Cerebral Palsy (CP) is a group of neurological disorders affecting movement, muscle tone, and posture due to brain injury occurring during early development. Dyskinetic CP, a subtype resulting from basal ganglia impairment, is characterised by involuntary movements such as dystonia and athetosis, leading to fluctuating muscle tone, postural instability, and difficulties with speech and feeding. This case report presents the clinical management of a four-year-old male diagnosed with dyskinetic CP treated by an optimised Ayurvedic regimen, highlighting Taila Dahana Agnikarma and Panchakarma treatments, encompassing Udwartana, Abhyanga, Swedana, Basti, kati basti and Pratimarsha Nasya. These therapies constituted the main therapeutic strategy, whereas physiotherapy and dietary management served as supporting components. The patient improved motor function, muscular tone, postural control, and gait after the intervention. Clinical examinations utilising the Modified Tardieu Scale and Composite Spasticity Index showed spasticity decrease, while goniometers showed range of motion improvements. The successful use of Taila Dahana Agnikarma in dyskinetic CP is unusual and suggests its potential as an additional therapy in paediatric neurorehabilitation.

To give a comprehensive overview of genetic findings in children with cerebral palsy, including a description of subtype, comorbidities and neuroimaging, providing insight into the clinical utility of genetics. A systematic review of previous literature using Embase and Medline as databases. All studies were published between 2000 and 2022, each including at least 10 individuals with cerebral palsy. A total of 19 studies were included, comprising a total of 3707 individuals with cerebral palsy. The overall diagnostic yield was 22.2%, with the highest yield (up to 55%) in cryptogenic cerebral palsy. Variants in a total of 377 unique genes were identified, most frequently CTNNB1, SPAST and ATL1. In addition, 59 different CNVs were identified, of which 32 were in known (micro)duplication/-deletion syndromes such as 22q11.2 microdeletion/-duplication and 14q12 microdeletion. Spastic and dyskinetic cerebral palsy were the most common phenotypes among genetically diagnosed cases. Findings also included variants in genes linked to epilepsy, specific neuroimaging patterns and potentially treatable conditions. These results highlight the importance of genetic evaluation for diagnostic clarification, targeted treatment, monitoring of comorbidities and genetic counseling. We recommend offering genetic testing to individuals with cryptogenic cerebral palsy to optimize management and prevention.

The Visual Function Classification System (VFCS) provides a standardised framework for grading visual functioning in children with cerebral palsy (CP). This study evaluated the reliability and construct validity of the Croatian VFCS, and its ability to distinguish visual functioning across CP subtypes and functional classifications. Ninety-five children with CP (mean age: 11.8 years, range: 4-18) were assessed using VFCS, Gross Motor Function Classification System (GMFCS), Manual Ability Classification System (MACS), and Communication Function Classification System (CFCS). Reliability was tested using quadratic weighted kappa and intraclass correlation coefficients (ICCs); differences across CP types with the Kruskal-Wallis, and construct validity with Spearman correlations. The Croatian VFCS showed excellent interrater ( κ = 0.87) and intrarater reliability ( κ = 0.92), with ICCs greater than 0.90. VFCS levels differed significantly by CP subtype ( χ ² = 8.30, P = 0.016), with preliminary evidence suggesting that unilateral spastic CP may be associated with better visual function than bilateral spastic or dyskinetic CP. Moderate correlations with CFCS ( ρ = 0.557), MACS ( ρ = 0.392), and GMFCS ( ρ = 0.308) revealed clinically relevant divergences between visual and motor abilities. The Croatian VFCS is a reliable, valid, and clinically sensitive tool. This study provides preliminary evidence of its discriminative utility across CP subtypes and supports integrating VFCS into multidisciplinary assessment and individualised rehabilitation planning.

Bilateral globus pallidus internus deep brain stimulation (GPi-DBS) is a proven safe and effective treatment in certain forms of idiopathic or inherited dystonia (ID/IN). Its effects in acquired dystonia such as in dyskinetic cerebral palsy (DCP) however vary widely. The impact of GPi-DBS on speech and swallowing, which significantly affect quality of life, remains poorly understood, especially in pediatric patients. To evaluate GPi-DBS effects on speech and swallowing using the Frenchay Dysarthria Assessment 2 (FDA-2), in pediatric patients with dystonia, and assess how the effects differ between DCP and ID/IN patients. This pro- and retrospective multicenter study analyzes speech and swallowing pre- and 12 months post-GPi-DBS using FDA-2, including prospective data from the STIM-CP trial and retrospective data from the GEPESTIM registry. Twenty-six patients were included (17 male, 9 female; 14 DCP, 12 ID/IN) with mean age of 12.2 years at DBS. No significant changes in FDA-2 total scores were observed pre- and post-DBS (pre: 46.3 ± 33.6; post: 46.3 ± 34.2). ID/IN patients showed consistently higher scores compared to DCP patients both pre- and post-DBS (P < 0.005). When adjusted for age, medication, and pre-surgical values, group differences narrowed, with minimal changes from baseline in both groups. GPi-DBS did not significantly change FDA-2 scores pre- and post-DBS. Assessing speech and swallowing in pediatric patients with dystonia, impaired expressive language and/or intellectual disability is challenging. More comprehensive and patient-centered assessment tools are needed to fully capture DBS effects on these domains in these complex disabled patients.

This study aimed to describe cerebral magnetic resonance imaging (MRI) findings in children with cerebral palsy (CP) and compare them with risk factors and clinical features. Data on 1867 children with CP born in 2000-2016 were retrieved from the Norwegian Quality and Surveillance Registry for CP and linked to the Medical Birth Registry. Descriptive statistics were used. MRI scans were performed in 1463 children, that revealed one predominant pathogenic finding: 44.6% white matter injuries, 28.4% grey matter injuries, 13.5% normal, 7.9% maldevelopments and 5.5% miscellaneous. The proportion of grey matter injuries increased as gestational age increased (p < 0.01). However, white matter injuries were more common in children born at term and small for gestational age (SGA) than in those not SGA (p = 0.03). In term-born children, grey matter injuries decreased with increasing 5-min Apgar scores (p < 0.01). Half of the children with spastic CP had white matter injuries, while half of those with dyskinetic CP had grey matter injuries. White matter injuries were the most common, regardless of the Gross Motor Function Classification System level. Children with epilepsy mainly had grey matter injuries. This study highlighted the vulnerability of white and grey matter in preterm and term infants with CP, respectively.

Clinical Phenotype, Predictors and Early Biomarkers of Dyskinetic Cerebral Palsy Prognosis.

We aimed to establish the clinical characteristics of children and young people (CAYP) currently receiving deep brain stimulation (DBS) therapy for the management of movement disorders in the UK to better inform planning of future service provision. Cross-sectional service evaluation of centres providing DBS for the management of movement disorders in childhood. A total of 139 CAYP were identified across three centres. Median age at surgery was 9.8 years (range 2.0-18.9 years), and median duration of DBS was 4.4 years (range from 1 week to 15.75 years). Modal Gross Motor Function Classification System level was V (n=66). The most common causes of movement disorder were dyskinetic cerebral palsy (69/139, 49.6%), dystonia due to mutations in the lysine methyltransferase 2B gene, aka DYT-KMT2B, (13/139, 9.4%) and dystonia due to mutations in the Torsin-1A gene, aka DYT-TOR1A, (9/139, 6.5%). A monogenetic cause of dystonia without evidence of central nervous system pathology on MRI was identified in 30 CAYP (21.6%). Clinically significant dystonia was present in all CAYP, with significant chorea in 47/139 (33.8%) and significant spasticity in only 13/139 (9.4%). No tone-reducing medications were currently used by 43/139 (30.9%) of CAYP. The remaining 96/139 CAYP were currently receiving 1-6 tone-reducing medications, most commonly gabapentin (n=58), clonidine (n=50) and a form of benzodiazepine (n=43). Despite care being provided by paediatric services, 37/139 (26.6%) of CAYP were >18 years of age. CAYP currently receiving DBS therapy represent a heterogeneous population in terms of dystonia aetiology, functional level and additional pharmacological management. Only 102 CAYP<18 years of age are currently receiving DBS therapy in the UK, representing a small proportion of the population who could benefit from this intervention.

Obstructive sleep apnea (OSA) can affect the growth and development of children, and serious OSA can lead to significant complications if left untreated. Children with cerebral palsy (CP) also experience sleep problems. Therefore, the present study examined the prevalence and differences of OSA in children with CP compared to healthy children. In addition, it sought to classify the children with CP according to different severities and clinical types in order to investigate whether there are differences in the severity of OSA and in sleep and respiratory characteristics among groups of children with different severities of CP. One hundred and fifty-six children with CP and one hundred and fifty healthy children completed the Pediatric Sleep Questionnaire (PSQ). Based on the PSQ results, a threshold of 0.33 was used to define high risk of OSA. Children identified as high risk underwent polysomnography (PSG) to analyze their sleep structure, breathing events, oxygen saturation, and heart rate. 48.7% of children with CP and 26.67% of healthy children identified as high risk of OSA. The prevalence of OSA in children with CP (23.72%) was higher than that in healthy children (7.3%). Thus, children with CP have both a higher risk and a higher prevalence of OSA than healthy children, but the difference in the severity of OSA was not statistically significant.Also, there were no differences in OSA severity among groups of children with varying severities and types of CP. The proportion of rapid eyes movement (REM) sleep time, minimum transcutaneous oxygen saturation (SpO2), and average SpO2 during the sleep period was lower in children with CP than in healthy children. However, the N2 ratio, sleep latency (SL), OAI, OAHI and ODI were higher in children with CP as compared to healthy children. Additionally, the average and maximum heart rates during sleep were higher in children with CP than in healthy children. According to the severity of CP and clinical classification, children with severe CP exhibited a lower proportion of REM sleep time, a lower N3 ratio, reduced total sleep time (TST), decreased sleep efficiency (SE), and lower average SpO2 during the sleep period compared to children with mild and moderate CP. However, they had a higher N2 ratio, increased OAI, elevated ODI, and a higher maximum heart rate during the sleep period compared to children with mild and moderate CP. Children with dyskinetic CP had a higher ODI index and N2 ratio than children with spastic CP. Children with CP have a higher risk and prevalence of OSA than healthy children. Although there were no differences in OSA severity among groups of children with varying severities of CP, children with severe CP exhibited more severe sleep disturbances and breathing issues compared to children with mild and moderate CP.

To identify gaze responses of children with cerebral palsy (CP), cerebral visual impairment (CVI), and severe intellectual and developmental disabilities (IDD) to unimodal and bimodal sensory stimuli. Forty children (27 female; mean age 8 years 3 months [range: 3-21 years] with two participants [ages 20 years and 21 years] representing outliers; Gross Motor Function Classification System [GMFCS] level V, Manual Ability Classification System level V, Communication Function Classification System level V) with spastic and dyskinetic CP and severe IDD (20 CVI, 20 no visual impairment) participated in an experimental-observational study. Unimodal (light, auditory, tactile) and bimodal (visual-auditory, visual-tactile) stimuli were presented in a darkened room. Standardized and validated microanalysis of video recordings assessed gaze frequency, duration, and latency. Compared to children with no visual impairment, children with CVI had significantly longer gaze latency to static visual and visual-tactile stimuli (p <0.05) and shorter overall gaze duration (p = 0.005). The CVI group showed sensory enhancement, responding more frequently to bimodal stimuli than unimodal non-visual stimuli (p = 0.014) and with significantly longer gaze duration to bimodal visual-auditory stimuli than unimodal auditory stimuli (p = 0.005). This study provides evidence that bimodal sensory stimulation can enhance visual engagement in children with CP, CVI, and severe IDD. Gaze frequency, duration, and latency are important considerations for interventions and adapting sensory environments.

Background/Objectives: Dyskinetic cerebral palsy (DCP) often implies severe motor impairment and risk of health problems. Our aim was to follow up a group of young adults with DCP that we previously examined as children, to describe health, function, and living conditions. Methods: Interviews regarding health issues, treatments, and living conditions, and quality of life (RAND-36) and fatigue questionnaires were completed. Gross and fine motor function, communication, and speech ability were classified, and weight, height, spasticity, and dystonia were assessed and compared to previous data. Joint range of motion (ROM) was compared to older adults with DCP. Results: Dystonia was present in all fifteen participants, and spasticity in all but two. A decrease was found mainly in those who received intrathecal baclofen (ITB). ROM limitations were most pronounced in shoulder flexion, abduction and inward rotation (while outward rotation was hypermobile), hip abduction, hamstrings, and knee extension. The majority had frequent contact with primary and specialist healthcare. Seven participants were underweight, eight had a gastrostomy, and seven had ITB. Upper gastrointestinal and respiratory problems were frequent. Orthopedic surgery for scoliosis was reported in five, and lower extremity in nine, while fractures were reported in six participants. RAND-36 revealed physical functioning, general health, and vitality as the greatest problem areas. Fatigue was significant in 64%. Eight participants lived with their parents. Participants at more functional levels completed tertiary education and lived independently. Conclusions: Most participants had severe impairment and many health issues, despite decreased dystonia and spasticity due to ITB. Sleep problems and pain were uncommon.

Background/objectives: Social media has significantly enhanced access to medical knowledge by enabling rapid information sharing. With YouTube being the second-most popular website, we intended to evaluate the quality of its content as a source of information for patients and relatives for information about cerebral palsy. Methods: The first 30 videos for search terms "Cerebral palsy", "Spastic cerebral palsy", "Dyskinetic cerebral palsy", "Worster-Drought syndrome", and "Ataxic cerebral palsy" were selected for inquiry. Out of 150 films, a total of 83 were assessed with a mixed method approach by two independent raters utilizing evidence-based quality scales such as Quality Criteria for Consumer Health Information (DISCERN), the Journal of the American Medical Association instrument (JAMA), and the Global Quality Score (GQS). Furthermore, audience engagement was analyzed, and the Video Power Index (VPI) was calculated for each video. Results: The mean total DISCERN score excluding the final question (subjective assessment of the video) was 30.5 ± 8.7 (out of 75 points), implying that the quality of the videos was poor. The global JAMA score was 2.36 ± 0.57 between the raters. The mean GQS score reached 2.57 ± 0.78. The videos had statistically higher DISCERN scores when they included treatment options, risk factors, anatomy, definition, information for doctors, epidemiology, doctor as a speaker, and patient experience. Conclusions: YouTube seems to be a poor source of information for patients and relatives on cerebral palsy. The analysis can contribute to creating more engaging, holistic, and informative videos regarding this topic.

This retrospective observational study investigates the clinical and neuroimaging profiles of children with cerebral palsy and explores the contribution of genetic factors to its etiology. We reviewed 302 pediatric cases diagnosed with cerebral palsy in Southern Brazil during 2023. Neuroimaging abnormalities were present in 92.1% of cases, with leukomalacia being most frequent. Neonatal encephalopathy emerged as the leading etiology, followed by prematurity and genetic conditions. Genetic testing was performed in 68 patients, identifying 29 distinct genes, notably in cases with preserved imaging or kernicterus. Dyskinetic and ataxic cerebral palsy were more often associated with normal neuroimaging, although not necessarily with positive genetic findings. Some patients with kernicterus also had genetic etiology, especially G6PD. The study reinforces that normal imaging does not exclude underlying genetic causes, especially in patients lacking perinatal complications or exhibiting dyskinetic patterns. These findings emphasize the complementary roles of neuroimaging and genetic in the multifactorial nature of cerebral palsy.

Dyskinetic cerebral palsy encompasses a group of predominantly perinatally acquired complex motor disorders that present with dystonia and/or choreoathetosis and are frequently associated with brain lesions in neuroimaging. Recently, lesion network mapping provided a tool to redefine neurological disorders as circuitopathies. Elucidating the common networks impacted by lesions in this condition could pave the way to identify new targets for neuromodulatory therapeutic approaches. In this study, we aim to assess lesion distribution in dyskinetic cerebral palsy and identify a related functional network derived from lesions. Here, we review the literature of MRI findings in dyskinetic cerebral palsy and perform literature-based lesion network mapping. Articles reporting conventional MRI findings clearly attributable to affected patients were included for review. Imaging findings and their anatomical distribution were extracted and quantified according to an established MRI classification system for cerebral palsy. Reviewed articles were searched for figures depicting lesions and these were traced onto a paediatric template. Whole-brain functional connectivity from lesions causing dyskinetic cerebral palsy was calculated using a paediatric resting-state functional MRI connectome. Individual maps were thresholded and later overlapped to derive a common network map associated with dyskinetic cerebral palsy. Results were contrasted with two control datasets for spatial specificity. Review of 48 selected articles revealed that grey matter injury predominated (51%), followed by white matter injury (28%). In 16% of cases MRI was normal. Subcortical lesions affected the thalamus, pallidum and putamen in >40% of reported patients, respectively. Figures available from 23 literature cases were used to calculate the lesion netwok map of dyskinetic cerebral palsy. The lesion-derived map revealed functional connectivity to a wide network including the brainstem, cerebellum, basal ganglia, cingulate and sensorimotor cortices. The strongest connectivity was found for the motor thalamus. This study confirms subcortical grey matter lesions as the most common MRI finding in dyskinetic cerebral palsy. The neural network identified with lesion network mapping includes areas previously implicated in hyperkinetic disorders and highlights the motor thalamus as a common network node. These results should be validated and their therapeutic implications explored in prospective trials.

Abstract Dystonic cerebral palsy (CP) is a type of dyskinetic CP and causes involuntary muscle contraction and abnormal posture. The symptoms tend to increase when triggered by attempts to move and interfere with activities of daily living and quality of life. The rehabilitation regimen includes medications, spasmolytic injections and tailored physical and occupational therapies.

Aim: To investigate the association between psychological problems and health-related quality of life (HRQOL) parameters in children with cerebral palsy (CP). Method: A convenience sample size of 68 children with CP (mean age: 6.82 years and standard deviation: 1.8; 31 males and 37 females) were recruited in the study. Parent-reported versions of the Strengths and Difficulties Questionnaire (SDQ) and Cerebral Palsy Quality of Life were used to detect the presence of psychological symptoms and HRQOL outcomes in children with CP. Psychological symptoms included emotional symptoms, conduct problems, hyperactivity/inattention, peer problems, and prosocial behavior. Results: Children with hemiplegic, diplegic, and ataxic CP were mostly found to be in scoring band of close to average on SDQ-Total difficulties Scale (TDS) (SDQ-TDS&amp;lt;14), whereas those with quadriplegic and dyskinetic CP were in very high scoring band (SDQ-TDS &amp;gt;20) on the SDQ-TDS. In terms of association, there were moderate to strong associations between scores for psychological symptoms and HRQOL (r=-0.45 to 0.860). Conclusion: The results suggest that the nature and extent of mental health problems vary among CP subtypes, with children with tetraplegic and dyskinetic CP more likely to experience mental health symptoms. Given the potential benefits of physical activity on both HRQOL outcomes and psychological mental symptoms, it is essential to encourage children with CP to participate in more physical activities.

To determine whether post-rewarming brain MRI enables individualized domain-specific prediction of neurodevelopmental outcomes at 2 years of age in infants treated with hypothermia for hypoxic-ischemic brain injury. We conducted a retrospective multicenter study of infants with moderate-to-severe hypoxic-ischemic encephalopathy (HIE) treated with hypothermia. Brain MRI abnormalities and the prediction of domain-specific 2-year neurodevelopmental outcomes were scored independently by 2 investigators after which consensus was reached for both imaging findings and outcome prediction. Neuroimaging patterns were categorized as normal, white matter (WM)/watershed-predominant, deep gray matter (DGM)-predominant, and near-total injury. Outcomes were predicted separately for mortality, cerebral palsy (CP) type and severity, cognitive delay, epilepsy, cerebral visual impairment (CVI), and feeding difficulties; these outcomes were predicted as highly unlikely, possible, probable, or highly likely. Of the 152 study infants, 27 (18%) died. The neurodevelopmental outcome at 2 years was available in all 125 survivors. CP was seen in 21 of 125 surviving infants (17%). No infants in the highly unlikely category developed CP while 90% in the highly likely category did. When CP was predicted as possible, 40% developed CP; all were mild and ambulatory. When CP was predicted as probable, 67% developed CP of whom 40% were severe and nonambulatory. Cognitive scores were available in 104 of 125 infants (83%). Cognitive delay was seen in 23 of 104 infants (22%) (15% mild and 7% severe). When cognitive delay was predicted as highly unlikely, 92% did not develop cognitive delay and the delay was mild in those who did. When cognitive delay was considered highly likely, this developed in 100%. When epilepsy, CVI, and feeding problems were predicted as highly unlikely, 98% did not develop epilepsy; for CVI and feeding problems, this was 100% and 97%, respectively. In 27 of 152 infants (18%), the investigators reached consensus that the overall injury was severe enough to consider redirection of care; 21 of 27 infants (78%) died. Of the survivors, 5 infants developed severe CP and 1 had a mild dyskinetic CP with swallowing problems and CVI. Individualized domain-specific categorical neuroprognostication mainly based on brain MRI is feasible, reliable, and highly accurate in infants with HIE.

Dyskinetic cerebral palsy (CP) is one of the most severe forms of CP, characterized by dystonia or choreoathetosis and can be classified into dystonic and choreoathetosis subgroups. The International Classification of Functioning, Disability, and Health-Child and Youth Version (ICF-CY) provides a framework for physical therapists to understand the health, functioning, activity, participation, and impact of dystonia and choreoathetosis. This review aimed to examine the clinical use of ICF-CY tools to assess body structure, function, and activity in children with dyskinetic CP. A systematic search was conducted in June 2024 using PubMed, Embase, Scopus, and Google Scholar databases. The search included terms related to cerebral palsy, dyskinesia, choreoathetosis, dystonia, body structure, function, and activity. After removing duplicates, 11,800 articles remained and 34 met the inclusion criteria. The review found that ICF-CY activity assessments focused primarily on fine-motor, communication, eating-drinking, bimanual fine motor, and speech functions following gross motor function. Some studies have evaluated ICF-CY body structure and function. Most studies used the Dyskinesia Impairment Scale. This review presents evaluations using instrumented assessments as objective outcome measures in patients with dyskinetic CP. Future studies should develop measurements that are applicable outside the laboratory by using new technologies.

Patient compliance is essential for stabilizing mandibular fractures and ensuring successful outcomes. However, uncontrollable movement disorders can greatly complicate the overall management. This case discusses the management of a mandibular body fracture in a 46-year-old male with dyskinetic cerebral palsy, without using maxillomandibular fixation. The patient fell from a wheelchair, resulting in a mandibular body fracture. He received open reduction and internal fixation, but maxillomandibular fixation was not feasible due to his condition. Severe bruxism led to excessive contractions of the masseter muscle, which complicated recovery. The patient underwent 2 revision surgeries, along with botulinum toxin injections and muscle relaxants. Three months after the final surgery, the patient recovered without complications, and radiographic follow-up confirmed proper fixation. This case demonstrates that for patients with dyskinetic cerebral palsy, combining open reduction and internal fixation with botulinum toxin injections can effectively manage mandibular body fractures and lead to successful healing.

https://onlinelibrary.wiley.com/doi/full/10.1111/dmcn.15237

This study aimed to explore the effects of a four-week intensive eye-tracking intervention on children with dyskinetic cerebral palsy (DCP), focusing on goal attainment, communication competencies, stress levels, subjective workload, and caregivers’ perception of psychosocial impact. A multiple case study design with non-concurrent, staggered multiple baselines was employed, involving three children aged 7, 12, and 13 years. The study included a randomized baseline period of two or three weeks, an intensive eye-tracking intervention, and a six-month follow-up. Two individual eye-tracking goals were identified and assessed using the Goal Attainment Scale, while communication competencies were evaluated with the Augmentative and Alternative Communication Profile: A Continuum of Learning. Stress levels were monitored through Heart Rate Variability measured by the Bittium Faros 360° ECG Holter during eye-tracking tasks. Subjective workload and psychosocial impact were assessed using pictograms and the Psychosocial Impact of Assistive Devices Scale, respectively. Descriptive statistics were applied for analysis. All participants attained and retained their eye-tracking goals, regardless of their initial functional profiles or prior experience with eye-tracking technology. Post-intervention improvements in communication competencies were maintained at the six-month follow-up. Variations in stress levels, subjective workload, and psychosocial impact were observed among participants across different phases of the study, aiding the interpretation of the results. The study concludes that a structured, tailored, four-week intensive eye-tracking intervention can yield successful results in children with DCP, irrespective of their baseline communication abilities or functional profile. Recommendations for future research, including more robust methodologies and reliable computerized tests, are provided.