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Animal models of developmental motor disorders: parallels to human motor dysfunction in cerebral palsy.

ABSTRACT: Cerebral palsy (CP) is the most common motor disability in children. Much of the previous research on CP has focused on reducing the severity of brain injuries, whereas very few researchers have investigated the cause and amelioration of motor symptoms. This research focus has had an impact on the choice of animal models. Many of the commonly used animal models do not display a prominent CP-like motor phenotype. In general, rodent models show anatomically severe injuries in the central nervous system (CNS) in response to insults associated with CP, including hypoxia, ischemia, and neuroinflammation. Unfortunately, most rodent models do not display a prominent motor phenotype that includes the hallmarks of spasticity (muscle stiffness and hyperreflexia) and weakness. To study motor dysfunction related to developmental injuries, a larger animal model is needed, such as rabbit, pig, or nonhuman primate. In this work, we describe and compare various animal models of CP and their potential for translation to the human condition.

SUBMITTER: Cavarsan CF

PROVIDER: S-EPMC6766736 | biostudies-literature | 2019 Sep

REPOSITORIES: biostudies-literature

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Animal models of developmental motor disorders: parallels to human motor dysfunction in cerebral palsy.

Cavarsan Clarissa F CF Gorassini Monica A MA Quinlan Katharina A KA

Journal of neurophysiology 20190814 3

Cerebral palsy (CP) is the most common motor disability in children. Much of the previous research on CP has focused on reducing the severity of brain injuries, whereas very few researchers have investigated the cause and amelioration of motor symptoms. This research focus has had an impact on the choice of animal models. Many of the commonly used animal models do not display a prominent CP-like motor phenotype. In general, rodent models show anatomically severe injuries in the central nervous s ...[more]

PMID: 31411933

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Project description:Purpose This study describes motor speech disorders and associated communication limitations in six variants of progressive supranuclear palsy (PSP). Method The presence, nature, and severity of dysarthria and apraxia of speech (AOS) were documented, along with scores on the Apraxia of Speech Rating Scale-Version 3 (ASRS-3) for 77 (40 male and 37 female) patients with PSP. Clinician-estimated and patient-estimated communication limitations were rated using the Motor Speech Disorders Severity Rating (MSDSR) Scale and the Communicative Effectiveness Survey (CES), respectively. Descriptive statistics were calculated for each of these dependent variables. One-tailed t tests were conducted to test mean differences in ASRS-3 and CES between participants with and without AOS and between participants with and without dysarthria. Spearman rank correlations were calculated between ASRS-3 scores and clinical judgments of AOS and dysarthria severity and between MSDSR and CES ratings. Results Nine participants (12%) had normal speech. Eighty-seven percent exhibited dysarthria; hypokinetic and mixed hypokinetic-spastic dysarthria were observed most frequently. AOS was observed in 19.5% of participants across all variants, but in only 10% exclusive of the PSP speech and language variant. Nearly half presented with AOS in which neither phonetic nor prosodic features clearly predominated. The mean ASRS-3 score for participants with AOS was significantly higher than for those without and correlated strongly with clinician judgment of AOS severity. Mean ASRS-3 was higher for participants with dysarthria than for those without but correlated weakly with dysarthria severity. Mean MSDSR and CES ratings were lower in participants with AOS compared to those without and moderately correlated with each other. Conclusions Motor speech disorders that negatively impact communicative effectiveness are common in PSP and occur in many variants. This is the first description of motor speech disorders across PSP variants, setting the stage for future research characterizing neuroanatomical correlates, progression of motor speech disorders, and benefits of targeted interventions. Supplemental Material https://doi.org/10.23641/asha.14111837.

Project description:BACKGROUND:Scalp acupuncture has been widely used as treatment for motor dysfunction in children with cerebral palsy in China. Previous studies have failed to provide high-quality evidence to demonstrate the effectiveness of this treatment in children with cerebral palsy. No high-quality randomized controlled trials on scalp acupuncture have been published. The aim of this study is to evaluate the effectiveness of Jiao's scalp acupuncture when combined with routine rehabilitation treatment versus routine rehabilitation treatment alone for motor dysfunction in children with cerebral palsy. METHODS/DESIGN:This is a four-centre randomized controlled trial. One hundred cerebral palsy patients with motor dysfunction were enrolled. Patients will be allocated in a 1:1 ratio into either an acupuncture treatment group or a control group. Cerebral palsy patients in the control group will receive conventional rehabilitation treatment, whereas patients in the acupuncture group will receive a combination of scalp acupuncture and conventional rehabilitation treatment. Thirty-six treatment sessions will be performed over a 12-week period. The Gross Motor Function Measure and the Fine Motor Function Measure Scale will be assessed as the primary outcome measures. The Paediatric Evaluation of Disability Inventory and the Cerebral Palsy Quality of Life Questionnaire for Children will be selected as secondary outcome measures. All assessments will be conducted at baseline, week 4 (treatment 12), week 8 (treatment 24), week 12 (treatment 36) and week 24 (follow-up). DISCUSSION:This is the first trial evaluating the efficacy and safety of scalp acupuncture as a treatment for motor dysfunction in children with cerebral palsy. The results of this trial are expected to provide relevant evidence demonstrating that scalp acupuncture can be used as an effective rehabilitation treatment method for improving motor dysfunction in children with cerebral palsy. TRIAL REGISTRATION:ClinicalTrials.gov, NCT03921281. Registered on 19 April 2019.

Dataset Information

Animal models of developmental motor disorders: parallels to human motor dysfunction in cerebral palsy.

Publications

Animal models of developmental motor disorders: parallels to human motor dysfunction in cerebral palsy.

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