Oral health care and service utilisation in individuals with Rett syndrome: an international cross‐sectional study

Abstract

Background There is a dearth of literature available on the comparative oral health status of those with Rett syndrome (RTT) despite diurnal bruxism being a supportive diagnostic criterion for the disorder. This study was designed to investigate the dental experiences of individuals with RTT in terms of perceived at‐home and professional dental care. Methods Using data in the InterRett database, provided by English‐speaking families of individuals with a confirmed MECP2 genetic mutation, the study investigated relationships between dental problems, oral care, child factors including bruxism and use of gastrostomy, and socioeconomic indicators. The study also explored relationships between dental presentations and socioeconomic, child, and family‐related factors. Results Individuals with RTT exhibiting bruxism were more likely to access dental treatment. Those who had full oral feeding had a higher incidence rate of dental treatment than those with full tube feeding. A conservative (under) estimation of the overall dental caries progression rate revealed that this may be similar to that of the normal population. Conclusions Drivers for dental treatment in RTT include bruxism as well as dental caries. Those who have full oral feeding experience more dental treatment than those with full tube feeding. A higher maternal education level may confer a protective effect for oral health outcomes in those with RTT. Nevertheless, families generally tended to value the importance of oral health despite reported difficulties in day‐to‐day mouth care.

Enablers and barriers in dental attendance in Rett syndrome: an international observational study

“…Barriers in the UK to care within the public sector include waiting list issues (14.1%) and cost (5%) 48 . One reason why cost may not have been perceived as a major barrier for most families in the present study was that the families placed a high value placed on oral health and therefore on dental attendance, as noted in a previous study 28 . Another reason why cost may not have been perceived by many as a major barrier for dental access may again be sociodemographic factors.…”

“…24 Diurnal bruxism is the most frequently reported oral presentation, 25,26 and there are various management approaches reported in the published literature in RTT, ranging from tooth restorations for worn or fractured tooth structure to more novel approaches such as bite planes and even acupuncture. 24 While bruxism in RTT has been explored and some studies of the overall oral health experiences of those with RTT have been completed, including oral disease incidence, type and modality of treatment received, 25,[27][28][29] little is known regarding barriers faced by individuals with RTT for accessing timely and appropriate oral health care. An Australian population-based retrospective study 27 found that dental extractions were more likely to have occurred in individuals with RTT living at home in households with a low annual household income.…”

“…[31][32][33] While dental attendance rates may differ for patients with special needs compared with counterparts in the general population, 34,35 it seems likely that maintaining oral health is valued by most individuals with special needs, or by their caregivers. 28,36 As well, oral health status impacts the quality of life of those with disability. 37 Possible barriers to utilising dental services by individuals with disabilities include inadequate training of providers, the challenging behaviours of the individual, 38 inadequate reimbursement and unprofitable care provision due to the additional time required, 39 and practitioner perceptions that dental problems may not be of major importance.…”

“…Those with full oral feeding receive more dental treatment than those with full tube feeding and may be attributed to the differences in both aspiration risk, and in tooth contact time and thereby opportunity for demineralisation to occur in the caries process. Moreover, higher maternal education levels may help drive better oral health outcomes 28 . A gap in past work is that it did not assess the impact of caregiver beliefs, nor did it assess barriers to accessing timely oral health care.…”

Movement disorders in patients with Rett syndrome: A systematic review of evidence and associated clinical considerations

“…Other stereotypies are also prevalent in patients with RTT, and these too can emerge differently, such as bruxism 26,28 and shifting of the weight 34 . Individuals with bruxism were also more inclined to access dental services 43 ; however, the risk of bruxism appears to decline with age 44 . Oral motility was also affected by dystonic and dyskinetic movements 45 .…”