Levetiracetam therapy for treatment of choreoathetosis in dyskinetic cerebral palsy

Abstract

Dyskinetic cerebral palsy (CP) is a movement disorder that is difficult to treat and which causes major disability. We report on two female patients (aged 5y and 8y) who experienced severe perinatal asphyxia and developed dyskinetic CP, clinically characterized by choreoathetosis. Neuropsychological testing of these children showed a low average developmental quotient and no attentional deficit. Monotherapy with levetiracetam was initiated to improve balance control and fine motor skills. Treatment was evaluated by use of video and the Visual Analog Scale. In both children an impressive improvement of balance control and fine motor skills was observed. No side effect occurred. Furthermore, both patients showed more interest and pleasure during activities according to their parents. In a recent multidisciplinary evaluation of the initiated therapy, the parents, the therapist, and the rehabilitation doctor all confirmed that the effect initially observed was still present at 14 and 26 months later. To our knowledge, this report on two patients with dyskinetic CP is the first suggesting that levetiracetam may offer an alternative to the standard therapy of involuntary, uncontrolled movements in this group of patients.Cerebral palsy (CP) is a non-progressive disorder of posture or movement caused by a lesion of the developing brain. The Surveillance of Cerebral Palsy in Europe (SCPE) has classified CP into three main groups (spastic, dyskinetic, and ataxic), which are based on clear neurological signs indicating pathology in the cerebral motor systems. Dyskinetic CP presents involuntary, uncontrolled, recurring, and, occasionally, stereotyped movements. SCPE uses dystonic and choreoathetotic subtypes for subgrouping dsykinetic CP.1 The pathophysiology of dyskinesia in CP is not fully elucidated, but existing evidence suggests that disorders of the basal ganglia play a role in this entity. The symptomatic treatment of this movement disorder is still very difficult. In a retrospective survey of 22 children with extrapyramidal CP, only a minority showed a functional improvement using trihexyphenidyl, which is a centrally active muscarinic antagonist. 2Several anticonvulsants seem to be efficacious for the treatment of (paroxysmal) involuntary movement disorders. On the basis of a recent report about levetiracetam efficacy in the treatment of movement disorders, we started levetiracetam in two patients with dyskinetic CP unresponsive to conventional drugs mostly used in such cases. 3To our knowledge, this report on two patients with dyskinetic CP, is the first suggesting that levetiracetam may offer an alternative to standard therapy of involuntary, uncontrolled movements in this group of patients. METHODInformed consent of the parents was obtained for both patients. Levetiracetam was only started when the other available drugs usually prescribed failed. Baclofen (GABAagonist) and artane (Muscarine-antagonist) were tried but ª

Treatment of Involuntary Movements with Carbamazepine in Dystonic-Choreoathetoid Cerebral Palsy

“…This is a first demonstration of effective treatment of dystonic‐choreoathetoid CP by CBZ. While Vles and colleagues have reported significant improvement of choreoathetoid movements with levetiracetam in two children with CP, in our patient switching to levetiracetam resulted in worsening of his involuntary movements. Numerous reasons could account for the inefficacy of levetiracetam and the effectiveness of CBZ, including the differing mechanism of action of the drugs, potential underdosing of levetiracetam, the age of the patients (pediatric vs. adult) and the potential anti‐anxiety or mood stabilizing effect of CBZ which could indirectly contribute to the improvement of the involuntary movements; Clinician should consider use of CBZ in CP patients with disabling and bothersome involuntary movements, before pursuing an expensive procedure such as GPi‐DBS.…”

“…CP remains a leading cause of disability in children affecting 2 to 3 per 1,000 live births, with ~10% to 15% of these children having dystonic‐choreoathetoid CP typically with little or no cognitive impairment ,. Medical treatment of dystonic‐choreoathetoid CP remains less than satisfactory, but there are reports of mild improvement with trihexyphenidyl and a report of 2 children who improved with levetiracetam . Many patients are currently undergoing bilateral globus pallidus interna (GPi‐DBS) stimulation for symptomatic improvement .…”

The Dyskinesia Impairment Scale: a new instrument to measure dystonia and choreoathetosis in dyskinetic cerebral palsy

“…Specific assessment of dystonia has mostly relied on the Barry-Albright Dystonia Scale (BADS). 20 Operationally, the BADS has become a criterion standard for scoring dystonia in CP, but several studies [10][11][12][13][14][15][16][17] have reported the difficulty of measuring dystonia reliably and ⁄ or questioned the sensitivity of the BADS.…”

Use of Antiepileptic Drugs for Hyperkinetic Movement Disorders

“…However, neuronal ion channelopathies have been documented in several paroxysmal movement disorders such as episodic ataxia type 1 (KCNA1) and type 2 (CANCA1) [12, 36, 49, 50] and in paroxysmal neurological disorders without associated movement disorders including familial hemiplegic migraine [51-54] and some forms of familial epilepsy [48, 55, 56]. …”

“…The newer AEDs, may represent an alternative in the treatment of paroxysmal kinesiogenic choreoathetosis; in fact, gabapentin and levetiracetam have been reported to be useful in the management of patient with hemichorea/hemiballism [78, 79], while topiramate is reported to improve both vascular hemichorea/hemiballism [80, 81] and vascular generalized chorea [55]. Moreover, recently Vlas and co-workers [50] described the efficacy of levetiracetam (start dose of 2.5mg⁄kg⁄day, titrated to a final dose of 10mg⁄kg⁄day) to reduce choreoathetosis in two patients with dyskinetic cerebral palsy, without the development of side effects.…”