Video Abstract

Video Abstract

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OBJECTIVE

Children with autism spectrum disorder (ASD) have difficulty participating in dental care and experience significant unmet dental needs. We examined the efficacy of parent training (PT) for improving oral hygiene and oral health in underserved children with ASD.

METHOD

Families of Medicaid-eligible children with ASD (ages 3–13 years, 85% boys, 62% with intellectual disability) reporting difficulty with dental care participated in a 6-month randomized controlled trial comparing PT (n = 60) with a psychoeducational dental toolkit (n = 59). Primary outcomes were parent-reported frequency of twice-daily toothbrushing and dentist-rated visible plaque. Secondary outcomes included parent-reported child behavior problems during home oral hygiene and dentist-rated caries. Dentists were blind to intervention assignment. Analyses were intention to treat.

RESULTS

Retention was high at posttreatment (3 months, 93%) and 6-month follow-up (90%). Compared with the toolkit intervention, PT was associated with increased twice-daily toothbrushing at 3 (78% vs 55%, respectively; P < .001) and 6 (78% vs 62%; P = .002) months and a reduction in plaque at 3 months (intervention effect, −0.19; 95% confidence interval [CI], −0.36 to −0.02; P = .03) and child problem behaviors at 3 (−0.90; 95% CI, −1.52 to −0.28; P = .005) and 6 (−0.77; 95% CI, −1.39 to −0.14; P = .02) months. Comparatively fewer caries developed in children receiving the PT intervention over 3 months (ratio of rate ratios, 0.73; 95% CI, 0.54 to 0.99; P = .04).

CONCLUSIONS

PT represents a promising approach for improving oral hygiene and oral health in underserved children with ASD at risk for dental problems.

What’s Known on This Subject:

Children with autism spectrum disorder have difficulty participating in dental care and experience significant unmet dental needs, which may be exacerbated by underserved status. No known interventions have improved oral hygiene or oral health in this population in randomized trials.

What This Study Adds:

In a randomized trial involving underserved children with autism spectrum disorder and dental care problems, parent training significantly improved oral hygiene and health relative to a psychoeducational toolkit. Parent training represents a promising oral health intervention for this high-risk population.

One of the most common unmet child health care needs, dental care presents a particular challenge for children with autism spectrum disorder (ASD),1  a population with an elevated risk for plaque, caries, and oral health problems across numerous studies,27  although not universally.8,9  Children with ASD exhibit greater distress during dental care and experience increased difficulty with participating in routine oral hygiene and dental visits than children without ASD.1012  Challenges with participating in dental care may compromise care quality13,14  and may adversely influence parental perspectives about future dental care.11  Multiple factors may impede dental care for children with ASD, including child ASD symptoms, comorbid intellectual disability (ID), communication difficulties, and challenging behaviors.1,1517  Barriers associated with low income and racial or ethnic minority status may compound difficulties in obtaining dental care.1,15 

Dental interventions for children with ASD have predominantly focused on improving compliance with dental office procedures.1833  Less attention has been devoted to oral hygiene, despite evidence of poor oral hygiene in children with ASD6,10,12,34,35  and close links among oral hygiene, oral health, and overall well-being.3638  Leveraging work with parents to promote oral hygiene in children with ASD holds promise given the fundamental role parents play in supporting oral hygiene and oral health for all children36,3841  and the heightened influence of parental support for children with ASD,42  particularly among children with comorbid ID.43 

Daily living skills have long been targeted through behavioral intervention,44  but few studies have focused specifically on oral hygiene in children with ASD. In a multiple-baseline study, investigators used gradual exposure to increase and generalize toothbrushing compliance in 3 children with ASD.45  Parent-implemented visual supports have been associated with increased toothbrushing participation4648  and improved oral health in children with ASD in pre- and postintervention designs,4850  and researchers of a randomized trial found promising, but nonsignificant effects of video modeling on oral health.51  Additionally, in an uncontrolled trial, researchers linked caregiver psychoeducation and training in toothbrushing to improved oral health in individuals with ASD.52  Collectively, findings reveal that parent-implemented intervention may be a useful approach for improving oral hygiene and oral health in children with ASD. However, randomized controlled trials (RCTs) are lacking, as are interventions that target both oral hygiene behavior and oral health outcomes. Additional attention to economically disadvantaged families is also needed.

In the current investigation, we have addressed these limitations by developing and testing a parent training (PT) intervention designed to improve primary outcomes of daily home oral hygiene and oral health in underserved families of children with ASD. In this RCT, we examined the efficacy of PT (R. Fenning, K. McKinnon-Bermingham, and E. Butter, unpublished manual, 2017) relative to a psychoeducational dental toolkit for increasing frequency of twice-daily home toothbrushing and reducing visible plaque. We expected that PT would significantly improve oral hygiene and oral health relative to the toolkit at postintervention (3 months, secondary end point) and that relative gains would be maintained at 6-month follow-up (primary end point). Secondary aims examined the efficacy of PT for reducing severity of child behavior problems during home dental care and in preventing caries. Post hoc exploratory analyses investigated changes in oral hygiene as mechanisms underlying PT effects on oral health.

Families of children with ASD and Medicaid eligibility were recruited from December 2016 to December 2018 for participation in a 6-month RCT at 2 hospital-affiliated sites in a large research network. Follow-up concluded in June 2019. Additional eligibility criteria included parent-reported difficulty participating in dental care, child age 3 to 13 years at enrollment, child ASD diagnosis confirmed through clinical best estimate on the basis of the Diagnostic and Statistical Manual of Mental Disorders, Fifth Edition,53  research-reliable administration of the Autism Diagnostic Observation Schedule-Second Edition,54  and parent-reported absence of child dental screenings and examinations in the previous 6 months. Exclusion criteria included an acute dental condition requiring immediate emergency treatment, medication-induced oral health adverse effects, and participation in nonstudy dental interventions. Study procedures were conducted in English.

Phone screening was completed with 266 families. Of these, 157 families consented to baseline assessments, and 119 were eligible for study participation. Eligible participants were randomly assigned by the data coordinating center using a concealed computer-generated, site-stratified, permuted block schedule that assigned participants equally to the PT (n = 60) or toolkit (n = 59) intervention. Our 3 study dentists were blinded to intervention assignment. Retention was high (90%), with 107 families completing the final follow-up (Fig 1).

FIGURE 1

Flow of families through an RCT of PT versus a psychoeducational dental toolkit.

FIGURE 1

Flow of families through an RCT of PT versus a psychoeducational dental toolkit.

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An independent data and safety monitoring board oversaw study procedures, which were approved by site institutional review boards (2016P001195/PHS, HS-2016-3043, IRB16-00736, HSR-17-18-580). Parents provided written informed consent, and children provided assent when appropriate. Phone screening was used to determine demographic eligibility. Prerandomization baseline assessments included parent questionnaires, a dental visit that yielded standardized ratings of oral health, and a clinic visit that involved testing of child intellectual functioning and ASD symptoms. Follow-up dental visits and parent questionnaires provided outcome measures at 3 and 6 months after baseline.

Participants were seen by the same board-certified pediatric dentist at each time point. Reliability for oral health measures was established prestudy using photographs; during the trial, dentists engaged in ongoing calibration through monthly calls and annual in-person exercises. Dental visits involved a standard chair, overhead light, visual examination of full-mouth caries followed by plaque ratings on index teeth, and preventive cleaning, adjusted for children’s presenting needs. Given our population, dental charting did not include a dental explorer or x-rays.

Dental Toolkit

The toolkit was an active, multicomponent comparator involving (1) the Autism Speaks Autism Treatment Network/Autism Intervention Research Network on Physical Health Dental Toolkit, a family-friendly psychoeducational resource about oral hygiene and dental visits; (2) an Oral-B Vitality Floss Action electric toothbrush to support plaque reduction55 ; (3) a 6-month supply of oral hygiene materials (toothbrush heads, fluoride toothpaste, and flossers), and (4) mock dental tools (eg, dental mirror). Manual toothbrushes were provided to encourage family-wide oral hygiene.

PT to Improve Dental Care

Families participating in PT received all toolkit components and the novel intervention curriculum (R. Fenning, K. McKinnon-Bermingham, E. Butter, unpublished manual, 2017) outlined in Table 1. Individual PT comprised 7 core in-person sessions, including a home visit and a dental office coach, and 4 phone booster sessions. PT included psychoeducation and was centered on behavioral techniques shown to be effective in the treatment of children with ASD and existing strategies used clinically in populations without ASD to address dental fear and maladaptive behaviors during dental care. An emphasis on optimizing parent engagement was embedded throughout treatment through the use of motivational interviewing to address expectancies, goal setting, and treatment barriers56 ; application of cognitive-behavioral techniques to enhance parenting self-efficacy57 ; and incorporation of mindful awareness to support nonjudgment of challenges related to dental care. Supplemental supports included a study library of video models, social stories, and parent handouts. Doctoral-level clinical or counseling psychologists and board-certified behavior analysts delivered treatment. The PT manual included detailed instructions and verbatim scripts. Therapists participated in annual in-person cross-site training and weekly cross-site case supervision to ensure fidelity and integrity of PT delivery. Therapists also provided ratings of intervention fidelity and parental adherence after each intervention session.

TABLE 1

Curriculum Overview of PT to Improve Dental Care in Children With ASD

WeekSessionLength, minContent
Overview, participation enhancement, and introduction to behavioral principles 90 Conduct adapted participation enhancement intervention56  to address parental expectations, motivation, goal setting, and treatment barriers. Provide treatment overview, review antecedent-behavior-consequence model and functions of behavior, assess oral aversions, and introduce antecedent interventions (eg, daily toothbrushing schedule, use of behavioral momentum, arranging the environment, providing choices). 
Introduction to toothbrushing 90 Introduce priming as an antecedent intervention (visual schedule, video models, social story). Present toothbrushing task analysis and introduce prompting. Integrate use of consequences (reinforcement, planned ignoring). Provide video models and develop home dental support plan. 
Toothbrushing with child 90 Introduce use of distraction and noncontingent escape (taking breaks). Present video models. Engage parent in in vivo practice with child. Refine home dental support plan. 
Home coaching 60 Practice in vivo toothbrushing with child. Update home dental support plan as needed. 
Flossing and diet 60 Introduce and demonstrate flossing (video models). Provide psychoeducation regarding diet and other factors influencing oral health. Update home dental support plan as needed. Forecast preparation for dental visit. Provide related video models. 
Preparing for the dental visit 60 Present task analysis of dental visit and introduce desensitization practice. Review antecedent interventions, distraction, and noncontingent escape as applied to the dental office. Address planned use of consequences. Develop dental visit support plan. 
Dental visit coaching 60 Engage parent in in vivo practice with child at the dental office. Update home and dental visit support plans as needed. 
10 Phone booster 1 15–20 Review home practice and implementation of interventions. Refine home and dental visit support plans as needed. 
12 3-month dental office visit   
13 Phone booster 2 15–20 Review home practice and implementation of interventions. Refine home and dental visit support plans as needed. 
16 Phone booster 3 15–20 Review home practice and implementation of interventions. Refine home and dental visit support plans as needed. 
21 Phone booster 4 15–20 Review home practice and implementation of interventions. Refine home and dental visit support plans as needed. 
24 6-month dental office visit   
WeekSessionLength, minContent
Overview, participation enhancement, and introduction to behavioral principles 90 Conduct adapted participation enhancement intervention56  to address parental expectations, motivation, goal setting, and treatment barriers. Provide treatment overview, review antecedent-behavior-consequence model and functions of behavior, assess oral aversions, and introduce antecedent interventions (eg, daily toothbrushing schedule, use of behavioral momentum, arranging the environment, providing choices). 
Introduction to toothbrushing 90 Introduce priming as an antecedent intervention (visual schedule, video models, social story). Present toothbrushing task analysis and introduce prompting. Integrate use of consequences (reinforcement, planned ignoring). Provide video models and develop home dental support plan. 
Toothbrushing with child 90 Introduce use of distraction and noncontingent escape (taking breaks). Present video models. Engage parent in in vivo practice with child. Refine home dental support plan. 
Home coaching 60 Practice in vivo toothbrushing with child. Update home dental support plan as needed. 
Flossing and diet 60 Introduce and demonstrate flossing (video models). Provide psychoeducation regarding diet and other factors influencing oral health. Update home dental support plan as needed. Forecast preparation for dental visit. Provide related video models. 
Preparing for the dental visit 60 Present task analysis of dental visit and introduce desensitization practice. Review antecedent interventions, distraction, and noncontingent escape as applied to the dental office. Address planned use of consequences. Develop dental visit support plan. 
Dental visit coaching 60 Engage parent in in vivo practice with child at the dental office. Update home and dental visit support plans as needed. 
10 Phone booster 1 15–20 Review home practice and implementation of interventions. Refine home and dental visit support plans as needed. 
12 3-month dental office visit   
13 Phone booster 2 15–20 Review home practice and implementation of interventions. Refine home and dental visit support plans as needed. 
16 Phone booster 3 15–20 Review home practice and implementation of interventions. Refine home and dental visit support plans as needed. 
21 Phone booster 4 15–20 Review home practice and implementation of interventions. Refine home and dental visit support plans as needed. 
24 6-month dental office visit   

To provide flexibility and maximize dosage for underserved families, 8% of core sessions were completed after the 3-month follow-up assessment. Therapists also had the discretion to substitute a home visit for 1 phone booster session during the maintenance phase.

Demographics and Family Information

Parents reported on parent and child age, sex, race, ethnicity, home language(s), marital status and cohabitation, household income, and child comorbid diagnoses, medication status, and intervention history.

Dental Experiences

Parents reported on children’s current oral hygiene practices and parent and child previous dental visit history.

Intellectual Functioning

Child intellectual functioning was estimated using the Stanford-Binet 5 abbreviated battery IQ,58  which yields a standard score with a mean of 100 and an SD of 15.

Adaptive Behavior

Adaptive behavior was measured using the Adaptive Behavior Composite score from the Vineland Adaptive Behavior Scales 3rd Edition Comprehensive Interview Form,59  which has a mean of 100 and an SD of 15.

Overall Behavior Problems

Parent-reported overall child behavior problems were assessed using the Total Problems T-score from the Child Behavior Checklist (CBCL),60  with higher scores denoting more behavior problems.

Frequency of Home Toothbrushing

Parents reported the number of days in the past week that children completed toothbrushing twice per day, with or without help.

Visible Plaque

Dentists used a standard Visual Plaque Index (VPI)55,61,62  to rate the buccal and lingual nonrestored surfaces of index teeth63  on a 0 to 5 scale (0 = no plaque, 5 = plaque on more than two-thirds of tooth surface). Interrater reliability was adequate to excellent (82%–100% within 1 scale point).

Severity of Behavior Problems During Home Oral Hygiene

Parents reported on the occurrence and severity of 8 behavior problems during the past week’s oral hygiene activities (not listening, difficulty sitting/standing still, actively resisting, eloping, fearful/anxious behaviors, screaming/yelling, aggression, and self-injury) using a 0 to 9 scale (0 = no problem, 9 = severe problem). Items were averaged to produce a single score (α = 0.91).

Caries

Dentists completed the Decayed, Missing, and Filled Teeth Index (dmft/DMFT)64  to document the presence and progression of full-mouth caries. The dmft/DMFT indicates the number of primary/permanent teeth that are decayed (d/D), missing due to caries (m/M), and filled (f/F). The d2/D2 code represents clinically detectable cavitated lesions. Interrater reliability was adequate to excellent (κ = 0.61–0.92).

Assuming at least 30% correlation between baseline and follow-up estimates and 2-tailed testing at α = 0.025 given 2 primary outcomes, a minimum sample size of 100 was selected to provide 80% power to detect an effect size of 0.6. Based on trial variance estimates, this effect size corresponds to a 6-month treatment difference of 1.2 days/week in twice-daily toothbrushing and a 0.3-unit change in VPI. Enrollment targets permitted balanced attrition of 15%.

Analyses were intention to treat. Missing baseline data were mean imputed for 1 participant who exited the study before completing outcome measures. Treatment-dependent changes in outcomes were analyzed using shared baseline generalized mixed models with fixed effects for visit and child age, interaction terms for child age by visit and treatment by postbaseline visit, and random intercepts for each participant and site. Sensitivity analyses included main effects and interactions with visit for primary caregiver sex and baseline toothbrushing frequency. A Gaussian distribution with an identity link was used for continuous measures. A Poisson distribution with log link was used for dmft/DMFT and d2/D2 counts. A binomial distribution with a logit link was used for number of days of completed toothbrushing. Within-group changes from baseline and between-group differences were estimated using linear contrasts of least-squares means. Post hoc causal mediation analysis tested mediation of significant 3-month changes in oral health by concurrent changes in oral hygiene (twice-daily toothbrushing, electric toothbrush use, and severity of interfering behaviors). Percentages of total effect attributed to mediation are reported. Tests were 2-sided at P < .025 for primary outcomes and P < .05 for other comparisons. Analyses were performed using SAS version 9.4 software (SAS Institute Inc, Cary, NC).

Individual deidentified participant data for the variables used in the analyses will be shared. The study protocol and statistical analysis plan will also be made available. Data will become available 18 months after the final study closeout date of August 31, 2020. Individuals interested in accessing the data should contact the corresponding author. Data will be made available to investigators pursuant Autism Intervention Network on Physical Health policies and institutional review board requirements.

Primary caregivers tended to be female (90%) and married (53%), with a substantial proportion identifying as Hispanic/Latino (40%). The majority of children met Diagnostic and Statistical Manual of Mental Disorders, Fifth Edition, criteria for comorbid ID on the basis of study assessments (62%) and presented with clinically elevated behavior problems at baseline (61% CBCL T-score >63). Baseline characteristics were similar between randomly assigned groups, except PT primary caregivers were more often women, and PT families reported less frequent baseline oral hygiene than toolkit families (Table 2).

TABLE 2

Baseline Demographics and Clinical Characteristics by Treatment Group

VariablePT (n = 60)Toolkit (n = 59)
Child   
 Boys, n (%) 49 (82) 52 (88) 
 Age, y   
  Mean (SD) 7.3 (2.6) 7.4 (2.7) 
  Range 3.1–13.0 3.0–13.3 
 Race, n (%)   
  White 34 (61) 31 (61) 
  Asian 7 (13) 5 (10) 
  Black/African American 5 (9) 5 (10) 
  Hawaiian/Pacific Islander 2 (4) 1 (2) 
  American Indian/Alaskan Native 1 (2) 
  Multiracial 8 (14) 8 (16) 
 Ethnicity, n (%)   
  Hispanic/Latino 22 (39) 20 (36) 
 IQ   
  Mean (SD) 66.2 (19.5) 71.2 (20.3) 
  Range 47–112 47–124 
 Adaptive behavior, mean (SD) 57.1 (16.1) 57.8 (19.1) 
 ID, n (%) 41 (68) 33 (56) 
 ASD symptom severity, mean (SD) 7.3 (1.7) 7.2 (1.9) 
 Behavior problems   
  CBCL T-score, mean (SD65 (9) 67 (10) 
  Clinically elevated (T-score >63), n (%) 32 (55) 38 (67) 
Primary caregiver   
 Age, y, mean (SD) 37 (7) 37 (9) 
 Women, n (%) 58 (97) 48 (83)* 
 Race, n (%)   
  White 37 (74) 34 (68) 
  Asian 7 (14) 6 (12) 
  Black/African American 3 (6) 4 (8) 
  Hawaiian/Pacific Islander 1 (2) 
  American Indian/Alaskan Native 1 (2) 
  Multiracial 2 (4) 5 (10) 
 Ethnicity, n (%)   
  Hispanic/Latino 24 (41) 22 (39) 
 Education, n (%)   
  Less than eighth grade 1 (2) 3 (5) 
  Some high school 6 (10) 3 (5) 
  High school graduate 8 (14) 12 (21) 
  Some college 32 (54) 24 (42) 
  College degree 7 (12) 8 (14) 
  Advanced degree 5 (8) 7 (12) 
 Living situation, n (%)   
  Married/domestic partner 32 (53) 30 (53) 
  Single/other adults in home 14 (23) 15 (26) 
  Single/no other adults in home 14 (23) 12 (21) 
 Annual gross family income, $, n (%)   
  <15 000 9 (16.1) 8 (15) 
  15 000–24 999 14 (25) 13 (24) 
  25 000–34 999 11 (20) 14 (26) 
  35 000–49 999 13 (23) 7 (13) 
  50 000–74 999 7 (13) 6 (11) 
  ≥75 000 2 (4) 6 (11) 
 Primary home language English, n (%) 50 (85) 47 (89) 
Baseline home oral hygiene   
 Past-week twice-daily toothbrushing, mean (SD) 2.48 (2.51) 3.74 (2.90)* 
 Behavior problem severity during oral hygiene, mean (SD) 2.92 (2.39) 2.78 (2.33) 
Baseline oral health   
 Visible plaque, mean (SD) 0.95 (0.68) 0.77 (0.50) 
 Caries, mean (SD) 3.19 (4.55) 2.72 (3.21) 
 No. of decayed teeth, mean (SD) 1.19 (2.12) 0.67 (1.39) 
 Child age at first dental visit, y, mean (SD) 3 (2) 3 (2) 
VariablePT (n = 60)Toolkit (n = 59)
Child   
 Boys, n (%) 49 (82) 52 (88) 
 Age, y   
  Mean (SD) 7.3 (2.6) 7.4 (2.7) 
  Range 3.1–13.0 3.0–13.3 
 Race, n (%)   
  White 34 (61) 31 (61) 
  Asian 7 (13) 5 (10) 
  Black/African American 5 (9) 5 (10) 
  Hawaiian/Pacific Islander 2 (4) 1 (2) 
  American Indian/Alaskan Native 1 (2) 
  Multiracial 8 (14) 8 (16) 
 Ethnicity, n (%)   
  Hispanic/Latino 22 (39) 20 (36) 
 IQ   
  Mean (SD) 66.2 (19.5) 71.2 (20.3) 
  Range 47–112 47–124 
 Adaptive behavior, mean (SD) 57.1 (16.1) 57.8 (19.1) 
 ID, n (%) 41 (68) 33 (56) 
 ASD symptom severity, mean (SD) 7.3 (1.7) 7.2 (1.9) 
 Behavior problems   
  CBCL T-score, mean (SD65 (9) 67 (10) 
  Clinically elevated (T-score >63), n (%) 32 (55) 38 (67) 
Primary caregiver   
 Age, y, mean (SD) 37 (7) 37 (9) 
 Women, n (%) 58 (97) 48 (83)* 
 Race, n (%)   
  White 37 (74) 34 (68) 
  Asian 7 (14) 6 (12) 
  Black/African American 3 (6) 4 (8) 
  Hawaiian/Pacific Islander 1 (2) 
  American Indian/Alaskan Native 1 (2) 
  Multiracial 2 (4) 5 (10) 
 Ethnicity, n (%)   
  Hispanic/Latino 24 (41) 22 (39) 
 Education, n (%)   
  Less than eighth grade 1 (2) 3 (5) 
  Some high school 6 (10) 3 (5) 
  High school graduate 8 (14) 12 (21) 
  Some college 32 (54) 24 (42) 
  College degree 7 (12) 8 (14) 
  Advanced degree 5 (8) 7 (12) 
 Living situation, n (%)   
  Married/domestic partner 32 (53) 30 (53) 
  Single/other adults in home 14 (23) 15 (26) 
  Single/no other adults in home 14 (23) 12 (21) 
 Annual gross family income, $, n (%)   
  <15 000 9 (16.1) 8 (15) 
  15 000–24 999 14 (25) 13 (24) 
  25 000–34 999 11 (20) 14 (26) 
  35 000–49 999 13 (23) 7 (13) 
  50 000–74 999 7 (13) 6 (11) 
  ≥75 000 2 (4) 6 (11) 
 Primary home language English, n (%) 50 (85) 47 (89) 
Baseline home oral hygiene   
 Past-week twice-daily toothbrushing, mean (SD) 2.48 (2.51) 3.74 (2.90)* 
 Behavior problem severity during oral hygiene, mean (SD) 2.92 (2.39) 2.78 (2.33) 
Baseline oral health   
 Visible plaque, mean (SD) 0.95 (0.68) 0.77 (0.50) 
 Caries, mean (SD) 3.19 (4.55) 2.72 (3.21) 
 No. of decayed teeth, mean (SD) 1.19 (2.12) 0.67 (1.39) 
 Child age at first dental visit, y, mean (SD) 3 (2) 3 (2) 
*

P < .05.

At baseline, 64% of families reported attempting toothbrushing daily, 51% reported completing brushing once per day, and 22% reported completing twice-daily toothbrushing. Most families used a manual toothbrush (75%), and 69% used fluoride toothpaste. A majority (75%) reported challenges with child behavior during toothbrushing. Regarding dental office care, 50% of children had received a past-year dental visit; use of pharmacologic interventions and physical restraint during previous visits was reported for 43% and 56%, respectively. For the 25 children (21%) without a previous dental visit, parental concerns about child behavior at the dental office emerged as the most commonly reported barrier (76%).

Of the 58 families who received PT, 48 (83%) completed all core sessions 1 to 7, and 37 (64%) completed all booster sessions 8 to 11. Therapist-rated intervention fidelity (95%) and adherence (94%) were excellent across all PT sessions (1–11). No adverse events were identified.

Results for primary and secondary outcomes are presented in Table 3 and Fig 2. Frequency of twice-daily toothbrushing improved significantly within each group. However, gains were significantly greater at both time points for children in PT relative to the toolkit intervention. Only PT significantly decreased visible plaque. PT resulted in greater plaque reduction than the toolkit intervention at 3 months. Group differences fell below significance at 6 months.

FIGURE 2

Estimated means with 95% CIs for oral hygiene and oral health outcomes.

FIGURE 2

Estimated means with 95% CIs for oral hygiene and oral health outcomes.

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TABLE 3

Oral Hygiene and Oral Health Outcomes at 3 and 6 Months

Outcome and Time PointPT (n = 60)Toolkit (n = 59)
Estimate(95% CI)Effect(95% CI)Estimate(95% CI)Effect(95% CI)Group DifferenceIntervention Effect (95% CI)
Home oral hygiene      
 Frequency of twice-daily tooth brushinga      
  Baseline  —  — — 
   Days 2.8 (2.2 to 3.5) — 2.8 (2.2 to 3.5) — — 
   % 40 (31 to 50) — 40 (31 to 50) — — 
  3 mo — 5.16*** (3.59 to 7.40) — 1.83** (1.27 to 2.63) 2.82*** (1.71 to 4.64) 
   Days 5.4 (4.8 to 5.9) — 3.9 (3.1 to 4.7) — — 
   % 78 (69 to 85) — 55 (44 to 67) — — 
  6 mo — 5.26*** (3.63 to 7.62) — 2.39*** (1.66 to 3.44) 2.20** (1.33 to 3.65) 
   Days 5.5 (4.8 to 6.0) — 4.3 (3.5 to 5.1) — — 
   % 78 (69 to 85) — 62 (50 to 72) — — 
 Severity of behavior problems during oral hygiene activities      
  Baseline 2.89 (2.51 to 3.27) — 2.89 (2.51 to 3.27) — — 
  3 mo 1.71 (1.21 to 2.22) −1.18*** (−1.64 to −0.71) 2.62 (2.12 to 3.11) −0.28 (−0.73 to 0.18) −0.90** (−1.52 to −0.28) 
  6 mo 1.37 (0.86 to 1.88) −1.52*** (−1.99 to −1.05) 2.14 (1.64 to 2.64) −0.75** (−1.21 to −0.29) −0.77* (−1.39 to −0.14) 
Oral health status      
 Visible plaque      
  Baseline 0.84 (0.75 to 0.94) — 0.84 (0.75 to 0.94) — — 
  3 mo 0.68 (0.55 to 0.81) −0.17* (−0.29 to −0.04) 0.87 (0.74 to 1.00) 0.03 (−0.10 to 0.15) −0.19* (−0.36 to −0.02) 
  6 mo 0.75 (0.62 to 0.88) −0.09 (−0.22 to 0.04) 0.80 (0.67 to 0.92) −0.05 (−0.17 to 0.08) −0.05 (−0.21 to 0.12) 
 Full-mouth caries (dmft/DMFT)b      
  Baseline 0.88 (0.33 to 2.33) — 0.88 (0.33 to 2.33) — — 
  3 mo 0.84 (0.31 to 2.26) 0.95 (0.75 to 1.21) 1.14 (0.43 to 3.06) 1.30* (1.03 to 1.63) 0.73* (0.54 to 0.99) 
  6 mo 0.93 (0.35 to 2.50) 1.06 (0.83 to 1.34) 1.09 (0.41 to 2.91) 1.23 (0.98 to 1.55) 0.86 (0.63 to 1.17) 
  No. of decayed teeth (d2/D2)b      
  Baseline 0.29 (0.19 to 0.45) — 0.29 (0.19 to 0.45) — — 
  3 mo 0.34 (0.21 to 0.55) 1.16 (0.82 to 1.66) 0.47 (0.29 to 0.77) 1.63* (1.11 to 2.39) 0.71 (0.43 to 1.19) 
  6 mo 0.34 (0.20 to 0.55) 1.16 (0.81 to 1.66) 0.51 (0.31 to 0.83) 1.75** (1.18 to 2.58) 0.66 (0.40 to 1.11) 
Outcome and Time PointPT (n = 60)Toolkit (n = 59)
Estimate(95% CI)Effect(95% CI)Estimate(95% CI)Effect(95% CI)Group DifferenceIntervention Effect (95% CI)
Home oral hygiene      
 Frequency of twice-daily tooth brushinga      
  Baseline  —  — — 
   Days 2.8 (2.2 to 3.5) — 2.8 (2.2 to 3.5) — — 
   % 40 (31 to 50) — 40 (31 to 50) — — 
  3 mo — 5.16*** (3.59 to 7.40) — 1.83** (1.27 to 2.63) 2.82*** (1.71 to 4.64) 
   Days 5.4 (4.8 to 5.9) — 3.9 (3.1 to 4.7) — — 
   % 78 (69 to 85) — 55 (44 to 67) — — 
  6 mo — 5.26*** (3.63 to 7.62) — 2.39*** (1.66 to 3.44) 2.20** (1.33 to 3.65) 
   Days 5.5 (4.8 to 6.0) — 4.3 (3.5 to 5.1) — — 
   % 78 (69 to 85) — 62 (50 to 72) — — 
 Severity of behavior problems during oral hygiene activities      
  Baseline 2.89 (2.51 to 3.27) — 2.89 (2.51 to 3.27) — — 
  3 mo 1.71 (1.21 to 2.22) −1.18*** (−1.64 to −0.71) 2.62 (2.12 to 3.11) −0.28 (−0.73 to 0.18) −0.90** (−1.52 to −0.28) 
  6 mo 1.37 (0.86 to 1.88) −1.52*** (−1.99 to −1.05) 2.14 (1.64 to 2.64) −0.75** (−1.21 to −0.29) −0.77* (−1.39 to −0.14) 
Oral health status      
 Visible plaque      
  Baseline 0.84 (0.75 to 0.94) — 0.84 (0.75 to 0.94) — — 
  3 mo 0.68 (0.55 to 0.81) −0.17* (−0.29 to −0.04) 0.87 (0.74 to 1.00) 0.03 (−0.10 to 0.15) −0.19* (−0.36 to −0.02) 
  6 mo 0.75 (0.62 to 0.88) −0.09 (−0.22 to 0.04) 0.80 (0.67 to 0.92) −0.05 (−0.17 to 0.08) −0.05 (−0.21 to 0.12) 
 Full-mouth caries (dmft/DMFT)b      
  Baseline 0.88 (0.33 to 2.33) — 0.88 (0.33 to 2.33) — — 
  3 mo 0.84 (0.31 to 2.26) 0.95 (0.75 to 1.21) 1.14 (0.43 to 3.06) 1.30* (1.03 to 1.63) 0.73* (0.54 to 0.99) 
  6 mo 0.93 (0.35 to 2.50) 1.06 (0.83 to 1.34) 1.09 (0.41 to 2.91) 1.23 (0.98 to 1.55) 0.86 (0.63 to 1.17) 
  No. of decayed teeth (d2/D2)b      
  Baseline 0.29 (0.19 to 0.45) — 0.29 (0.19 to 0.45) — — 
  3 mo 0.34 (0.21 to 0.55) 1.16 (0.82 to 1.66) 0.47 (0.29 to 0.77) 1.63* (1.11 to 2.39) 0.71 (0.43 to 1.19) 
  6 mo 0.34 (0.20 to 0.55) 1.16 (0.81 to 1.66) 0.51 (0.31 to 0.83) 1.75** (1.18 to 2.58) 0.66 (0.40 to 1.11) 

—, not applicable.

P < .10;

*

P < .05;

**

P < .01;

***

P < .001.

a

Binomial model with logit link presents estimates as n (%) of days per week and effects as ORs or intervention effects as ratio of ORs.

b

Poisson model with log link presents estimates as counts and effects as rate ratios or intervention effects as ratio of rate ratios.

In the PT group, parents reported significantly reduced severity of child behavior problems during home oral hygiene activities at both time points. In the toolkit group, parents reported reduced behavior problems at 6 months only. Significant reductions in child behavior problems in the PT relative to the toolkit intervention were reported at both 3 and 6 months.

Children in toolkit group had significantly more caries over time than children in the PT group. At 3 months, group differences in full-mouth caries were significant. Although the toolkit intervention continued to exhibit a greater relative increase in full-mouth caries and decayed teeth over time, differences fell short of significance at 6 months.

Adjustment for baseline differences revealed an identical pattern of results, with a slight reduction in estimated group differences in 3-month caries (Supplemental Table 4).

Neither improved frequency of oral hygiene (VPI, −2% [95% confidence interval [CI], −25% to 21%; P = .86]; dmft/DMFT, −8% [95% CI, −30% to 15%; P = .51]), increased use of an electric toothbrush (VPI, 0.2% [95% CI, −5% to 5%; P = .95]; dmft/DMFT, 0.5%, 95% CI, −9% to 8%; P = .92), nor reduction of behavior problems during oral hygiene (VPI, 22% [95% CI, −19% to 63%; P = .29]; dmft/DMFT, 21% [95% CI, −20% to 61%; P = .32]) significantly mediated 3-month PT effects on plaque and caries. However, the 95% CI for behavior problems included mediation effects that would be considered substantial.

Both PT and a psychoeducational dental toolkit were associated with improved frequency of toothbrushing, but PT was associated with greater relative gains in oral hygiene and a consistent reduction in the severity of interfering child behavior problems. Moreover, PT was associated with improved oral health by reducing plaque and minimizing progression of caries at immediate follow-up, whereas plaque was not reduced, and caries significantly worsened with the toolkit intervention. To our knowledge, this RCT is the first to reveal significant treatment effects on oral hygiene and oral health in children with ASD. Beneficial effects of PT suggest that this approach is viable for addressing one of the most common unmet health care needs of children with ASD: problematic oral hygiene and poor oral health.27 

The PT intervention was numerically superior to the toolkit intervention for all outcomes assessed, which represents a stringent test of PT advantage given the multiple interventions contained in our active comparator. The toolkit was associated with meaningful improvement in frequency of oral hygiene and some reduction of child behavior problems during home dental care. Indeed, toolkit improvements may partly account for the reduced group differences in oral health seen at 6 months. Evidence of meaningful benefits from the relatively low-touch toolkit highlights the need to provide underserved families with access to high-quality, user-friendly psychoeducational materials and basic oral hygiene resources.

One-half of our sample had not visited the dentist in the past year, and 21% had never been to the dentist. For these families in particular, study facilitation of 3 dental visits may have functioned as an additional intervention. Our study did not focus on barriers to preventive dental visits. However, delivery of appropriate and timely access to dental services remains a critical need for children with ASD, which may be remedied in part through adoption of comprehensive care coordination.17 

Poor oral hygiene is posited to underlie oral health problems in children with ASD.6,8  Although mediation results were nonsignificant, CIs included potentially substantial effects for child behavior problems, suggesting the possibility that improved behavior management may reflect a core treatment mechanism. Future trials would benefit from being powered specifically for mediation testing and from using measures with enhanced sensitivity to detect change in quality of oral hygiene. Integrating smart toothbrush technology may assist with measurement precision.

Strengths of the study include our use of a multisite RCT design with an active comparator, blinded dentist evaluation of oral health, and follow-up at 3 and 6 months after baseline. We also demonstrated strong retention of our underserved sample comprising families with Medicaid eligibility. The socioeconomic and ethnic diversity of our sample is rare in RCTs for children with ASD conducted in the United States.65,66  Moreover, the majority of the participating children with ASD presented with comorbid ID, a subpopulation that is strikingly underrepresented in research literature.67 

Consistent with existing studies, we relied on unblinded parent report of toothbrushing frequency. Use of an active comparator minimized potential response bias, but future work would benefit from multimethod measurement and use of a comparator matched for contact and intensity. Previously significant oral health differences also lessened at follow-up, which may suggest the need for greater PT supports during maintenance. Alternatively, long-term follow-up might yield more pronounced treatment effects because caries progression slowed in the PT group and increased in the toolkit group. PT oral health improvements, although statistically significant, were relatively small, and it is possible that augmenting oral health measures would enable detection of larger and more clinically significant effects. To reduce participant burden, dentists used plaque ratings of index teeth to represent full dentition.63  An examination of plaque on all teeth might enhance sensitivity. Finally, the results reveal the efficacy of PT as administered by highly trained therapists with a select population. Community-based replication is needed.

The PT intervention was efficacious relative to a psychoeducational dental toolkit for improving oral hygiene and oral health in underserved children with ASD. The PT intervention also reduced the severity of child behavior problems interfering with home oral hygiene. Given the feasibility and preliminary efficacy of our manualized PT program, we recommend additional study of our novel application of PT to promote oral health in children with ASD, a population at high risk for unmet dental needs.

We thank the members of the clinical coordinating center and data coordinating center at Massachusetts General Hospital, including Lily Altstein, Justin Farmer, Siobhan Glynn, Hilda Gutierrez, Richard Morse, James Perrin, and Brian Winklosky, as well as the members of the Massachusetts General Hospital data safety and monitoring board, including Ann Neumeyer (chair), Amanda Bennett, and Larry Lin. Additionally, we thank project team members at each site, including Charles Astley, Melissa Brown, Breana Cervantes, Jean Gehricke, Taylor Harlan, Haley Kitchens, Anna Laakman, Michele Ocen, Meghan Orr, Erin Roby, and Lisa Tully, and gratefully acknowledge the contributions of Cathy Flaitz, Tanya Matthew, David McAdam, Richard Udin, Konita Wilks, and all the families who made this work possible. This work was conducted through the Autism Speaks Autism Treatment Network serving as the Autism Intervention Research Network on Physical Health.

Drs Fenning and Butter conceptualized and designed the study, developed the treatment curriculum and data collection instruments, supervised data collection, drafted the initial manuscript, and critically reviewed and revised the manuscript; Dr Macklin conceptualized and designed the study, developed data collection instruments, supervised data collection, performed analyses, drafted the initial manuscript, and critically reviewed and revised the manuscript; Drs Norris, Kuhlthau, and Steinberg-Epstein conceptualized and designed the study, developed data collection instruments, supervised data collection, drafted the initial manuscript, and critically reviewed and revised the manuscript; Dr Coury and Ms Hess conceptualized and designed the study and critically reviewed and revised the manuscript; Drs Albright, Guijon, Hammersmith, Scherr, Spaulding, and Stephenson and Ms Moffitt supported development of the data collection instruments, collected data, and critically reviewed and revised the manuscript; Ms McKinnon-Bermingham developed the treatment curriculum, collected data, and critically reviewed and revised the manuscript; Mr Chan performed analyses, drafted the initial manuscript, and critically reviewed and revised the manuscript; Ms Lu supervised data collection and critically reviewed and revised the manuscript; and all authors approved the final manuscript as submitted and agree to be accountable for all aspects of the work.

This trial has been registered at www.clinicaltrials.gov (identifier NCT03003221).

FUNDING: This project was supported by the Health Resources and Services Administration of the US Department of Health and Human Services under cooperative agreement UA3 MC11054–Autism Intervention Research Network on Physical Health. The funding source had no role in the design or conduct of the study; data collection, management, analysis, or interpretation; and preparation, review, approval, or submission of the manuscript. This information or content and conclusions are those of the authors and should not be construed as the official position or policy of, nor should any endorsements be inferred by, the Health Resources and Services Administration, US Department of Health and Human Services, or the US government.

CONFLICT OF INTEREST DISCLOSURES: Dr Macklin reports serving on advisory boards for Biogen, Cerevance, and Stoparkinson Healthcare Systems, serving on data monitoring boards for Novartis and Takeda Pharmaceuticals, and receiving research funds through his institution on his behalf from Amylyx Pharmaceuticals, Biohaven Pharmaceuticals, Clene Nanomedicine, GlaxoSmithKline, Mitsubishi Tanabe Pharma America, Prilenia Therapeutics, Ra Pharmaceuticals, and the National Institutes of Health, and Dr Coury has received research grant support from GW Biosciences, Stalicla, and Stemina and serves on advisory boards for BioRosa, Cognoa, GW Biosciences, MaraBio, Quadrant, and Stalicla; the other authors have indicated they have no conflicts of interest to disclose.

A randomized controlled trial revealed efficacy of a novel parent training intervention for improving oral hygiene and oral health in underserved children with autism spectrum disorder.

     
  • ASD

    autism spectrum disorder

  •  
  • CBCL

    Child Behavior Checklist

  •  
  • CI

    confidence interval

  •  
  • dmft/DMFT

    Decayed, Missing, and Filled Teeth Index

  •  
  • ID

    intellectual disability

  •  
  • PT

    parent training

  •  
  • RCT

    randomized controlled trial

  •  
  • VPI

    Visual Plaque Index

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