An electronic search took place in September 2020 in the following databases: Embase, PubMed, LiLACS, and EBSCO. The search strategy was adapted to each database with the following key terms and Boolean operators, alone or in combination, where applicable: (conocimientos OR knowledge OR perceptions OR attitudes OR opinion OR education OR understanding OR awareness) AND (caregivers OR parents OR families OR cuidadores OR padres) AND (oral health OR salud oral OR mouth care OR oral hygiene OR oral care) AND (down syndrome OR sindrome de down OR trisomy 21).

Inclusion criteria were primary studies published in English or Spanish that measured the knowledge or perception of oral health of caregivers of people with DS through questionnaires or interviews. There were no limits regarding participants’ age or publication date. Exclusion criteria were publications with no full text available, populations that did not include DS, interventions without an instrument to measure desired outcomes/results, or that did not assess knowledge/perception.

Three members of the research group independently selected the articles, following the inclusion and exclusion criteria. The team compared and discussed the studies, emphasizing research purpose, methodology, population, and main results obtained. Article registration met Arksey and O'Malley’s (12) criteria, to include: authors, title, place of study, purpose, participants, methodology, outcomes measures and main findings.

Figure 1 presents the study selection process using a PRISMA flowchart (13). The electronic search returned 447 results of which we eliminated 12 duplicates, leaving 435 titles for screening. The screening process eliminated 419 articles after reading titles and abstracts for not being relevant to answer the PCC question or because they were not primary studies, giving a total of 16 eligible studies for full-text reading. Then 7 did not have full-text available, did not include any instrument (i.e., interview or questionnaire) to measure desired outcomes, or was not conducted with the specific population with DS looked at in this review. After completing the full-text readings and having identified studies from other sources (i.e., references from articles selected) which added 7 titles. Three of the latter articles were excluded due to duplications and one did not have full text available. The final sample consisted of 12 articles.

FIGURE 1 FIGURE 1 PRISMA Flowchart of the Exploratory Review (13) Source: the authors.

The 12 studies included in the review are in Table 1. The places of the studies included 9 countries: Australia (15), Turkey (16), Belgium (17), Brazil (18,19,20), United States (21), Saudi Arabia (22,23), United Kingdom (24), Sweden (25), and France (26). Most studies were from Europe and one in Brazil.

TABLE 1

(F/M: fathers and mothers. DS: Down Syndrome. yo: years old)

Parents who reported their children had good general health also considered their oral health to be adequate (17,25). In general, most families rated the oral health of their children with DS as good or very good (16,17,19,20,22,25). However, a study (21) showed that formal or paid caregivers outside the family, with a statistically significant difference, have a more negative perception of oral health than family members who play the caregiver role. This could suggest the existence of an emotional-family component in the perception of oral health of relatives with DS. On the other hand, a negative perception was more likely when there were malocclusions or the child was between 4 and 9 years of age, the latter due to a greater need for adult supervision since, in most cases, people with DS cannot brush their teeth autonomously (19). Parents who perceive poor oral health in their children also stated that the clinical examination and dental treatment were difficult procedures to perform (25).

Regarding the greatest concerns of caregivers about oral health issues, these focused on caries, pain, and periodontal disease (19,21,22,24). Oliveira, et al. (20) recognized an important cultural component in which mothers believe that the main cause of caries in their children is the constant consumption of antibiotics due to recurrent infections, which can be common at an early age of a child with DS (5,9).

The caregivers identified in their children the presence of halitosis, inflammation, and bleeding gums (21,22). Likewise, mothers noted that pain affected their children's mood or general behavior, perceiving crying and anger as indicative signs of the presence of dental pain in children (22).

Knowing how to maintain proper oral hygiene is also another major concern for caregivers. This is due to the need to reinforce or brush teeth, given the lack of manual dexterity or intellectual disability that people with DS may have (19,24). In addition, they reported that home oral care routines are interfered with by behavioral problems, lack of time, or lack of sufficient help to carry it out (21).

Caregivers were concerned about oral hygiene and taking their children for frequent dental check-ups, attending at least once a year (16,17,24). The fathers and mothers who participated in those studies reported the need for an annual check-up, presence of caries, pain, being referred by a health professional, or accompanying siblings as the main reasons for visiting the dentist (16,17).

However, this study also found that 24.2% of families had never taken their children to the dentist (16). When examining the justifications for not doing so, it was found that most families thought their children were too young to go, which was consistent with another study by Descamps, et al. (17), in which it was pointed out that children between 2 and 4 years old had never been to the dentist, because their parents considered them too young. Caregivers also reported as reasons for being apprehensive about dental care: assuming that children would be difficult to care for, being unaware of the need to visit the dentist preventively and considering that general health problems were more important than oral health issues (16).

Access to dental care was another issue mentioned by caregivers who reported that they found challenging to find a competent dentist (16,18,26). This affects the frequency of annual visits since they are fewer for not finding a trained professional (26). When comparing the experience of children with DS with that of siblings without this condition, those who did not have this syndrome had a significantly higher probability of having received dental care than the sibling with DS (26). Also, the main dental procedures performed on children with DS were extractions and restorations and that, except for hygiene instruction, very few received preventive care, such as the application of sealants or fluoride varnish (24). In 35% of the families, the child with DS saw the same dentist as her siblings (25) and 57.5% saw the same dentist as their relatives (24). Caregivers also expressed that it would be useful for them to have a list of dentists trained to provide dental care to their children with DS (24).

Finally, caregivers indicated having different dental care experiences; however, the majority (84%) was satisfied with the professional caring for their child (25). In their opinion, it is more important for dentists to be friendly, reassuring, and able to explain procedures before treatment, involving the patient, than someone specialized in treating people with special needs (16,17,24).

Although there are no differences regarding how oral health status impacts the quality of life of families of children with or without DS, the parents of the former perceived that situations such as presenting dental caries and leaving them untreated, do have a negative impact on the quality of life of those around them (18). For example, a poor oral condition can directly affect the mood of mothers who reported feeling guilty and neglectful if their children had dental pain (22). Likewise, poor oral condition, halitosis, or aesthetic compromises (20) could alter the child's quality of life and, consequently, the quality of life of the people who care for them. Mothers saw themselves as the most affected member within the family circle, especially when there is pain involved, indicating that they felt irritated, angry, and depressed, preferring to isolate themselves socially until the child felt better (22). Regarding the latter, problems affecting oral health would constitute an obstacle to the child's social acceptance, since pain or dentomaxillary anomalies associated with alterations in orofacial functions, such as speech, breathing and chewing, can disturb their emotional status, leading to a restriction in family activities and generating conflicts within the family (20,22).

Most caregivers recognized the importance of oral health in chewing, aesthetics, and speech. In addition to being aware that having good oral hygiene is necessary for good general health, they reported having adequate knowledge of dental preventive care and self-care and recognized the influence of a high-sugar diet as the main cause of caries development (23). Likewise, they affirmed the importance of frequent visits to the dentist and brushing teeth, emphasizing this oral hygiene habit as a rule within the home (15,17,23). Regarding the use of fluorides, most caregivers had heard of its role in preventing cavities and only 25% attributed the property of favoring teeth whitening to it. The majority (93.3%) recognized toothpaste as the main source of fluoride and to a lesser extent (20%) drinking water (23).

For the most part, families received information on these topics from a dentist (16,17,21,23,24,25). Other recognized sources of information were teachers, professionals working in rehabilitation centers, and the Internet (16,17,23). Talking with other parents of children with DS was also considered an important way of learning (24,25); even in one study (24) this practice was pointed out as more useful than asking a health professional directly. It was further noted that, unlike unpaid caregivers, formal or paid caregivers commonly receive training and formal apprenticeship through courses (21). Despite this, a study reported that 20 % of parents had never received any type of guidance regarding the oral status of child with DS (17).

Regarding the knowledge acquired, caregivers declared that they have received education on prevention and promotion measures; for example, oral hygiene instruction, dietary counseling, and use of fluorides, as well as information on common oral conditions in DS, including tooth agenesis and delayed tooth eruption (17,25). 47.5 % rated such teaching as appropriate, and the majority stated that it would be useful to have a brochure with advice on oral health problems commonly associated with DS (24).