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A 12-year-old male with a chronic disruptive behavior disorder presented to the Emergency Department with tongue protrusion, inability to speak, eye rolling, and involuntary body tremors manifesting on the same day. His mother reported that this was the fifth episode in the past month, all of which resolved spontaneously within a few hours. There was no history of fever, trauma, or ingestion of an unknown substance. The boy had been treated with paliperidone 3 mg daily for several years, but the dose had to be increased to 6 mg/day during the previous month.
Physical examination revealed eye, mouth, and tongue dystonia, and increased neck tone with left-sided deviation (Figure 1). No other abnormalities were found on neurologic examination. Vital signs were stable, and the Glasgow Coma Scale score was 15.
A 3-mg intravenous infusion of biperiden was administered with complete resolution of symptoms in five minutes (Figure 2).
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Figures 1 Physical examination at presentation on ED: eye, mouth, and tongue dystonia and increased neck tone with left-sided deviation
Discussion
Extrapyramidal symptoms (EPS) are movement disorders that can be caused by brain damage or induced by drugs.1) They include dystonia, akathisia, and parkinsonism, which occur within a few days of starting or increasing medication, or tardive dyskinesia, which is a late side effect.2,3
EPS are the most common side effect described in patients treated with antipsychotics, especially first-generation antipsychotics.1,3,4) Severe or fatal cases have been reported.1)
Paliperidone is an atypical or second-generation antipsychotic (SGA) approved for the treatment of schizophrenia in adolescents and also used off-label for behavioral disorders in children and adolescents.5 This type of antipsychotic exhibits antagonism to both dopamine D2 and serotonin type 2A receptors.2,3) Drug-induced extrapyramidal syndrome can be explained by the affinity of antipsychotics for D2 receptor blockade in the nigrostriatal tract, which interferes with normal dopamine and acetylcholine neurotransmission.6) Studies have shown that the risk of EPS is higher with typical antipsychotics, but several cases have been described in patients treated with SGA.2,4,6) This type of symptom is usually associated with high doses and can occur when an antipsychotic is initiated or when the dose is increased, regardless of previous treatment duration.3,5
As observed in this case, dystonia may present as spasmodic torticollis and tongue protrusion or as blepharospasm, trunk extension, and forced jaw opening.1-3)
Extrapyramidal side effects in children can be treated with anticholinergic medications such as benztropine, biperiden, or diphenhydramine.1-3 In some cases, an extended treatment period of two to five days should be considered to prevent recurrence of symptoms.3 The antipsychotic agent should be discontinued or, if antipsychotic treatment is required, the dose reduced.1-3
The number of mental disorders in children for whom treatment with SGA is recommended has increased in recent years.3) Despite their weak effect on D2 receptors, with an expected low liability for inducing EPS, recent studies show that the risk of side effects is higher than previously thought.2-4) Therefore, it is essential to use SGA cautiously in children and adolescents and to quantify the risk-benefit ratio.2,4 Parents should be informed about the possibility of extrapyramidal symptoms, and pediatricians should be familiar with the acute presentation of adverse effects of these drugs.2,4
Authorship
Mafalda Moreira - Conceptualization; Investigation; Project administration; Visualization; Writing - original draft; Writing - review & editing
Diana Alba - Validation; Writing - review & editing
Inês Paiva Ferreira - Validation; Writing - review & editing
Cláudia Monteiro - Investigation; Supervision; Validation; Writing - review & editing
