Oral manifestations in a group of adults with autism spectrum disorder

Objective: A number of studies have evaluated the oral health of patients with autism spectrum disorder (ASD), though most have involved children, and no specific oral manifestations have been described. The present study describes the buccodental disorders and hygiene habits in a group of adults with ASD. Study Design: A prospective case-control study was made of a group of patients with ASD (n=30), with a mean age of 27.7±5.69 years, and of a healthy age- and gender-matched control group (n=30). An evaluation was made of the medical history, medication, oral hygiene habits and oral diseases, with determination of the CAOD, CAOS and OHI-S oral hygiene scores. Results: Most of the patients in the ASD group used two or more drugs and were assisted in brushing 2-3 times a day. The most frequent manifestations were bruxism, self-inflicted oral lesions and certain malocclusions. The CAOD and CAOS scores were significantly lower than in the controls. Conclusions: Adults with ASD and assisted dental hygiene presented fewer caries than the non-disabled population. However, bruxism, ogival palate and anterior open bite were frequent in the patients with ASD. Key words:Autism spectrum disorder, caries, dental hygiene, oral manifestations.


Introduction
Patients with autism spectrum disorder (�SD) are the indi�iduals with special needs that pose the �reatest challenge for dentists, due to their complex and varied clinical manifestations. Described by Leo Kanner in 1943� these manifestations first appear in individuals under three years of age, and are characterized by deficiencies in social interaction, communication� beha�ior� interests and acti�ities (1)� as well as complex sensory alterations (2). Mana�ement is of a multidisciplinary nature� the most effecti�e strate�ies bein� based on educational pro-�rams (3). In order to educate children with �SD� it is necessary to facilitate structured situations helpin� the patients to anticipate what is �oin� to happen (4)� since they show great fear and anxiety in the face of unknown situations (5), such as visiting the dental clinic. The dental care of these patients poses great difficulties, and in most cases treatment is pro�ided under �eneral anesthesia (4�6- 8).
No specific oral manifestations of ASD have been described (1�7�9)� thou�h the oral hy�iene of these subjects is known to be deficient (1,5,10,11). Nevertheless, many authors ha�e found the pre�alence of caries and of periodontal disease to be no different compared with non-autistic indi�iduals (4�5�9�10)� and in some cases the pre�alence of caries in children with �SD may e�en be comparati�ely lower (6�8�12). Between 60-95% of all patients with ASD have an unusual sensory profile, in-cludin� dysfunction in re�isterin� oral sensiti�ity. Most studies of patients with �SD report oral patholo�y in children� and in some isolated instances both children and adults ha�e been e�aluated. Very few studies have examined only adults with ASD, and no case-control series have been published. The present study was therefore desi�ned to describe the habits of oral hy�iene and buccodental disease in a �roup of adults with �SD in our community.

Material and Methods
Study design � prospecti�e case-control study was carried out� de-scribin� the habits of oral hy�iene and oral patholo�y in a �roup of adults with �SD� compared with a �roup of healthy a�e-and �ender-matched controls.

Sample screening
The study population consisted of individuals with ASD pertainin� to two day centers for people with autism in the Valencian Community (Spain) (one in the pro�ince of Castellón and the other in the pro�ince of Valencia). Of the 40 indi�iduals in these centers� 30 met the study inclusion criteria. The latter were defined before sample screenin� and consisted of the followin�: a dia�nosis of �SD; the understandin� at least of �ery simple instructions; and the obtainment of written informed consent from the care�i�ers for participation in the study. The study sample consisted of 27 males and 3 females (n=30)� a�ed between 20-41 years (mean 27.7±5.69). �ll patients presented some de�ree of mental impairment: mild in 8 cases (26%)� moderate in 11 (37%) and se�ere in 11 (37%). � total of 63.33% of the patients were institutionalized. The control group in turn consisted of 30 non-disabled indi�iduals randomly selected from amon� the accom-panyin� persons in the Red Cross Special Patients Clinic of Valencia (Spain): 23 males (76.67%) and 7 females (23.33%)� with a mean a�e of 27.83±5.84 years.

Methodology
The parents / caregivers of the participants received an explanation of the study, together with an information sheet and informed consent form. Those who agreed to participate completed a questionnaire evaluating the medical history and habits of oral hy�iene of the patients. The data not adequately recorded in this way were obtained by inter�iewin� the parents / care�i�ers. The study received the support and collaboration of the technical personnel of the different participatin� centers. Two dentists specialized in the oral care of disabled patients participated� to�ether with a stomatolo�ist and a psycholo�ist specialized in �SD. One to 5 anticipatory workshops or sessions were held per subject before the dental examination. To this effect we used photo�raphs� picto�rams� macromodels� tooth brushes and real objects such as caries probes� dental mirrors, masks and gloves. Each workshop lasted 20 minutes� with the participation of 1-2 people. Desensitization was achie�ed throu�h successi�e approaches� and use was made of the Tell-Show-Do (TSD) technique, visual pedagogy, in vivo modeling, audiovisual modelin�� beha�ioral testin� and self-modelin� from photo�raphs. Sensory processin� was assessed with the widely validated Sensory Profile Questionnaire. The sensory profile reflects the response to sensory stimuli. In this way we can identify the search for sensations� emotional reacti�ity� tone / resistance� oral sensory sen-siti�ity� inattention / distractibility� poor re�istry� sensory sensitivity, sedentarism and fine motor perception. All clinical dental examinations were made by the same dentist. � number 5 dental mirror was used� to�ether with a caries exploratory probe. The exploration was carried out in an adequately conditioned room with limited decoration� silence and no distractions. � stretchertype chair and guidable light source of sufficient power for adequate intraoral exploration were used, and all the data were recorded in the patient clinical history. At the end of the examination, the parents / caregivers recei�ed a report on the oral disorders found� with su�-�estions re�ardin� the necessary treatments and dental care. The CAOD index was determined in both groups, as-e417 sessin� the caried� absent and obturated teeth corresponding to all the individuals examined per group (considering only 28 permanent teeth). The CAOS index was also determined under the same conditions as before� thou�h in this case the basic unit was the dental surface. Specifically, 5 surfaces were considered in posterior teeth� and four surfaces in anterior teeth. Oral hygiene was rated using the Simplified Oral Hygiene Index (OHI-S). The criteria of this index are related to the sum of the Plaque Index Score (PI-S) and the Calculus Index Score (CI-S). In both cases the surfaces of index teeth were examined: vestibular surfaces of 11, 16, 26 and 31; lin�ual surfaces of 36 and 46. In the absence of index teeth, we explored the entire sextant and recorded the maximum degree (except third molars). The PI-S was scored as 0: no plaque; 1: plaque covering no more than 1/3 of the examined surface; 2: > 1/3 but < 2/3; 3: > 2/3 of the surface. The CI-S was scored as 0: no calculus; 1: supragingival calculus < 1/3; 2: >1/3 but <2/3 of the surface or isolated sub�in�i�al points; 3: > 2/3 of the surface or subgingival in the form of a continuous band.

Data analysis
The SPSS version 18 statistical package for Microsoft Windows ® was used to analyze the data. � descripti�e study was made involving the frequencies of the different �ariables� to�ether with a comparati�e analysis of the two �roups. The Student t-test was used to compare means of quan-titati�e �ariables between the two �roups� while the Mann-Whitney �-test was used for continuous / discontinuous variables showing a non-normal (skewed) distribution. The Fisher test was used for comparing qualitative variables. Statistical significance was accepted for p<0.05 and p<0.01.

Clinical research ethics committee
The study was evaluated and approved by the Clinical Research Ethics Committee of the University of Valencia.

Results
The medical history of the patients with ASD revealed only two cases (6.67%) of gastroesophageal reflux, and 5 cases (16.67%) of epilepsy. Fifty percent suffered selfinflicted lesions (80% affecting the hands and arms, 47% the head and neck, and only 13% the rest of the body). A full 77% were receiving some kind of medication. The drug distribution was as follows: anxiolytics 57%� antipsychotics 48%� anticon�ulsi�ants 39%� neuroleptics 22%� antidepressants 17%� and other medications 22%. Of the 23 patients under medication� 78% recei�ed two or more dru�s. �s re�ards oral hy�iene� almost one-half of the patients with �SD (46.67%) underwent fully assisted tooth brushing. Thirty percent were initially autonomous, but finally required the help of the parents / caregivers. Only 23.33% were fully autonomous for tooth brush-in�. In turn� 46.67% brushed at least three times a day� 40% twice a day� and 13.33% once a day. Only a manual toothbrush was used in 25 cases (83.33%)� and an electric toothbrush in 5 cases (16.67%). Extraoral examination of the patients with ASD re-�ealed cheilitis in 13.33% of the cases and droolin� in 7%. Intraoral examination in turn revealed self-inflicted lesions in 13% of the cases� correspondin� to bitin� of the lips and inside of the cheek, and intraoral ulcers. O�er one-half of the patients (60%) showed wear fac-ets� affectin� the enamel and dentin in 61% of the cases. Twenty percent suffered dental traumatisms -all in the upper anterior sector. Re�ardin� the presence of maloc-clusions� 36.67% presented o�i�al palate� 46.67% dental crowdin�� and 30% anterior open bite. Caries were present in 60% of the subjects. The CAOD score for the 30 patients was 3.7� with �alues of 1.33� 1 and 1.37 for caried� absent and obturated teeth� respectively. The OHI-S score was 1.92, and the PI-S score (1.57) was �reater than the CI-S score (0.41). On comparin� oral hy�iene amon� the patients with �SD and the controls� only 23.33% of the former were seen to brush their teeth autonomously� �ersus 100% of the subjects without �SD. In most of the patients with ASD the brushing frequency was at least three times a day, versus twice a day in the controls (Table 1). On comparin� the oral patholo�y in the two �roups� a significantly greater presence of ogival palate and anterior open bite was recorded in the �SD �roup (p<0.05 and p<0.01, respectively) ( Table 2). Table 3 shows the caries and oral hygiene index scores for both groups. Statistically significant differences were observed in relation to the CAOD index (p<0.05) The amount of plaque was significantly greater in the �SD �roup than in the controls without �SD (p<0.01).
The time taken to complete the dental examination in the �SD �roup was 8.13 minutes (ran�e 5.33-15.5). In comparison, the time taken to complete the dental examination in the control �roup was 5.52 minutes (ran�e 4.4-6.2) -the intergroup difference being statistically significant (p<0.01). One half of the patients with ASD showed altered processin� in �arious sensory modalities -this evidencing the presence of a global sensorial disorder. Inadequate oral sensitivity registry was docu-mented in 25% of the cases -a situation that could have influence the habits of oral hygiene and oral disease in the study sample.

Discussion
The medical history of patients with autism spectrum disorder (�SD) could condition the appearance of certain oral lesions or may influence the definition of a dental treatment plan. In our study there were fewer patients with gastroesophageal reflux than in other studies such as that published by Ibrahim et al. (13). Likewise, we obser�ed a lesser pre�alence of epilepsy (9�14). �owever, self-inflicted injuries are one of the manifestations that may pose a problem for the dental mana�ement of these patients. The prevalence of these lesions was 13% in our series -a proportion similar to that reported by Ba�hdadli et al. (15)� but far lower than in other studies (in the order of 68%) (6�16).
Most of the patients with �SD were recei�in� dru� treatment� and a lar�e percenta�e recei�ed 2-3 dru�s a day. These values are far higher than those reported by Loo et al. (12)� and may be due to the fact that our series in�ol�ed only adult patients� most of which were more-o�er institutionalized� while most other studies found in the literature are about pediatric patients. The most frequently prescribed drugs were antipsychotics and anxiolytics that can affect salivation and favor bacterial plaque formation and caries. Nevertheless, we found no differences between the two �roups in terms of the presence of caries.
Most of the patients with ASD required assistance in tooth brushin�� in coincidence with the obser�ations of other authors (9,17,18). Indeed, only one-quarter of the patients were able to complete brushin� without help. However, the frequency of assisted brushing was high (2-3 times a day)� and mostly in�ol�ed manual brushin� rather than the use of an electric toothbrush (6,17). This may be explained by the fact that most of the patients were institutionalized adults following a habit acquired with their care�i�ers.
In any case, dental hygiene tends to be insufficient due to dietary problems� since these patients show a preference for certain flavors and textures (e.g., sweet, soft and sticky foods  In contrast, significant differences were observed in the case of the complete CAOS index and the scores by sur-faces� which were both lower in the �SD �roup. On examining the number of dental traumatisms, our data coincide with those found in the literature (20% and 21%) (9�19). �ll of these injuries affected teeth in the anterior sector� and were due to falls secondary to walking problems and the lack of patient caution in situations of risk -which increases vulnerability to accidents.
Patients with ASD do not present very specific oral disorders (1�7�9)� thou�h they do show a �reater tendency towards certain malocclusions (e.�.� o�i�al palate and anterior open bite). Oz�en et al. (20) also obser�ed these differences regarding malocclusion. Likewise, our patients with �SD showed dental crowdin� in a lar�er proportion of cases than in the study of DeMattei et al. (21)� thou�h the pre�alence was comparati�ely hi�her in the control �roup.
Bruxism was more frequent than in other studies (21% to 44%) (6,19,21). This may be because the mentioned studies e�aluated mainly children� and the patients with bruxism in our study were institutionalized and presented important mental retardation. Lastly� while we also recorded self-inflicted oral lesions and drooling, the proportions were lower than in other studies (21).
In conclusion� the present study shows that institutionalized adults with �SD and assisted dental hy�iene present fewer caries than the non-disabled population.
The patients do show some characteristic oral manifestations in the form of self-inflicted lesions, bruxism and malocclusions such as o�i�al palate and anterior open bite.