follow-up period.

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History and evolution of surgical treatment for spasticity: a journey from neurotomy to selective dorsal rhizotomy

Jorge Cespedes, Oscar Andrés Escobar Vidarte, María José Uparela, Enrique Osorio-Fonseca, and Jorge E. Alvernia

The evolution of neurosurgical approaches to spasticity spans centuries, marked by key milestones and innovative practitioners. Probable ancient descriptions of spasmodic conditions were first classified as spasticity in the 19th century through the interventions of Dr. William John Little on patients with cerebral palsy. The late 19th century witnessed pioneering efforts by surgeons such as Dr. Charles Loomis Dana, who explored neurotomies, and Dr. Charles Sherrington, who proposed dorsal rhizotomy to address spasticity. Dorsal rhizotomy rose to prominence under the expertise of Dr. Otfrid Foerster but saw a decline in the 1920s due to emerging alternative procedures and associated complications. The mid-20th century saw a shift toward myelotomy but the revival of dorsal rhizotomy under Dr. Claude Gros’ selective approach and Dr. Marc Sindou’s dorsal root entry zone (DREZ) lesioning. In the late 1970s, Dr. Victor Fasano introduced functional dorsal rhizotomy, incorporating electrophysiological evaluations. Dr. Warwick Peacock and Dr. Leila Arens further modified selective dorsal rhizotomy, focusing on approaches at the cauda equina level. Later, baclofen delivered intrathecally via an implanted programmable pump emerged as a promising alternative around the late 1980s, pioneered by Richard Penn and Jeffrey Kroin and then led by A. Leland Albright. Moreover, intraventricular baclofen has also been tried in this matter. The evolution of these neurosurgical interventions highlights the dynamic nature of medical progress, with each era building upon and refining the work of significant individuals, ultimately contributing to successful outcomes in the management of spasticity.

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Hoffmann’s sign in cervical spondylotic myelopathy patients: pathological insights from neuroimaging

Jianchao Chang, Kun Zhu, Ying Wang, Siya Zhang, Yan Li, Junxun Zuo, Bingyong Xie, Haoyu Ni, Jiyuan Yao, Zhibin Xu, Sicheng Bian, Tingfei Yan, Xianyong Wu, Senlin Chen, Peng Xu, Peiwen Song, Yuanyuan Wu, Cailiang Shen, Jiajia Zhu, Yongqiang Yu, and Fulong Dong

OBJECTIVE

Hoffmann’s sign testing is a commonly used physical examination in clinical practice for patients with cervical spondylotic myelopathy (CSM). However, the pathophysiological mechanisms underlying its occurrence and development have not been thoroughly investigated. Therefore, the present study aimed to explore whether a positive Hoffmann’s sign (PHS) in CSM patients is associated with spinal cord and brain remodeling and to identify potential neuroimaging biomarkers with diagnostic value.

METHODS

Seventy-six patients with CSM and 40 sex- and age-matched healthy controls (HCs) underwent multimodal MRI. Based on the results of the Hoffmann’s sign examination, patients were divided into two groups: those with a PHS (n = 38) and those with a negative Hoffmann’s sign (NHS; n = 38). Quantification of spinal cord and brain structural and functional parameters of the participants was performed using various methods, including functional connectivity analysis, voxel-based morphometry, and atlas-based analysis based on functional MRI and structural MRI data. Furthermore, this study conducted a correlation analysis between neuroimaging metrics and neurological function and utilized a support vector machine (SVM) algorithm for the classification of PHS and NHS.

RESULTS

In comparison with the NHS and HC groups, PHS patients exhibited significant reductions in the cross-sectional area and fractional anisotropy (FA) of the lateral corticospinal tract (CST), reticulospinal tract (RST), and fasciculus cuneatus, concomitant with bilateral reductions in the volume of the lateral pallidum. The functional connectivity analysis indicated a reduction in functional connectivity between the left lateral pallidum and the right angular gyrus in the PHS group. The correlation analysis indicated a significant positive association between the CST and RST FA and the volume of the left lateral pallidum in PHS patients. Furthermore, all three variables exhibited a positive correlation with the patients’ motor function. Finally, using multimodal neuroimaging metrics in conjunction with the SVM algorithm, PHS and NHS were classified with an accuracy rate of 85.53%.

CONCLUSIONS

This research revealed a correlation between structural damage to the pallidum and RST and the presence of Hoffmann’s sign as well as the motor function in patients with CSM. Features based on neuroimaging indicators have the potential to serve as biomarkers for assessing the extent of neuronal damage in CSM patients.

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Introduction. Functional neurosurgery for pain and movement disorders with a special emphasis on spasticity

Kim J. Burchiel, Francisco Ponce, Albert Lee, Ahmed M. Raslan, Sharona Ben-Haim, and Marc Sindou

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Letter to the Editor. Considerations for the value of extended reality versus ex cathedra format for neuroanatomy education

Marios Salmas, Dimitrios Chytas, George Noussios, Theano Demesticha, Angelo V. Vasiliadis, and Theodore Troupis

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Long-term follow-up of pallidal deep brain stimulation for craniocervical dystonia: is the globus pallidus internus the best target?

Zhuoling Zhao, Zhiwei Ren, Yongsheng Hu, Kaijia Yu, Fangzhao Yin, Jianyu Li, and Guojun Zhang

OBJECTIVE

Craniocervical dystonia (CCD) is a common type of segmental dystonia, which is a disabling disease that has been frequently misdiagnosed. Blepharospasm or cervical dystonia is the most usual symptom initially. Although deep brain stimulation (DBS) of the globus pallidus internus (GPi) has been widely used for treating CCD, its clinical outcome has been primarily evaluated in small-scale studies. This research examines the sustained clinical effectiveness of DBS of the GPi in individuals diagnosed with CCD.

METHODS

The authors report 24 patients (14 women, 10 men) with refractory CCD who underwent DBS of the GPi between 2016 and 2023. The severity and disability of the dystonia were evaluated using the Burke-Fahn-Marsden Dystonia Rating Scale (BFMDRS). The BFMDRS scores were collected preoperatively, 6 months postoperatively, and at the most recent follow-up visit.

RESULTS

The mean age at onset was 52.0 ± 11.0 years (range 33–71 years) and the mean disease duration was 63.3 ± 73.3 months (range 7–360 months) (values for continuous variables are expressed as the mean ± SD). The mean follow-up period was 37.5 ± 23.5 months (range 6–84 months). The mean total BFMDRS motor scores at the 3 different time points were 13.3 ± 9.4 preoperatively, 5.0 ± 4.7 (55.3% improvement, p < 0.001) at 6 months, and 4.5 ± 3.6 (56.6% improvement, p < 0.001) at last follow-up. The outcomes were deemed poor in 6 individuals.

CONCLUSIONS

Inferences drawn from the findings suggest that DBS of the GPi has long-lasting effectiveness and certain limitations in managing refractory CCD. The expected stability of the clinical outcome is not achieved. Patients with specific types of dystonia might consider targets other than GPi for a more precise therapy.

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Neurosurgical treatment of spasticity: a potential return to the cerebellum

Daniel D. Cummins, Hyun Joo Park, and Fedor Panov

OBJECTIVE

Neurosurgical targeting of the cerebellar dentate nucleus via ablative dentatotomy and stimulation of the dentate nucleus was historically used for effective treatment of spasticity. Yet for decades, neurosurgical treatment of spasticity targeting the cerebellum was bypassed in favor of alternative treatments such as intrathecal baclofen pumps and selective dorsal rhizotomies. Cerebellar neuromodulation has recently reemerged as a promising and effective therapy for spasticity and related movement disorders.

METHODS

In this narrative review, the authors contextualize the historical literature of cerebellar neuromodulation, comparing it with modern approaches and exploring future directions with regard to cerebellar neuromodulation for spasticity.

RESULTS

Neurosurgical intervention on the cerebellum dates to the use of dentatotomy in the 1960s, which had progressed to electrical stimulation of the cerebellar cortex and dentate nucleus by the 1980s. By 2024, modern neurosurgical approaches such as tractography-based targeting of the dentate nucleus and transcranial magnetic stimulation of cerebellar cortex have demonstrated promise for treating spasticity.

CONCLUSIONS

Cerebellar neuromodulation of the dentate nucleus and cerebellar cortex are promising therapies for severe cases of spasticity. Open areas for exploration in the field include the following: tractography-based targeting, adaptive cerebellar stimulation, and investigations into the network dynamics between the cerebellar cortex, deep cerebellar nuclei, and the subcortical and cortical structures of the cerebrum.