Case Report of a Severe Recurrent Tongue Self-Injury in an Infant With Dystonia

Pediatrics. 2016 Nov;138(5):e20160738. doi: 10.1542/peds.2016-0738. Epub 2016 Oct 6.


Dystonia is characterized by sustained or intermittent muscle contractions causing abnormal, often repetitive movements, postures, or both that are typically patterned, twisting, and sometimes tremulous. It is often initiated or worsened by voluntary action and associated with overflow muscle activation. In this article we report a case of severe oromandibular dystonia, which is a specific form of dystonia characterized by involuntary, action-induced tonic or clonic spasms of the masticatory, lingual, and pharyngeal musculature. Episodes of repeated tongue biting in a 17-month-old girl caused her to stay in the PICU for 4 weeks. These episodes were the consequence of dystonia induced by a perinatal stroke. We highlight the specific dental management that enabled us to treat the child without extractions. Facing this type of complex illness, we insist on the importance of interdisciplinary work with the goal of avoiding outdated techniques. The use of botulinum toxin seemed relevant.

Publication types

  • Case Reports

MeSH terms

  • Adrenergic Uptake Inhibitors / therapeutic use
  • Botulinum Toxins / therapeutic use
  • Dystonic Disorders / complications*
  • Dystonic Disorders / drug therapy
  • Female
  • Humans
  • Infant
  • Mouth Protectors
  • Neuromuscular Blockade
  • Patient Care Team
  • Recurrence
  • Self-Injurious Behavior / complications*
  • Self-Injurious Behavior / therapy
  • Tetrabenazine / therapeutic use
  • Tongue / injuries*
  • Tongue / surgery
  • Tooth Extraction
  • Trismus / complications
  • Trismus / prevention & control*


  • Adrenergic Uptake Inhibitors
  • Botulinum Toxins
  • Tetrabenazine