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PubMed1992

Alternating hemiplegia in childhood: 23 cases in Japan.

Alternating hemiplegia in childhood (AHC) has clinically characteristic features which are easily defined and recognizable. Laboratory investigations were basically normal although they were extensively examined during and between attacks. There is still much debate about its etiology, particularly its relation to migraine or epilepsy. Clinical characteristics of identified 23 AHC in Japanese were presented, which were obtained from a large Japanese cooperative study in 1988. AHC may prove not to be as rare as has been thought.

Child↗
PubMed1997

Alternating hemiplegia of childhood: report of a case having a long history.

We examined a patient with alternating hemiplegia of childhood (AHC) who had over a 23-year history of AHC to investigate the origin of the neurological deterioration with increasing age. Hemiplegic attacks had occurred consistently at a frequency of a few per week since infancy, and he first experienced attacks of cerebellar ataxia at the age of 23 years. Intellectual impairment, dysarthria, dystonic posturing, and a wide-based gait had been slowly progressive, but they had been stable since he turned twenty. The electromyographic response to transcranial magnetic stimulation was normal between attacks and showed reversible alteration during an attack. MRI revealed slight dilatation of the lateral ventricles, and MR angiography showed normal cerebral blood flow. Proton MR spectroscopy between attacks showed normal peak area ratios for N-acetyl groups, choline-containing compounds, and creatine and phosphocreatine, and it also demonstrated no lactic peak. 123I-IMP SPECT between attacks demonstrated diffuse cerebral hypoperfusion despite no evidence of ischemic change in the above MR study. These results suggest that the slowly progressive neurological deficits are due to the primary underlying pathology rather than the secondary neuronal loss as a result of frequent ischemic attacks.

Adult↗
PubMed1993

[Post-traumatic hemiplegia in a patient with fibromuscular dysplasia of the carotid artery].

A 44-year-old patient, without remarkable medical history, was admitted with a head trauma with initial loss of consciousness and a thoracic trauma. The initial treatment included the insertion of a chest drain for evacuation of a pneumothorax and intrapleural analgesia with bupivacaine. The day after admission, the patient experienced a generalized epileptic crisis, without prodomes. Later, a left proportional hemiplegia with aphasia was recognized. The CT scan obtained immediately after the crisis, as well as the carotid Doppler ultrasonography and echocardiography were normal. The bilateral carotid angiography showed an image of fibromuscular dysplasia of the extracranial segment of the right internal carotid artery. The migration of a carotid thrombus initiated by the trauma was hypothetized. A treatment with a platelet aggregation inhibiting drug was started and associated 20 days later with low molecular weight heparin. The patient recovered a normal motility within 10 days; only the aphasia remained. Trauma of the carotid artery is not a frequent cause of cerebrovascular accident. The occurrence of the latter is favoured by a pre-existing lesion of this artery. This case demonstrates that in a trauma patient not all central nervous system manifestations are initiated by a head trauma.

Adult↗
PubMed2003

Functional reorganization of brain in children affected with congenital hemiplegia: fMRI study.

Using functional magnetic resonance imaging, the brain activation related to unilateral sequential finger-to-thumb opposition was studied in six children with a right congenital hemiplegia of cortical origin. They were compared to six age-matched controls. In the control group, movements with either hand asymmetrically activated the sensorimotor cortex and premotor areas in both cerebral hemispheres with a typical contralateral predominance. By contrast, paretic finger movements activated both hemispheres in the hemiplegic patients, with a strong ipsilateral predominance favoring the undamaged hemisphere. The activation induced by nonparetic finger movements was restricted to the contralateral undamaged hemisphere. Furthermore, the level of activation in the undamaged cortex was partly related to residual finger dexterity, according to covariance analysis. These activation patterns indicate an adaptive reorganization of the cortical motor networks in this group of patients, with a prominent involvement of the undamaged hemisphere in the control of finger movements with either hand.

Adolescent↗
PubMed2003

Long-latency motor evoked potentials in congenital hemiplegia.

OBJECTIVE: To investigate long-latency motor evoked potentials (MEPs) elicited by transcranial magnetic stimulation in congenital hemiplegia (CH) and to seek for correlation with paretic hand movement deficits. METHODS: MEPs were recorded from the first dorsal interosseous of both hands in 12 CH patients and 12 age-matched controls; dexterity and upper limb function were quantitatively assessed in both groups. RESULTS: In CH patients, long-latency MEPs, occurring much later than the commonly reported MEPs, were frequently observed in the paretic and non-paretic hands. Four distinct groups of long-latency MEPs were found, each cluster being identified by its mean latency, namely 35, 85, 160 and 225 ms. The residual dexterity of the paretic hand was correlated with the presence of contralateral MEPs with a 20 and 225 ms latency and was negatively correlated with ipsilateral MEPs, irrespective of their latency. In controls, only few MEPs with a latency of 225 ms were found in 4 out of 12 subjects. CONCLUSIONS: The pattern of MEPs found in CH patients differs dramatically from that reported in adult stroke patients, suggesting that long-latency MEPs are a rather distinctive consequence of early corticospinal lesions. The hypothesis that a given cluster of long-latency MEPs is mediated by a particular pathway appears very unlikely. Rather, we suggest that an exacerbation of cortical and/or spinal excitability is at the origin of these long-latency MEPs.

Adolescent↗
PubMed2001

Comparison of three ankle-foot orthosis configurations for children with spastic hemiplegia.

The purpose of this study was to examine the effectiveness of the hinged ankle-foot orthosis (HAFO), posterior leaf spring (PLS), and solid ankle-foot orthosis (SAFO), in preventing contracture, improving efficiency of gait, and enhancing performance of functional motor skills in 30 children (21 male, 9 female; mean age 9 years 4 months; age range 4 to 18 years,) with spastic hemiplegia. Following a 3-month baseline period of no ankle-foot orthosis (AFO) use, each AFO was worn for 3 months after which ankle range of motion, gait analysis, energy consumption, and functional motor skills were assessed. The HAFO and PLS increased passive ankle dorsiflexion and normalization of ankle rocker function during gait. Normalization of knee motion in stance was dependent upon the knee abnormality present and AFO configuration. The HAFO was the most effective in controlling knee hyperextension in stance, while PLS was the most effective in promoting knee extension in children with >10 degree knee flexion in stance. Energy efficiency was improved in 21 of the children, with 13 of these children demonstrating the greatest improvement in HAFO and PLS. Improvements in functional mobility were greatest in the HAFO and PLS.

Adolescent↗
PubMed2002

Effect of a rigid ankle-foot orthosis on hamstring length in children with hemiplegia.

Eighteen children with hemiplegia, mean age 8 years 5 months, underwent gait analysis and musculoskeletal modelling using specially designed software. The maximum lengths of the hamstrings were determined for each child walking in and out of an ankle-foot orthosis (AFO). The muscles were deemed to be short if shorter than the normal average -1SD. In bare feet 8 participants had short medial hamstrings with a higher proportion of these in the less involved individuals. All participants showed an increase in maximum hamstring length when wearing an AFO. In all but one child this was sufficient to restore hamstring length to within normal limits. These finding suggest that hamstring pathology in hemiplegic gait is usually secondary to more distal lower limb pathology.

Ankle Joint↗
PubMed2002

Comparing tests of tactile sensibility: aspects relevant to testing children with spastic hemiplegia.

Children with hemiplegic cerebral palsy (OP) commonly exhibit tactile sensory deficiencies in their hands in addition to their motor problems. The aim of this study was to compare and evaluate the usefulness of some common tests of tactile sensibility for use with children with hemiplegic CP. Twenty-five children with hemiplegia aged between 5 and 18 years, and 19 control individuals participated. All children were examined with Semmes-Weinstein monofilaments, two-point discrimination (2PD), stereognosis of familiar objects, stereognosis of forms, and functional sensibility. Dexterity, spasticity, and bimanual task performance were also assessed. Results from the different sensory tests deviated greatly. We found three tests to be useful: 2PD of 3 mm spacing, which was the most sensitive test, stereognosis of familiar objects, and functional sensibility assessed through the Pick-up test (comparing performance with and without the influence of vision). Stereognosis of forms and threshold values of touch (Semmes-Weinstein monofilaments) are seemingly less useful tests for children with CNS impairments. Deficient sensibility was strongly related to dexterity. Aspects concerning the testing methodology are discussed.

Adolescent↗
PubMed2005

Muscle force generation and force control of finger movements in children with spastic hemiplegia during isometric tasks.

Force control ability was investigated in 10 males and 10 females, between 5 and 15 years old with spastic hemiplegia (mild and moderate hand dysfunction), and an aged-matched control group (eight males, 12 females). An isometric force production task at five different levels of maximum voluntary contraction (MVC) was performed. Results showed that MVC generated with the affected hand (AH) was only one-third of that generated by the non-affected hand (NAH; p < 0.001), time to peak was almost twofold at the highest force level (p < 0.001), and the coefficient of variation was twice as high (p < 0.001). Results for the NAH did not differ from those of the control children. Correlations between clinical and experimental variables were significant for the relation between Ashworth score for elbow flexors, MVC and variability at the highest force level. In conclusion, the findings for the AH suggests that strength training should be considered for agonist spastic muscles.

Adolescent↗
PubMed1996

Reciprocal inhibition in hemiplegia: correlation with clinical features and recovery.

BACKGROUND: Previous reports have described changes in reciprocal Ia inhibition in hemiplegic patients, but correlations between the amount of Ia inhibition and the clinical deficits have not been well established. METHODS: We studied reciprocal inhibition between ankle flexors (tibialis anterior) and extensors (soleus) in 16 hemiplegic patients at various stages following a stroke and in 26 control subjects. The amount of disynaptic Ia inhibition was determined from the short latency suppression of the soleus or tibialis anterior H-reflexes by conditioning stimulation of the antagonistic muscle nerves. RESULTS: Disynaptic Ia inhibition from peroneal nerve afferents to soleus motoneurones was increased in patients who showed good recovery of function with mild spasticity. However, it was not changed, or even sometimes diminished, in patients who made a poor recovery and had more marked extensor spasticity. In patients where serial recordings were obtained there was an increase in Ia inhibition during the recovery period following stroke. Ia inhibition to the tibialis anterior motoneurones tended to be greater in the poor recovery patients with marked spasticity than in the good recovery patients. The late (D1) inhibition, presumably due to presynaptic inhibition, was decreased in the patients, although consistent correlations between the amount of this inhibition and the clinical features were not clearly demonstrated. CONCLUSIONS: Changes in excitability of Ia inhibitory pathways can be correlated with some of the clinical features seen in hemiplegia. Increased Ia inhibition of soleus motoneurones during recovery may be a mechanism to compensate for loss of descending motor commands.

Adult↗
PubMed1995

Evidence of altered dominance in children with congenital spastic hemiplegia.

The study aimed at investigating lateralization effects and signs of transfer and crowding in children with congenital lateralized brain damage with the aid of a dichotic listening test, a chimeric test, and verbal and nonverbal neuropsychological tests. Thirty-three children with spastic hemiplegia and 86 control children (age 5.0-12.0 yr) were assessed. Children with left-hemisphere damage (n = 17) were found to have a pathological left-ear advantage for verbal material, and children with right-hemisphere damage (n = 16) were found to have a pathological right visual half-field advantage for visual material. Children with left-hemisphere damage and a left-ear advantage on the dichotic test were also found to have a right visual half-field advantage on the chimeric test, which was regarded as a sign of reversed dominance. No verbal or nonverbal differences emerged between the left-hemisphere and the right-hemisphere damage groups, nor did differences emerge when the children were reclassified by considering children with left hemisphere damage and signs of reversed dominance as having damage to the nondominant hemisphere. It was concluded that although lateralized brain damage may alter the dominance for verbal and visual functions, there is still considerable inter-individual variability with respect to inter- and intrahemispheric neural adjustment to damage. The dichotic and the chimeric tests did not indicate the presence of brain damage accurately, but they indicated the lateralization of damage in children with stated abnormality with a high degree (91.3%) of accuracy.

Attention↗
PubMed1998

Hemiplegia following general anaesthesia: an unusual presentation of migraine.

We report the case of a 33-year-old woman who developed a dense hemiplegia immediately after an uncomplicated general anaesthetic for diagnostic laparoscopy. She had a history of recurrent hemiplegic migraine with a strong family history. Her migraine was normally associated with visual disturbances and a unilateral headache followed by a left-sided weakness such that she was unable to walk. Symptoms would last up to 24 h. Her post-operative state was atypical of her normal migraine, in that she had no headache or visual disturbance and initially all four limbs were affected.

Adult↗
PubMed2001

Classification of gait patterns in spastic hemiplegia and spastic diplegia: a basis for a management algorithm.

Classifications of gait and postural patterns in spastic hemiplegia and spastic diplegiía are presented, based on the work of previous authors. The classifications are used as a biomechanical basis, linking spasticity, musculoskeletal pathology in the lower limbs, and the appropriate intervention strategies. The choice of target muscles for spasticity management, the muscle contractures requiring lengthening and the choice of orthotics are then linked to the underlying gait pattern.

Algorithms↗
PubMed2001

Botulinum toxin type A injections in the spastic upper extremity of children with hemiplegia: child characteristics that predict a positive outcome.

Evidence is increasing to suggest that botulinum toxin type A (BTX-A) plays a role in the management of upper extremity spasticity in the paediatric population. However, little information is available on the clinical characteristics of the child that predict a response to this intervention. Our research group previously published a randomized controlled trial demonstrating that BTX-A injection improves function of the upper extremity of children with spastic hemiplegia. In the present paper, we evaluate the child characteristics that predict a positive response to the BTX-A injections in the randomized treatment group. The treatment group was divided into positive functional responders and nonresponders using a cut score of a change of 10 points on the Quality of Upper Extremity Skills Test (QUEST). A two-way analysis of variance procedure was done comparing the following baseline characteristics: function as scored on the QUEST and the Pediatric Evaluation of Disability Inventory (PEDI) self-care domain, grip strength, upper extremity spasticity and age. Grip strength was significantly higher in responders with a P-value of 0.001. Young age approached significance with a P-value of 0.05. Correlation of change scores on the QUEST with baseline characteristics in the treatment group yielded similar results. BTX-A causes a reduction in spasticity and strength; we postulate that if the hand is weak initially, BTX-A can decrease hand function. Two case reports are presented that highlight the importance of grip strength and age.

Activities of Daily Living↗
PubMed2002

Prediction of reflex sympathetic dystrophy in hemiplegia by evaluation of hand edema.

OBJECTIVE: To determine the predictive value of measurements of hand edema for the development of reflex sympathetic dystrophy (RSD). DESIGN: Cohort study. SETTING: Departments of rehabilitation medicine in 3 general hospitals and 1 rehabilitation hospital in Japan. PARTICIPANTS: Thirty-four stroke patients. INTERVENTIONS: Not applicable. MAIN OUTCOME MEASURES: Measurement of the circumference of the middle finger was used to evaluate hand edema. The degree of hand edema was expressed by the ratio of circumference of the middle finger (RCMF) in the affected side to that in the uninvolved extremity. RESULTS: Eight of 34 patients developed clinical RSD from 2 to 4 months after stroke. Hand edema showed a significant relationship to the development of RSD (ie, the patients who had an RCMF of above 1.06 at 4 weeks poststroke had significantly higher incidence of RSD than those with a lower RCMF; P=.0127). CONCLUSION: It is possible to predict the development of RSD in hemiplegia by measuring hand edema 4 weeks poststroke.

Adult↗
PubMed2000

Successful trial of amantadine hydrochloride for two patients with alternating hemiplegia of childhood.

We report here the efficacy of amantadine hydrochloride for two patients with alternating hemiplegia of childhood (AHC) that did not respond to flunarizine. Amantadine was administered to one patient at age one year and seven months and to the other at age 25 years. The frequencies and duration of the hemiplegic attacks significantly improved in both patients. However, the attacks gradually returned to the previous level after a significant reduction in seizures for three years in the younger patient with ongoing AHC. Our therapeutic results further support the hypothesis that glutamate and NMDA receptors are involved in inducing alternating hemiplegic attacks, because amantadine as well as its derivative, memantine, are clinically available non-competitive N-methyl-D-aspartate (NMDA) receptor antagonists, with neuroprotective effects. Amantadine is worth trying when treating patients with AHC as a first trial or a substitute for flunarizine once the latter agent looses effect.

Adult↗
PubMed1992

["Palatolaryngeal hemiplegia" in transient brain stem ischemia--a contribution to neurogenic dysphagia].

A 65-year old man suffering from dysphagia with aspiration was examined. ENT examination showed a Horner syndrome and cranial nerve palsy with paralysis of the soft palate and one vocal cord (palatolaryngeal hemiplegia, Avellis' syndrome). Pharyngeal manometry and videofluoroscopy depicted an asynergic swallowing with cricopharyngeal achalasia. CT scans of mediastinum, head, neck, and skull base showed no signs of abnormality. MR imaging of the brain stem demonstrated an enrichment of contrast medium in the dorsal region of the upper medulla oblongata in the level of the centre of the glossopharyngeal and vagus nerve. This case demonstrates an uncommon cause of dysphagia which was related to transitory brain stem ischaemia. After a period of three weeks the patients' complaints vanished as well as the clinical features. In a follow-up of MR-imaging three months later no focal enhancement of contrast medium was seen confirming the diagnosis of a brain stem ischaemic lesion.

Aged↗
PubMed1987

Flunarizine in alternating hemiplegia in childhood. An international study in 12 children.

Twelve children with alternating hemiplegia were treated with the calcium-entry blocker flunarizine for 4 months. All but one patient responded favourably with a reduction in frequency and/or duration and severity of attacks. Interictal symptoms decreased and mental development improved in several patients. Nine of the patients entered a subsequent double-blind placebo-controlled withdrawal study lasting another 4 months. Relapses were observed in part of the placebo as well as of the flunarizine-treated patients. The reason for this is not clear, since it is unlikely that the favourable response during the initial open-label study would be due to a placebo effect, or that tolerance to the drug had developed. Feed-back from the parents rather suggests that stress and tension, which were known trigger factors in a majority of these patients, played a role when the patient was switched to the double-blind treatment. Although the present study is not fully conclusive, apparently because of the use of an inadequate trial design, flunarizine, the first truly promising drug in this disease, deserves further study. An appeal is made to join another international double-blind study.

Child↗