SEVERE TONGUE INJURY IN AN EPILEPTIC ADOLESCENT: A CASE
REPORT
Daniella Ferraz Cerqueira, DDS
Postgraduate Student, Department of Pediatric Dentistry and Orthodontics, School of
Dentistry, Federal University of Rio de Janeiro, Rio de Janeiro, Brazil
Áurea Simone Barrôso Vieira, DDS
Postgraduate Student, Department of Pediatric Dentistry and Orthodontics, School of
Dentistry, Federal University of Rio de Janeiro, Rio de Janeiro, Brazil
Lucianne Cople Maia, MSD, PhD
Associate Professor, Department of Pediatric Dentistry and Orthodontics, School of
Dentistry, Federal University of Rio de Janeiro, Rio de Janeiro, Brazil
Eric Sweet, MD
Associate Professor, Department of Anatomy, Federal University of Rio de Janeiro, Rio
de Janeiro, Brazil
KEY WORDS: Epilepsy, seizure, adolescent, oral health, injury
CORRESPONDING AUTHOR:
Name: Lucianne Cople Maia
Address: Rua Gastão Gonçalves, 47 / 501 Niterói - Rio de Janeiro, Brazil
Zip Code: 24240-030
Phone number: (55) (21) 2629 3738
E-mail: rorefa@microlink.com.br
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ABSTRACT:
Epilepsy and other neurological disorders can have profound social, physical and
psychological consequences especially, when they begin in childhood. Moreover, seizures
episodes may cause fractures, burns and head or dental/oral injuries. Therefore, this article
presents a case of an adolescent with a severe tongue injury related to refractory epileptic
seizures and outlines the proposed treatment; placement of an upper silicon bite guard,
which, within a few months, successfully promoted the healing of tongue injury.
INTRODUCTION
Epilepsy is defined by the WHO (2001) 1, as a chronic affection of multiple
etiologies, characterized by recurring episodes of paroxysmal brain dysfunction caused by
a sudden, disorderly, and excessive neuronal discharge. The diagnosis of epilepsy is
reserved for those who have recurring seizures and at least two unprovoked ones 1, 2
. The
International League Against Epilepsy (ILAE) 2
has developed a classification of
epilepsies in which the primary distinguishing factor is based on the type of seizures, focal
or generalized. Seizures may vary from the briefest lapses of attention or muscle jerks to
severe and prolonged convulsions. They may also vary in frequency, from less than one a
year to several per day.
In 1998, ILAE 3 revised the seizure classification based exclusively on ictal seizure
semiology produced by epileptic interference of one of the four “spheres”: sensorial,
consciousness, autonomic and motor sphere (Table 1). No EEG findings or other test
results influence the classification.
Epilepsy occurs in both genders and can begin at any age, but is most frequently
diagnosed in infancy, childhood, adolescence or old age 4. Anyone can be affected by
seizures; and in fact; up to 5% of the world's population may have a single seizure at some
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point in their lives 1, 5
. From many studies around the world it has been estimated that the
mean prevalence of active epilepsy is approximately 8.2 per 1,000 of the general
population 1.
The elective treatment is usually the administration of anti-epileptic drugs
(phentoin, carbamezapine, valproic acid, phenobarbital, primidone and others) for the
appropriate type of seizure 1, 5, 6
. However a number of drugs used in seizure control have
implications for oral care or dental treatment (Table 2) 6,7,8,9.
Epilepsy can have profound social, physical and psychological consequences 1
especially when it begins in childhood, because all basic functions such as motor and
cognitive tasks and acquisition of social skills are in developmental process 3. Moreover,
seizures may result in, the most common of which are fractures, burns, head and
dental/oral injury 10,11,12
. According to Andreasen & Andreasen (1994)13, epileptic patients
present risks related to dental trauma and tongue injuries, such as lesions from bitings in
the border of the tongue caused by traumas, which are very common during the seizures.
Therefore, the aim of this article is to present a case of an adolescent with a severe tongue
injury related to recurrent epileptic episodes and describes the proposed treatment.
CASE REPORT
A 16-year-old right-handed boy, accompanied by his mother, arrived at the
Pediatric Dentistry Department of a public University of Rio de Janeiro-Brazil, after
seeking for treatment without success in 2 different public hospitals. His chief-complaint
was a terrible pain in his tongue and the anxiety to resolve the discomfort.
Medical history
During anamneses the mother reported that the patient’s “crises” began at 7 years
of age when he bit his tongue involuntarily at school. Since then, there were frequent
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episodes in which he turned his eyes, shaked, stiffened and contorted his body, and
clenched his fists. She could not ascertain if there had been any loss of consciousness
during the crises, which worsened without medication. The patient attributed his crises to
his emotional state (anxiety). The mother also reported that he usually injured himself. At
age 14, he fell from a roof with a cranial trauma, remaining in coma for several days. As
she was not aware of the diagnosis and could only describe his medication (carbamezapine
200mg twice/day and risperidone 1 mg/day), the patient was referred to a neurologist for
evaluation.
During the neurological examination, the patient presented some spontaneous
myoclonic jerks of the right arm. Strength and motor coordination were decreased on the
right side. The diagnosis was a lesion of the left cerebral hemisphere with right
hemiparesis and right focal epileptic fits, which were secondarily generalized (See Table
1). Language and learning difficulties were also diagnosed (the patient has been repeating
the fifth grade for 5 years). The medical therapy was changed by increasing the
carbamezapine dosage to 600mg/day.
Oral examination and dental management
At first examination, the patient was sweating, with an altered emotional state
(extremely anxious), and could barely speak due to the pain. He was holding a tissue
napkin, soaked with blood-streaked saliva between his teeth, in an attempt to avoid biting
his tongue because of the recurrent muscular spasms (almost every three minutes). Oral
examination revealed a wound, extending from the middle dorsum to the tip of the tongue,
and from one border to other. There were edema, bleeding and lacerations and a
pseudomembrane covering the wound which was caused by recurrent bites (Figure 1a,1b).
An impression of his upper arch was taken in order to construct a silicon bite guard
(Figure 2) in an attempt to protect his tongue from cutting edges of the teeth, and
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consequently avoid more injuries. The silicon bite guard was placed on the patient on the
same day and he was advised to use it all day long with his mother’s supervision. The
excess material was cut from the borders with scissors and trimmed in order to minimize
physical stimuli to his tongue. Dietary instructions (avoidance of acid food/beverages) and
toothpaste without alcohol ingredients (menthol, eucalyptol) were provided, in order to
minimize any burning sensation in his tongue.
One week later, his tongue was in the process of healing (Figure 3a). Oral
examination was then performed revealing a poor oral hygiene, gingival bleeding,
halitosis and dental caries in the majority of his teeth, confirmed by radiographic
examinations. Dental treatment was proffered consisting of fluoride therapy (application
of fluoride varnish on teeth with enamel caries and the prescription of sodium fluoride
solution-0.05% for daily use), teeth restorations, endodontic treatment and teeth extraction
according to each tooth’s condition. The treatment continued in follow-up visits which
were scheduled to observe the healing process of tongue (Figure 3b, 3c). Oral hygiene
instructions were reinforced at every dental appointment. After one month, the patient was
advised to discontinue the use of the bite guard as the beginning of repappilation and
appearing of gustatory buds was observed.
DISCUSSION
Seizures frequently cause injuries, including bites of the tongue or other areas of
the mucosa caused by caused by the forceful contraction of the masticatory muscles .
13
The patient from the present case exhibited a severe tongue injury resulting from recurring
clonic contractions of the jaws muscles, with no change in his level of consciousness.
The silicon bite guard was chosen in view of its efficacy in therapeutic or
preventive dentistry and medicine, as documented in the literature 6,14
. This modality of
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treatment is applicable to a wide range of pediatric patients from autism 15
to epileptic
children/adolescents once it has the potential to preserve the oral structures. In this case
described in this article, a simple silicon device protected the patient’s tongue from
recurrent bites and promoted healing within a few months. However, it should be stressed
that in patients with severe mental retardation and seizure disorder, the use of removable
prosthesis is not advisable because of the risk of displacement during a seizure. In some
cases, the tooth extraction is a radical but an alternative treatment for repeated trauma in
severely handicapped patients 16.
Recent WHO data suggested that psychiatric and neurological disorders, including
epilepsy, are among the most important contributors to the global burden of human
suffering1. Epilepsy usually begins in childhood, potentially impeding education,
employment, social relationships and the development of a sense of self-worth 5. In the
present case, the disorder began when the patient was 7 years-old and slightly affected his
cognitive skills.
Karolyhazy et al (2003)17, in an epidemiologic study, found that in almost all
aspects of oral health and dental status (i.e. the state of the remaining teeth and
periodontium, the extent of restorative and prosthetic treatment), epileptic patients had a
significantly worse condition when compared to healthy subjects. According to these
authors, the higher index of decayed and missing teeth as a result of decay, injury or
periodontal disease, is consequence of the combined effect of neglected oral hygiene, oral
cavity injury and the socioeconomic background. Other studies also demonstrated that
epileptic patients have poor dental status. Rajavaara et al (2003) 18 showed that the rate of
dental restorations due to caries was higher in epileptic girls (aged from 8-18 years) than
in the matching control group; while Ogunbodede et al (1998) 19 found that almost half of
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Nigerian patients, required dental prophylaxis with oral hygiene instruction and various
types of restorative treatment.
The patient from the present case had a deficient oral hygiene. His right-side
hemiparesis made mechanical control of dental biofilm difficult to perform, leading to the
development of gingivitis and caries. These conditions justified the treatment proposed as
well as the application of a fluoride therapy, and reinforcement of oral hygiene habits at
every subsequent visit to dental clinic.
Dentists should be aware of the oral complications associated with antiepileptic
pharmacological therapy. The most common side effects of carbamezapine are diplopia,
dizziness, ataxia, headache, drowsiness and neutro/thrombocytopenia. The oral
complications are xerostomia, increased incidence of microbial infections, delayed
healing, and excessive bleeding 8. In this case, the patient did not present xerostomia (in
fact, he had sialorrhea), although he exhibited excessive gingival bleeding which could
have been caused by the medication as well as from excess of dental biofilm. It must be
taken into account that the patient had severe recurring tongue injuries leading to a longer
healing process: the wound took more than six months to heal completely, with normal
aspect of tongue’s mucosa, with the appearance gustatory buds.
Therefore it stands clearly that epilepsy (recurrent seizures) and its medical
management may affect oral health, and the interaction of a medical and dental team is
often essential to prevent oral diseases and dental/oral injuries, as well as planning of
dental treatment to provide well being and quality of life for epileptic patients.
REFERENCES
1- World Health Organization: Epilepsy: etiology, epidemiology and prognosis. Fact
sheet N°165, 2001.
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2- COMMISSION ON CLASSIFICATION AND TERMINOLOGY OF THE
INTERNATIONAL LEAGUE AGAINST EPILEPSY. Proposal for revised
classification of epilepsies and epileptic syndromes. Epilepsia 1989; 30:389-399.
3- COMMISSION ON CLASSIFICATION AND TERMINOLOGY OF THE
INTERNATIONAL LEAGUE AGAINST EPILEPSY. Semiological clinical
classification. Epilepsia 1998; 39 (9):1006-1013.
4- CAMFIELD P & CAMFIELD C. Epileptic Syndromes in Childhood: Clinical
Features, Outcomes, and Treatment. Epilepsia 2002; 43 (Suppl. 3): 27-32.
5- BLUME WT. Diagnosis and management of epilepsy. Canadian Med Assoc J 2003;
168 (4): 441-8.
6- FISKE J, BOYLE C. Epilepsy and oral care. Dent Update. 2002; 29 (4):180-7.
7- FEELY M. Drug treatment of epilepsy. BMJ 1999; 318: 106-109.
8- STOOPLER ET, SOLLECITO TP, GREENBERG MS. Seizure disorders: Update of
medical and dental considerations. DART (Dental article review and testing) 2003.
9- SEYMOUR RA, ELLIS JS, THOMASON JM. Risk factors for drug-induced
gingival overgrowth. J Clin Periodontol 2000; 27: 217-223.
10- NAKKEN KO, LOUSSIS R. Seizure-Related Injuries in Multihandicapped Patients
with Therapy-Resistant Epilepsy. Epilepsia 1993; 34 (5): 836-40.
11- BUCK D, BAKER GA, JACOBY A, SMITH DF, CHADWICK DW. Patients'
experiences of injury as a result of epilepsy. Epilepsia 1997; 38 (4): 439-44.
12- ROBERGE RJ, MACIEIRA-RODRIGUES L. Seizure-related oral lacerations:
incidence and distribution. J Am Dent Assoc 1985; 111: 279-80.
13- ANDREASEN JO, ANDREASEN, FM. Textbook and color atlas of traumatic
injuries to the teeth. 3rd, Edition. Munksgaard, Kopenhagen: Welbury RR; 1994: 185.
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14- SEALS RR, DORROUGH BC. Custom mouth protectors: a review of their
applications. J Prosthet Dent 1984; 51: 238-42.
15- POLYZOIS GL. Custom mouth protectors: an aid for autistic children. Quintessence
Int. 1989; 20 (10): 775-7.
16- TSAI T-P. Extraction as a treatment alternative follows repeated trauma in a severely
handicapped patient. Dental Traumatol 2001; 17: 139-142.
17- KAROLYHAZY K, KOVACS E, KIVOVICS P, FEJERDY P, ARANYI Z. Dental
status and oral health of patients with epilepsy: an epidemiologic study. Epilepsia
2003; 44 (8): 1103-8.
18- RAJAVAARA P, VAINIONPAA L, RATTYA J, et al. Tooth by tooth analysis of
dental health in girls with epilepsy. Eur J Paediatr Dent 2003; 4 (2): 72-7.
19- OGUNBODEDE EO, ADAMOLEKUN B, AKINTOMIDE AO. Oral health and
dental treatment needs in Nigerian patients with epilepsy. Epilepsia 1998; 39(6):590-
4.
Table 1: Semiological Seizure Classification
Epileptic Aura Autonomic Dialeptic Seizure b Motor Seizure a Special Seizure
Seizure Seizure a
Sphere Sensorial Autonomic Consciousness Motor
Subtypes Somatosensory auraa Typical dialeptic seizure b 1- Simple motor seizure a Atonic seizure a
Auditory aura a - Myoclonic seizure a Hypomotor seizure b
Olfactory aura - Epileptic spasm a Negative myoclonic seizures a
Visual aura a - Tonic-clonic seizure Astatic seizure
Gustatory aura - Tonic seizure a Akinetic seizure a
Autonomic aura a - *Clonic seizure a Aphasic seizure b
Psychic aura - Versive seizure a
Abdominal aura 2- Complex motor seizure b
Hypermotor seizure b
Automotor seizure b
Gelastic seizure
a- Left/right/axial/generalized/bilateral asymmetric
b- Left hemisphere/right hemisphere.
* Patient’s diagnosis: Simple motor seizures in which the motor movements are relatively “simple,” unnatural, and consist of movements similar to
movements elicited by electrical stimulation of the primary motor areas. Clonic seizures are a series of myoclonic contractions that regularly recur at a rate
of 0.2-0.5/s.
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Table 2: Overview of current drugs used for epilepsy (adapted from Feely 1999 7, and Fiske & Boyle 2002 6).
Drugs Resumé Oral complications
Carbama- First-line: for partial or generalized tonic-clonic seizures. Tolerability generally good - Ulceration, xerostomia, glossitis, stomatitis
zepine in children and young adults (relatively less good in elderey people). Allergic - Increased incidence of microbial infections,
reactions (rash) fairly common. Enzyme-induced drug. delayed healing, and excessive bleeding 8
Valproate First-line: for generalized (both tonic-clonic and absence) and partial seizures. Weight Decrease on platelet count and function
gain often a problem. Allergic reactions uncommon. Not an enzyme-induced drug. causing clot problems.
Lamotrigine Recently promoted (monotherapy license), wide spectrum of activity; may join first
line. Allergic reactions (rash) fairly common and severe.
Phenytoin Formerly in first-line, now less used because of side effects. Spectrum of activity Gingival overgrowth - gingival hyperplasia 9
similar to that of carbamezapine. Narrow therapeutic window plus complex
pharmacokinetics demand monitoring of drug concentration.
Vigabatrin Recent warning about visual field defects makes specialist review desirable.
Gabapentin Not very effective as an additional treatment in severe epilepsy, but may have a future
as monotherapy.
Clobazam Has valuable special uses, particularly when seizures occur in clusters.
Topiramate For treating severe epilepsy
Tiagabine Just launched in UK. Its place in clinical practice is still to be established.
Pheno- Formerly used widely in cases of refractory epilepsy; may still have a role when other
barbitone treatment fail. Specialist opinion on withdrawal of drug is advisable.
Primidone Formerly used widely in cases of refractory epilepsy
Clonazepan Formerly used widely in cases of refractory epilepsy; may still have a role when other
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treatment fail. Specialist opinion on withdrawal of drug is advisable.
Ethosuximide Alternative to valproate for petit-mal seizures only
