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A 10-year-old child presented to the emergency department with a sudden onset of right hemiparesis and dysphasia. A thorough evaluation, including CT scan of the brain and cerebral angiography, resulted in a diagnosis of acute childhood hemiplegia. The diagnosis of acute childhood hemiplegia depends on characteristic cerebral angiographic findings. The treatment of this particular entity is supportive, often with incomplete recovery.
An insulin-dependent 26-year-old woman was complaining of nocturnal attacks of transient right hemiplegia. Hypoglycemia was found to be present at the time of each attack, and further investigations showed that these neurological disorders were related to an overdose of insulin. The authors discuss the effects of hypoglycemia on the brain and the pathogenetic theory of "hypoglycemic hemiplegia", involving selective neuronal vulnerability, regional blood flow disturbances or underlying ischaemic disease.
OBJECTIVE: To assess the feasibility and reliability of ankle plantar flexor stiffness measurements in hemiplegia. DESIGN: Repeated measurements in five consecutive weeks. BACKGROUND: In hemiplegia, an equinovarus positioning of the foot might be caused by an increased stiffness of the m. triceps surae. METHODS: In eight hemiplegic patients the net joint torque of passive muscle stretch was measured as a function of ankle-angle by a dynamometer, at both sides. Ankle-stiffness was characterised and also a biomechanical model of the passive muscle was fitted. RESULTS: In the vast majority of measurements it was possible to obtain measurements that were not distorted by involuntary muscle contraction. These measurements showed for the angle at which a passive plantar moment of 10 N m was reached a standard error of measurement of less than 2.7 degrees. The muscle model showed the increased stiffness as a shortening of the muscle-fibre-length. CONCLUSION: The feasibility of this method to measure muscle stiffness was fair to good in hemiplegic patients. Provided the abandoning of involuntary muscle activity, the reproducibility warrants application in clinical practice at an individual level. The use of the model relates this changes to a shortened m. soleus and/or m. gastrocnemius. RelevanceEffective clinical decisions for treatment of equinovarus positioning of the foot in the hemiplegic individual, should consider excessive involuntary contractions of the m. triceps surae complex (i.e., spasticity), as well as shortened muscle tissue resulting in high stiffness. Despite the importance of increased stiffness there have been no validated methods of measurement.
A case of Eales' disease with hemiplegia is reported. The patient had a right hemiparesis and a left hemiplegia occurring after a sudden loss of vision in the left eye two years earlier. The diagnosis was based on fluorescein angiography showing neovascularisation, fluorescein leakage, perivascular sheating and hemorrhages and on CT scan showing multiple hypodense lesions in right and left hemispheres and on IDSA showing bilateral occlusion of anterior cerebral arteries. The patient did not have further relapses.
Fifteen children between the age of 3 and 7 years who had had unilateral seizure followed by a transitory hemiplegia (without fever) were observed. This seizure was the only one in 13 of the children; a second fit appeared in 2 others. The average duration of the observation period was 10 years, 6 months. The evolution of the EEG shows that after a post-ictal delta focus contralateral to the hemiplegia, spike foci of variable localisations were formed in 12 cases. These cases, chosen because of their benign evolution, seem to be related closely to rolandic paroxysmal epilepsy (EPR).
Herpes zoster infection has been rarely reported to cause angiitis of the central nervous system in children. We describe a 4-year, 8-month-old female with acute hemiplegia and central facial palsy 6 weeks after she had had zoster ophthalmicus. The findings of magnetic resonance angiography, the clinical picture, and a preceding history of herpes zoster ophthalmicus suggested zoster vasculitis. Herpes zoster vasculitis is thus another consideration when examining a child with acute hemiplegia and a recent herpes zoster infection.
A case of alternating hemiplegia of childhood is reported. Tonic fits and generalized tonic-clonic seizures developed during her infancy. Frequent twitching and apneic seizures appeared at 16 years of age. Zonisamide transiently suppressed the tonic, twitching and apneic seizures, as well as the facial and neck dystonia. Cranial computed tomography and magnetic resonance imaging revealed progressive vermian atrophy. Cerebellar dysfunction may play a role in the clinical features of some patients with alternating hemiplegia of childhood.
BACKGROUND: Chickenpox encephalitis is usually mild and often presents as an acute cerebellar ataxia. Seizures and coma are uncommon. CASE REPORT: A 4-year old girl presented generalized seizures. Two days after the onset of chickenpox, she developed ataxia and left hemiparesis on day 3, followed by left hemiplegia after 24 hours. The cerebral scan showed areas of subcortical hypodensity compatible with leuko-encephalitis. The EEG showed lesions of acute leuko-encephalitis located in the right sub-cortical temporo-fronto-parietal area. The patient was given cortico-steroid and recovered slowly but completely. CONCLUSIONS: Hemiplegia, an exceptional event during the course of chickenpox, makes the diagnosis of chickenpox leuko-encephalitis difficult; IRM appears to be a contributing factor.
The purpose of this study was to investigate the impact of three different ankle foot orthoses (AFO) configurations on the function and kinematics of stair locomotion in children with spastic hemiplegia. Nineteen children were evaluated barefoot and with a hinged, posterior leaf spring (PLS) and solid AFO during stair ascent and descent. Stair specific items from the Pediatric Evaluation of Disability Inventory (PEDI) were used to evaluate function, while a motion measurement system was used to evaluate kinematics. The PEDI revealed no significant differences between AFOs and barefoot, although a greater percentage of children were able to keep up with their peers while wearing a hinged AFO. At the ankle, the hinged AFO provided the greatest amount of dorsiflexion during stance. All AFOs reduced plantarflexion in comparison to barefoot. The results of this study indicate that for children with spastic hemiplegia the use of an AFO did not impair stair ambulation.
Two forms of unilateral tonic (hemitonic) seizure were observed in two adult epileptic patients with infantile hemiplegia. Their ictal EEGs showed diffuse bilaterally synchronous spike bursts without focal features. In one case, a 31-year-old male, the VTR/EEG recording clearly demonstrated hemitonic seizure which simultaneously involved his paretic limbs and body and were associated with loss of consciousness and autonomic signs. In the other case, a 33-year-old male, partial seizures with secondary unilateral generalization were demonstrated by VTR/EEG recording. The reason why these cases do not exhibit motor seizure activity on the unaffected side despite diffuse bilateral EEG changes, a lower motor threshold in the paretic limbs and inhibitory effect in the brain-stem on undamaged side are postulated. These two cases suggest that hemitonic seizures include heterogeneous characteristics of partial and generalized epilepsy. In patients with diffuse brain damage, such as those with infantile hemiplegia, seizures appear to exhibit complicated patterns, with characteristics of partial and generalized seizures. There is a gradiation of expression of unilateral seizure, range from clearly partial seizures to those with mostly features of generalized seizures, except for an asymmetry of motor expression.
Hemiconvulsions-hemiplegia-epilepsy syndrome (HH/HHE) is a rare epileptic syndrome consisting of a prolonged unilateral convulsion producing a persisting hemiplegia, sometimes followed by epilepsy. We report on a 13-month-old male who presented with febrile left-sided HH syndrome with right hemispheric unilateral cytotoxic oedema followed by hemispheric atrophy on magnetic resonance imaging (MRI). Six months later the child progressively developed refractory focal epilepsy, including right hemiclonic seizures, and nearly continuous left frontal rhythmic spikes, suggesting the presence of a focal cortical dysplasia (FCD). A repeat MRI at 2 years of age showed left frontal FCD. This unusual case of dual pathology--right HH syndrome and left FCD--suggests that some other factor than the malformation determined the prolonged status and brain atrophy. The kinetics of regional cortical maturation could explain this unusual condition.
The level of agreement between clinicians' ratings of gait patterns in children with spastic hemiplegia was investigated using the Winters, Gage, and Hicks (WGH) classification scale. Sixteen clinicians (nine physiotherapists, seven orthopaedic surgeons) from six gait laboratories in five different countries rated gait patterns in 34 children with spastic hemiplegia (22 males, 12 females; mean age 10y [SD 3y], range 6-18y). Archived three-dimensional gait reports (kinematic and video data) and videos (video clips only) were rated for each child. Agreement between clinicians was substantial for reports (weighted kappa [wkappa] 0.77, range 0.62-0.89). Agreement was lower for videos (wkappa 0.63, range 0.39-0.97) and for clinicians' agreement between reports and corresponding videos (wkappa 0.62, range 0.47-0.76). Exact agreement was unacceptable for some gait patterns using reports (mean 65%, range 32-74%) and videos (mean 53%, range 35-94%). Not all gait patterns could be rated using the original WGH categories. It was concluded that: (1) agreement is acceptable using the WGH tool; (2) kinematic data from 3D instrumented gait analysis and video should be used together when using the WGH scale; and (3) further refinement of this classification is required.
INTRODUCTION: Alternating hemiplegia of childhood (AHC) is a rare disorder mainly characterized by attacks of hemiplegia and mental retardation. It has been often associated with migraine. The CACNA1A gene on chromosome 19 is involved in familial hemiplegic migraine and other episodic cerebral disorders, but also with progressive neuronal damage. METHODS: We performed mutation analysis in this gene in four AHC patients, using single strand conformation polymorphism analysis. RESULTS: We found nine polymorphisms, but no mutations in any of the 47 exons. CONCLUSIONS: Other cerebral ion channel genes remain candidate genes for AHC.
This paper lays out the service and cost consequences of supporting a nationally representative sample of young adults with hemiplegic cerebral palsy. The total costs burden and the additional costs that accrue to the public and independent sector due to their disabilities are calculated. Most of the young adults live at home. Many lead "ordinary" lives and attend further or higher education establishments or go to work. A small number of study members, however, make intensive use of expensive, often segregated facilities such as residential homes or schools and make considerable use of other support services. In total, this group of 81 people cost just over 1 million pounds to support during the year prior to interview, 43% of which was related to their impairments. When the sample is divided into two groups, people with a combination of associated conditions are found to have hemiplegia-associated costs almost 50 times greater than those with simple hemiplegia.
OBJECTIVE: To determine whether percutaneous (intramuscular) neuromuscular electric stimulation (perc-NMES) is less painful than transcutaneous neuromuscular electric stimulation (trans-NMES) for treating shoulder subluxation in hemiplegia. DESIGN: Double-blind, crossover trial. SETTING: University-affiliated tertiary care hospital. PARTICIPANTS: A convenience sample of 10 hemiplegic subjects with at least 1 fingerbreadth of glenohumeral subluxation. INTERVENTIONS: All subjects received 3 randomly ordered pairs of perc-NMES and trans-NMES to the supraspinatus and posterior deltoid muscles of the subluxated shoulder. Both types of stimulation were optimized to provide full joint reduction with minimal discomfort. MAIN OUTCOME MEASURES: Pain was assessed after each stimulation with a 10-cm visual analog scale (VAS) and the McGill Pain Questionnaire, using the Pain Rating Index (PRI) scoring method. Subjects were asked which type of stimulation they would prefer for 6 weeks of treatment. Wilcoxon's signed-rank test was used to compare median differences in VAS and PRI between perc-NMES and trans-NMES. RESULTS: Median VAS scores for perc-NMES and trans-NMES were 1 and 5.7, respectively (p = .007). Median PRI scores for perc-NMES and trans-NMES were 7 and 19.5, respectively (p = .018). Nine of the 10 subjects preferred perc-NMES to trans-NMES for treatment. CONCLUSION: Data suggest that perc-NMES is less painful than trans-NMES in the treatment of shoulder subluxation in hemiplegia.
OBJECTIVE: To examine the effect of cordopexy, laryngoplasty, and cordopexy combined with a modified laryngoplasty on airway mechanics. STUDY DESIGN: Experimental airway mechanics were determined by subjecting equine cadaveric larynges to airflows similar to inspiratory airflow of exercising horses. ANIMALS OR SAMPLE POPULATION: Twenty equine larynges. METHODS: Using cadaveric larynges, we developed and tested a new technique of arytenoid cartilage abduction. All larynges had the right arytenoid cartilage abducted to mimic the degree of arytenoid abduction that occurs at maximal exertion in live horses. Three surgical techniques were used to stabilize the left arytenoid cartilage of treated larynges; the left arytenoid cartilage was not stabilized in control larynges. Technique 1: Cordopexy--a suture was placed between the vocal ligament and the lamina of the thyroid cartilage. Technique 2: Standard laryngoplasty--a suture was placed between the muscular process of the arytenoid cartilage and the caudomedial aspect of the cricoid cartilage. Technique 3: Cordopexy plus modified laryngoplasty--the cordopexy suture was placed with a second suture between the horizontal ridge rostral to the muscular process of the left arytenoid cartilage and the lamina of the thyroid cartilage. Translaryngeal impedances (TI) were determined for each surgical technique by subjecting the larynges to increasing airflows and measuring the translaryngeal pressure differences. The arytenoid right to left angle quotient (RLQ) and the glottic cross-sectional area (CSA) were also measured. RESULTS: At maximal airflow, the adjusted means for the arytenoid RLQ and the TI for the cordopexy plus modified laryngoplasty (1.48 +/- 0.04, 0.69 +/- 0.05 cm H2O/L/s) and the standard laryngoplasty (1.39 +/- 0.04, 0.78 cm H2O/L/s) were different (P < .05) from values obtained after cordopexy alone (2.74 +/- 0.37, 1.76 +/- 0.48 cm H2O/L/s) or in control larynges (3.66 +/- 0.54, 4.16 +/- 0.96 cm H2O/L/s). Overall, a cordopexy plus modified laryngoplasty (9.69 cm2), a standard laryngoplasty (9.34 cm2), and a cordopexy alone (9 cm2) resulted in an increased glottic CSA greater than that for control larynges (6.94 cm2; P = .0001). CONCLUSIONS: Cordopexy alone did not improve airflow in a left laryngeal hemiplegic model. Cordopexy plus modified laryngoplasty was as efficacious as the standard laryngoplasty in alleviating the effects of left laryngeal hemiplegia on TI, glottic CSA, and arytenoid RLQ. CLINICAL RELEVANCE: Fixation of the vocal cord (cordopexy) in addition to a laryngoplasty procedure may prove useful in the surgical treatment of equine laryngeal hemiplegia.
Hemiconvulsions-hemiplegia (HH) syndrome is an acquired condition in which hemiplegia develops after a preceding febrile unilateral status epilepticus in a previously healthy child. Although viral encephalitis or vascular diseases may be the underlying etiology, the pathogenesis remains unknown in the majority of cases. We measured both plasma and cerebrospinal fluid cytokine levels in a girl with HH syndrome, and found elevated plasma concentrations of soluble interleukin-2 receptor and tumor necrosis factor-alpha, and a slightly increased plasma level of interleukin-6. Furthermore, she had a high serum concentration of soluble E-selectin, which is a marker of inflammatory endothelial activation. These findings suggest that proinflammatory cytokine-induced cerebrovascular endothelial injury could play a role in the pathogenesis of HH syndrome.
Alternating hemiplegia of childhood (AHC) is a rare disorder mainly characterised by attacks of hemiplegia and mental retardation. AHC has often been associated with migraine. Previously, we have excluded the involvement of the familial hemiplegic migraine (FHM) CACNA1A gene in four patients with AHC. A second gene for FHM was discovered recently: the ATP1A2 gene on chromosome 1q23, coding for the alpha 2 subunit of Na+,K+-ATPase. We performed a mutation analysis of the ATP1A2 gene in six patients, using direct sequencing, but found no mutations in any of the 23 exons. Other cerebral ion channel genes remain candidate genes for AHC.