ISSN - 0973-0958

Pediatric Oncall Journal

Acute Oromandibular Dystonia

Inês Afonso Belo1, Ana Rita Ramos2, João Nuno Franco2.
1Department of Pediatrics, Centro Hospitalar de Leiria, Portugal,
2Department of Pediatrics, Hospital Garcia de Orta, Portugal.

Inês Belo, Hospital de Santo André - Rua das Olhalvas, Leiria, Portugal
Postal address: 2410-197 Leiria, Portugal
Metoclopramide, acute dystonia, side effects

A previously healthy 14-year-old girl was brought to the Emergency Department by the paramedics due to a sudden onset of painful involuntary oromandibular muscle contraction and swollen lips. She also referred sporadic vomiting, once or twice a day, and abdominal pain for three days. On physical examination, she exhibited an oromandibular muscle contraction, deviating to the right side and angioedema of the lips (Figure 1). Her physical and neurological examination was otherwise unremarkable. She had no sialorrhea, no dyspneia, no rash and no tongue swelling. She weighted 41 Kg (between the 10th and 25th percentiles) and her height was 156 cm (25th percentile).
On the previous day, her primary care physician prescribed metoclopramide, 10 mg three times a day (0.7 mg/kg/day) for the vomiting, in the context of a probable gastrointestinal infection.
Due to the hypothesis of an extrapyramidal effect of metoclopramide, intravenous biperiden was administered and there was a resolution of the dystonia and the edema within a few minutes (Figure 2).

Figure 1. Adolescent at admission showing an orofacial dystonia.
<b>Figure 1.</b> Adolescent at admission showing an orofacial dystonia.

Figure 2. Adolescent after biperiden if you agree.
<b>Figure 2.</b> Adolescent after biperiden if you agree.

Figure 1. Adolescent at admission showing an orofacial dystonia
  Acute Oromandibular Dystonia
What is the diagnosis?

Metoclopramide is a dopamine receptor antagonist. It is used in various gastrointestinal disorders such as nausea, vomiting and gastroparesis.1 Although metoclopramide is one of the most frequently prescribed anti-emetics, the side effects have been reported in up to 20%2 of patients and, in children, extrapyramidal reactions are the most common ones, with an incidence of 9%.3 Acute dystonia typically occurs within 24 to 48 hours of initiating treatment and includes opisthotonus, torticollis, oculogyric crises and tonic muscular contractions.4,5 Pharyngeal muscle spasms or laryngospasm are rare but potentially life-threatening.6,7 Neurological adverse effects of metoclopramide are idiosyncratic and are not dose dependent, although it is more frequent with higher doses.8 Therefore, if metoclopramide must be given, doses should not exceed 0.5 mg/kg/day.8 These reactions are usually self-limited.
Acute dystonic reactions may represent a challenging diagnosis in the emergency room, as there is a high probability of misdiagnosis with other conditions like tetanus, partial seizures, encephalitis, meningitis or allergic reactions.5,9
In our case, besides the dystonia, the adolescent also presented angioedema that resolved after treatment. This finding is uncommon but has yet been described either with metoclopramide or other drugs such as haloperidol.10,11 When appropriate, other antiemetics such as ondansetron should be used, preferably as a single dose, thus reducing the risk of extrapyramidal reactions.
Compliance with ethical standards
Funding:  None  
Conflict of Interest:  None

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Cite this article as:
Belo I A, Ramos A R, Franco J N. Acute Oromandibular Dystonia. Pediatr Oncall J. 2023;20: 62-63. doi: 10.7199/ped.oncall.2023.20
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