Abstract
The following case report outlines the impact of motor tics linked to Tourette’s syndrome on dental development and healing following a dental injury to a maxillary central incisor. Emergency care and splinting of a mobile extruded maxillary left central incisor tooth was carried out at the local dentist on the same day as the dental trauma. A subsequent referral was made to the paediatric dental department for continued mobility of the maxillary central incisor on splint removal approximately 2 weeks later. A clinical and radiographic examination revealed shortened root length and apical root blunting associated with both maxillary anterior teeth. Further questioning revealed the likely cause of this to be related to the clenching and biting oral tics which the patient has experienced over the past 4 years. A removable splint has been fabricated for night-time wear and a mouthgaurd has been recommended for use during contact sports. Regular reviews will be conducted using a shared care approach between the patient’s local dentist and the paediatric dental department.
Keywords: Dentistry and oral medicine, Neurology
Background
A pre-teen boy, attended the paediatric dental traumatology clinic having sustained dental trauma to the maxillary left central permanent incisor (UL1). The patient reported falling face first when jumping on his bed at home a month before attendance to the clinic. This injury was described as an extrusive luxation injury with the patient’s mother reporting that the tooth looked ‘longer and wobblier’. Emergency treatment was conducted at the local dentist involving placement of a flexible splint for a period of 2 weeks. The patient was referred into the secondary care setting regarding concerns surrounding the continued mobility of the traumatised tooth.
The parent reported a medical history of attention deficit hyperactivity disorder and Tourette’s syndrome (TS), specifically complex motor tics. Further questioning revealed a constant change in oral motor tics with a history of clenching and biting tics occurring over the course of a 4-year period, commencing at aged 7. The parents described the tics as having an episodic onset, lasting between 1 and 2 weeks and occurring monthly. During tic periods, mandibular protrusion was noted with only the maxillary and mandibular incisors contacting and resulting in repetitive blunt trauma to the maxillary incisors during the root development period.
Case presentation
A thorough history and examination including trauma investigations revealed grade 2 mobility associated with the UL1. The tooth, however, was otherwise clinically sound and well aligned, with no signs of soft tissue pathology, discolouration or pain on palpation. The UL1 also responded normally to sensibility tests. The orthodontic examination reported mild anterior segment crowding and class 1 incisor and molar relationships. The overbite and overjet were within normal remits (see figure 1).
Figure 1.
(A–C) A set of images taken on presentation 1-month postdental trauma.
Investigations
In light of the mobility associated with the UL1, two intraoral radiographs were taken using the parallax technique in order to rule out root fracture, and to assess both periapical pathology and root morphology injury. The radiographs did, however, show shortened root length with blunted root apices of both the UR1 and UL1 (see figure 2).
Figure 2.
(A, B) Upper standard occlusal radiograph and periapical radiograph to assessed the maxillary central incisors following a traumatic dental injury.
Treatment
After a lengthy discussion regarding the oral findings, several treatment options were discussed with the parent and patient. The patient was advised to avoid biting into hard foods with the anterior dentition. Through the social history, it was identified that the patient was an avid rugby player. Use of a mouthguard for contact sports was reinforced with the importance explained at length. Finally, in conjunction with the history of tics, a removable splint was advised for regular night-time wear with full time use being advised when the oral tics of clenching and biting manifested (see figure 3).
Figure 3.
(A, B) A set of images taken on provision of removable splint.
Outcome and follow-up
The plan regarding the patient’s ongoing management of his maxillary incisors was to ensure a removable splint was provided for daily night time wear (see figure 3), with full-time splint wearing when oral tics manifest (for duration of tic period). He was also advised to use a mouthguard when partaking in contact sport. Regular dental reviews with primary care dental practitioner for monitoring of tooth mobility and vitality was advised. The follow-up care regimen recommended was an initial 3-month review, with 6-monthly reviews thereafter as per the patient’s caries risk assessment and the dental trauma guidelines.
Discussion
TS has been described as an early childhood-onset neurodevelopmental disorder.1 Motor tics are associated hyperkinectic movements that resemble voluntary actions. While they share many neurophysiological characteristics, tics differ in that they are usually proceeded by a sensory premonition or urge. They manifest as an exaggeration of normal movements and for some patients can be uncontrollable.2 The management of tics in TS patients can be challenging which the medical team using behavioural, pharmacological and in some cases, surgical methods.3 Sixty-six per cent of patients with TS present with facial tics as their first symptoms, with reports of self-injurious behaviours occurring in approximately a quarter of this patient group.4 The severity of tics is estimated to peak at approximately 8–12 years of age.5
There has been little published cases of dental manifestations in TS patients. Leksell and Edvardson6 reported a case of a 4-year-old patient with self-injurious behaviour. She presented to a paediatric dentist after self-extraction of her primary teeth, secondary to her oral motor tics. Repetitive grinding of teeth in a monophasic lateral motion resulting in tooth luxation was reported with subsequent oral and facial pain. This is in keeping with our patient’s tic pattern and tooth mobility.
Commencement of root development of the maxillary central permanent incisors occurs at the age of 5.5 years with full formation and apical closure by the age of 10.5 years. Repetitive blunt trauma through regular oral tics during the root development period as described in this case can lead to root blunting and shortening. This was seen radiographically on both the traumatised UL1 and the contralateral central incisor that did not sustain a dental trauma injury. As a result of repetitive blunt trauma, these teeth are more prone to mobility. This case highlights the potential side effects of oral tics on the developing dentition, with non-invasive splint provision by the general dental practitioner being a recommended management strategy during oral tic periods.
Patient’s perspective.
Written by the patient’s mother (names were substituted remain anonymous):
Following my sons incident where he fell off the bed and knocked his tooth, we were referred to the dental hospital. He was seen by a dentist and X-rays were taken. All staff made my son feel comfortable and less nervous. It was discovered that he has shortened roots of his teeth. I was asked if he had previous trauma to his teeth, but he had not. I mentioned that my son has Tourette’s syndrome, mainly motor tics and one of his common tics was chomping his teeth (biting them together sharply). It was established that this could be the trauma to his teeth, the prolonged tics of biting his teeth together.
My son was fitted for a retainer (clear plastic) to wear when he has these tics to try and prevent his teeth from clashing and causing more damage. Finding this out was quite worrying at first as he is adventurous and is into sports and I though he wouldn’t be able to play in his rugby team anymore. However, we came to terms with it and he is living a normal life but just being cautious and makes sure he wears a mouth guard for sports.
Learning points.
While the patient’s initial presentation was regarding a dental trauma injury, subsequent investigations revealed shortened roots. Further questioning revealed oral tics as the probable cause of repetitive traumatic injury affecting the maxillary incisors during its root development period. Awareness of oral tics in patients with neurodevelopmental disorders and the impact on oral health is essential for clinicians, especially paediatric clinicians given the early onset of oral tics and possible self-injurious behaviours.
Early identification, medical liaison and parent support are increasingly important for this cohort of patients.
As paediatric clinicians, we need to maintain a holistic approach to patient management and understand the oral tics patterns to be able to appropriately advise and treat patients.
Footnotes
Contributors: RB: assessment of the patient, requesting special investigations, follow-up care of patient and writing of article. MK and MV: supervision during assessment of patient and supervision of follow-up care. Assistance in article production.
Funding: The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.
Case reports provide a valuable learning resource for the scientific community and can indicate areas of interest for future research. They should not be used in isolation to guide treatment choices or public health policy.
Competing interests: None declared.
Provenance and peer review: Not commissioned; externally peer reviewed.
Ethics statements
Patient consent for publication
Consent obtained from parent(s)/guardian(s).
References
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