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

Abstract

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 Facing this type of complex illness, we insist on the importance of interdisciplinary work with the goal of avoiding outdated techniques.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.

Post-Stroke Lingual Dystonia: Clinical Description and Neuroimaging Findings

“…This patient had multiple old ischemic lesions in the bilateral thalami also. Two other chronic cases of post-stroke (left anterior inferior cerebellar infarct and right basal ganglia infarct) oromandibular dystonia with lingual dystonia have been reported (Akin et al, 7 Brissaud et al 8 ). Another case of post-stroke (right frontal hematoma) cranial lingual dystonia was reported, but the onset was not described in the paper (Alarcón et al 9 ).…”

“…We identified 20 cases of post-stroke lingual movement disorders following a PubMed search using the search terms described in Methods. [7][8][9][10][11][12][13][14][15][16][17][18][19][20] Thirteen cases had acute presentation and seven had a chronic presentation. Rippling tongue movements were the most common acute movement disorders described in five cases, followed by myoclonus (n53), dyskinesia (n52), tremor (n52), and dystonia (n51).…”

“…Two chronic cases of oromandibular dystonia with lingual dystonia have also been reported. 7,8 None of our lingual dystonia patients had associated cranial or oromandibular dystonia.…”

“…6 Two other chronic cases of post-stroke oromandibular dystonia with lingual dystonia have been reported. 7,8 Another case of post-stroke cranial lingual dystonia was reported; however, the onset was not described in the paper. 9 Here, we describe a case series of 11 patients who developed lingual dystonia following stroke.…”

Dental appliance therapy in pantothenate kinase‐associated neurodegeneration: Case report

“…Severe protrusive lingual dystonia is characteristic of the condition . This can cause significant distress for both patient and carers and can result in trauma to the lingual tissues with occasional severe consequences …”