Oral motor therapy with

palatal plates in children

with Down syndrome
A systematic review

Hanna Svensson
Ida Eriksson

Supervisor: Gunilla Klingberg

Master Thesis in Odontology (30 hp) Malmö University

Program in Dentistry Faculty of Odontology
February, 2017 205 06 Malmö
ABSTRACT
Aim: The aim of this study was to evaluate the effect of treatment with stimulating
palatal plates on the oral motor function in children with Down syndrome. The study
also aims to investigate if the treatment is cost-effective.

Material and Method: The study is a systematic review made according to the
PRISMA criteria. The articles were quality reviewed using Swedish Agency for
Health Technology Assessment and Assessment of Social Services - SBU’s manual.
The databases used for the literature search were PubMed, Cochrane Library, Scopus
and CINAHL.

Result: Screening of 107 unique papers resulted in 14 eligible publications. The

quality of the articles was overall low. Nine articles were rated moderate risk of bias
and 5 articles were rated high risk of bias. All 14 included articles showed a positive
effect in one or more aspects on oral motor function but there is no consensus
regarding evaluation methods for treatment with palatal plates, treatment times or
which orofacial variables that should be investigated. No meta-analysis was made due
to the lack of consensus.

Conclusion: This literature review identified a number of studies, which

investigated the effect of treatment with stimulating palatal plates. Due to the
unstandardized methods, different treatment times, and different orofacial measuring
variables, no consensus can be drawn from these studies. More RCT studies with
larger groups of children and standardized methods for evaluation are required.

2
Oralmotorisk behandling
med gomplattor hos barn
med Downs syndrom
- En systematisk litteraturöversikt

Hanna Svensson
Ida Eriksson

Handledare: Gunilla Klingberg

Examensarbete (30 hp) Malmö högskola

Tandläkarprogrammet Odontologiska fakulteten
Februari, 2017 205 06 Malmö

3
SAMMANFATTNING
Syfte: Syftet med denna studie var att utvärdera effekten av stimulerande gomplattor
på den orala motoriken hos barn med Downs syndrom. Studien syftar också till att
undersöka om behandlingen är kostnadseffektiv.

Material och Metod: Studien är en systematisk litteraturstudie enligt PRISMAs

kriterier och artiklarna kvalitetsgranskades med hjälp SBU: s handbok. De databaser
som användes för litteratursökningen var PubMed, Cochrane Library, Scopus och
CINAHL.

Resultat: Screeningen av 107 unika artiklar resulterade i 14 relevanta publikationer.

Kvaliteten på artiklarna var överlag låg och 9 artiklar bedömdes måttlig risk för bias
och 5 artiklar bedömdes ha hög risk för bias. Alla 14 inkluderade artiklar visade en
positiv effekt på minst en orofacial variabel men det finns ingen konsensus gällande
utvärderingsmetoder för behandling med gomplattor, behandlingstider eller vilka
orofaciala variabler som bör undersökas.

Slutsats: I denna litteraturgenomgång identifierades ett antal studier, som

undersökte effekten av behandling med stimulerande gomplattor. På grund av att
artiklarna använde icke standardiserade metoder, hade olika behandlingstider och
använde olika variabler för att mäta effekten, kan ingen slutsats dras från dessa
studier. Fler RCT studier med större grupper av barn och standardiserade metoder för
utvärdering behövs.

4
INDEX
1. Introduction ................................................................................................................... 6
1.1 Down syndrome................................................................................................................................ 6
1.2 History of “Orofacial regulation therapy” .............................................................................. 8
1.3 Treatment with palatal plates .................................................................................................... 8
1.4 Other oral motor treatments .................................................................................................... 10
1.5 Health economy and priorities in health care .................................................................. 11
1.6 Ethics .................................................................................................................................................. 12
Aim and issue ......................................................................................................................................... 12
2. Material och methods ............................................................................................... 13
3. Results ............................................................................................................................ 15
4. Discussion ..................................................................................................................... 21
4.1 Method discussion ........................................................................................................................ 21
4.2 Material discussion ....................................................................................................................... 22
4.3 Result discussion ........................................................................................................................... 22
4.3 Ethical discussion .......................................................................................................................... 26
5. Importance of project ............................................................................................... 26
6. References..................................................................................................................... 27
Appendix ............................................................................................................................ 31
1. PubMed- search ............................................................................................................................... 31
2. CINAHL – search ............................................................................................................................... 31
3. Scopus – search ................................................................................................................................. 32
4. Cochrane – search ............................................................................................................................ 32
5. Assessment tool - SBU .................................................................................................................. 33
6. Excluded articles .............................................................................................................................. 36
References excluded articles ........................................................................................................... 37

5
1. INTRODUCTION

1.1 Down syndrome

Down syndrome is not a disease - it is a diagnosis. The definition of “syndrome” is:

“a group of signs and symptoms that occur together and characterize a particular
abnormality or condition” (1). Down syndrome is a developmental disability caused
by chromosomal disorder. One of 800-900 neonates is born with Down syndrome.
The degree of intellectual disability varies between different individuals and a child
with the diagnosis can have a more or less characteristic appearance (2). Children
with Down syndrome have a lowered immune system (3). They also entail a greater
risk of medical problems such as congenital heart defects, congenital blockage in the
digestive tract, childhood leukemia, metabolic and endocrine disorders, vision
problems, hearing impairments, sleep apnea and Alzheimer's disease (4).

Some common physical signs include characteristic body posture, a small head,
flattened face, short neck, up-slanted eyes, and sloping underchin. Another
characteristic feature is a single deep crease across the centre of the palm, so called
single transverse palmar crease (2).

It is known that 95 percent of all cases of Down syndrome occurs due to an incorrect
division of sex cells, i.e. before the actual fertilization takes place. The mechanism
itself is still not fully clarified. Approximately 75% of all children with Down
syndrome are born to mothers younger than 35 years old. The largest group of
pregnant women are between 27 and 32 years old and thereby most children with
Down syndrome are born by women in this age (5-7).

1.1.1 Different forms of Down syndrome

Down syndrome can appear in three different forms. Trisomy 21 is the most common
form, in which all of an individual’s cells contain an extra copy of chromosome 21.
That extra chromosome is the result of random, abnormal events in cell division that
occur during embryonic development or during the development of egg or sperm cells
(8).

Mosaic Down syndrome is a more uncommon form of the syndrome. In this form the
person has a mix of some cells containing 46 and some 47 chromosomes. The
affected person will have some of the usual characteristics of the syndrome. How
pronounced the characteristics are is depending on the number of and which cells that
are involved (8).

Translocation Down syndrome is the third form of the disorder and it is the only form
of the disorder that may be inherited. It occurs when the extra chromosome in the 21
pair breaks off and attaches itself to another chromosome (8).

1.1.2 Orofacial features in Down syndrome

In infants with Down syndrome the muscle tone is low and this is very apparent in the
orofacial region. Lip closure is mostly poor and the mouth is often kept open with a

6
protruded tongue resting inactively between the lips (Figure 2a) (2,9). Tongue
diastasis (Figure 1) is not unusual and the lingual frenulum is often hypoplastic. The
upper lip is often narrow and sparsely active, whereas the lower lip is hypotonic,
externally folded and very active. Even the chin muscles are hyperactive and raise the
lower lip, which is particularly clear when swallowing. Even during speech, the lower
lip is more active than the upper lip (10).

The palate may appear small and narrow and with time the hard palate sometimes
develops unfavorably. The palate mucosa hypertrophies and adopts a horseshoe shape
and the palate may seem relatively high in relation to the narrow dental arch. This
could be one reason why children with Down syndrome often show severe
articulation disorders (10).

For some children with Down syndrome nasal breathing is impossible, due to narrow
nasal meatuse (11). This means they have to keep their mouth open to be able to
breathe and can therefore not be treated with palatal plates (10). In many cases
salivation is decreased but because an open mouth and hypotone orofacial muscles,
drooling is common (9).

Figure 1. Tongue diastasis

Photo: Gunilla Klingberg

1.1.3 The oral perspective of Down syndrome

Intraoral features like relative prognathism, hypodontia, microdontia, enamel
hypoplasia, delayed eruption in both dentitions and delayed exfoliation of primary
teeth are more commonly seen in children with Down syndrome than in other children
(7).

Individuals with Down syndrome also have more pronounced periodontal illness than
healthy individuals and the bacteria Actinobacillus actinomycetemcomitans (Aa)
occurs more frequently in subgingival plaque than in healthy control children
(12,13). It seems to be a combination of anatomical differences, mouth breathing,
cooperation difficulty, autonomy and immune system defects (13-15). According to
the systematic review from 2015 made by T.D. Deps, children with Down syndrome
have no increased risk regarding dental caries, possibly even lower risk than healthy
children (16).

7
Individuals with Down syndrome often have a general hypotonia of varying degree
and too agile joints. The temporomandibular joint ligaments are slackened, which
makes the mandible drop and moves forward. It is also common that the oral cavity
appears small and it is most likely a result of that the tongue is displaced. The tongue
can be placed between the alveolar ridges, between the lips or protrude from the
mouth. This makes it hard for the palate to keep its width, which ultimately can lead
to cross bite. Individuals with Down syndrome have because of the characteristics
mentioned often atypical or dysfunctional swallowing pattern with a strong tongue
pressure that contributes to an anterior open bite. These phenomena often contribute
to a negative growth pattern as the jaw drops and moves anteriorly, the mandibular
protrusion leads to angle Class III malocclusion, which is commonly seen in children
with Down syndrome. Later articulation problems (especially labial sound making)
can occur because the tongue is positioned between the front teeth (17).

1.2 History of “Orofacial regulation therapy”

In the 1970s Castillo Morales developed a program to reduce orofacial dysfunction.

The components in "Orofacial regulation therapy" are functional diagnostics and a
special physiotherapy program. The program consists of three parts - orofacial
therapy, body therapy and stimulating plate therapy. The orofacial and body therapy
consist of different types of touch, caress, pressure, elongation and vibration exercises
- to improve mouth and body posture. The objective of “Orofacial regulation therapy”
is a dorsal cranial shift of the tongue, combined with automatic training of the
muscles and stimulation of the inactive upper lip (10).

Figure 2a. Patient with Down syndrome Figure 2b. Same patient with a
without a stimulating plate (9). stimulating plate inserted (9).

1.3
Treatment with palatal plates

There are two main purposes of treatment with palatal plates, 1) improve oral motor
function, and 2) improve articulation. This study will focus on palatal plates for
improved oral motor function.

8
 Figure 3a. Schematic
picture of a child with
hypotonic orofacial
muscles and a protruded
tongue without
stimulating plate (51).
Figure 3b. Schematic
picture of a child with
hypotonic orofacial muscles
and a protruded tongue with
stimulating plate (51).

Palatal plates constitute one part of Castillo Morales concept of orofacial regulation
therapy. The children come to a dentist, usually after referral from a speech and
language pathologist in the department of child and adolescent habilitation.
Indications for stimulation plates are a hypotonic tongue with an interdental or Indicação
interlabial position, a protruded tongue with lingual diastasis, and a hypotonic inactive
upper lip with a position behind the lower lip (Figure 2a and Figure 3a). Possible
contraindications for treatment with palatal plate could be: mouth breathing, if parents
lack compliance, coping problems or if the child has multiple general diseases (18).

An initial assessment is done as early as possible, according to Castillo Morales, Inicio da Terapia
preferably before 6 months of age (10). An impression is taken for manufacturing of a
palatal plate in acrylate. In all, the treatment require many visits (19). The plate
should be extended back to the A-line and have a vestibular designed for good
retention. Palatal plates always have a crater; an elevation of acrylate at the A-line. It
can be round or oval; oval if there is a tongue diastasis - otherwise it is not stimulating Características
the entire area of the dorsal tongue. If there are no diastases, the crater is round da PPM
(Figure 4). The crater creates a reflex in the tongue, which makes the tongue contract
upwards and backwards in the mouth. This promotes contact with the palate and
increases the activity of the tongue (Figure 2b and Figure 3b) (10). There are also
buccal stimulators e.g. ridges for activating of the orbicularis oris muscle.

How long and how often the palatal plate should be used varies between different
studies. In the article by Carlstedt et al. the palatal plates were used 1/2 - 1 hour twice Uso diário
a day (20). When the child grows and teeth erupt the palatal plate should be replaced.
Another alternative is to rebuild on the existing plate. Other stimulators, e.g. metal
knobs or beads can then be used. What type of stimulation to use varies from case to
case, because the children are in need of different stimuli for efficiency (10,21).
Generally metal stimulators are considered to be stronger, which some children may

9
need, to get desired effect (10,21). According to Carlstedt et al. palatal plates should
be used at least during the child's first year (18).

Figure 4. Example of different stimulating plates.

To the left: Stimulating plate for a child without dentition with an oval crater and
vestibular ridges.
To the right: Stimulating plate for a child with dentition with a round crater and
vestibular metal knobs.
Photo: Gunilla Klingberg

1.4 Other oral motor treatments

There are different kind of tools for children who have problems with chewing,
sucking or blowing that can be used for diagnostics as well as oral exercises (22).

An electric toothbrush can be used for sensorimotor stimulation. When an electric

toothbrush applied to the tongue this will result in the tongue moving towards the
stimulus. Thus, the tongue may be drawn upwards and backwards if the stimulus is
applied on the dorsal part of the tongue. The stimulation also aims to develop tactile
stimulation and to raise awareness about the tongues position in the oral cavity (22).

Figure 5. Oral screen. Figure 6. Training with oral screen.

Photo: Gunilla Klingberg Photo: Gunilla Klingberg

10
 Figure 7. Training with oral screen.
Photo: Gunilla Klingberg

Training with an oral screen (Figure 5) can help to strengthen the muscles in lips,
cheeks and pharynx. The objective with oral screen-training is improved lip closure,
improved swallowing and decreased drooling. The training involves trying to keep the
oral screen within the lips while someone is pulling the ring on the oral screen to train
the muscle strength in the orofacial region (Figure 6 and Figure 7) (23).

1.5 Health economy and priorities in health care

The health economic aspects on the efficiency of the health care have a great
importance regarding priority on the clinical and political level. The efficiency in
different methods for diagnostics and treatment are in the health economic defined as:
the relationship between resources and outcomes for patients. This with respect to
healthcare objectives: to relieve and cure, prevent early death, decrease morbidity and
increase or maintain the quality of life (24).

Often the scientific evidence is insufficient or lacking for measuring the performance
of the health care system. A presumption for an assessment of effectiveness is the
knowledge of the benefit of various medical methods, i.e. its effects on the health and
quality of life. Such knowledge is generated primarily through applied clinical
research focused on profit measurement. On the basis of such studies, the cost
effectiveness of different measures are estimated (24).

Information about what various treatments costs can not be the basis for decision-
making when prioritizing treatments. The health economic analysis will be slightly
closer when they are extended to highlight the cost effectiveness - a term referring to
comparisons of costs and the effects achieved for the patient when comparisons
between treatments are made. There are however many difficulties with determining
the cost effectiveness of medical practices. It is in fact dependent on and intimately
connected with clinical research. In order to determine the cost effectiveness it is
essential - in addition to information about cost – also to collect outcome data from
clinical studies. For example, sensitivity and precision in diagnose, therapies effect on
survival and quality of life, as well as their risks and side effects (24).

11
The Swedish government and Ministry of social affairs appointed a commission
inquiry on priorities in health care (SOU 1995:5) The commission proposes three
principles when prioritizing health care, the most important ranked first:
1. The principle of human dignity; all people have equal value and equal rights
regardless of personal characteristics or role in society.
2. Needs and solidarity principle; resources should be invested in the human or
activity that have the greatest needs.
3. Cost-effectiveness principle; when choosing between different activity areas
or actions - a reasonable relationship should be between cost and effect
pursued measured in improved health and quality of life (24).

Those who have the greatest need of care should be prioritized, which make the
principle of human dignity valued higher than the needs and solidarity principle, and
cost-effectiveness principle is subordinate to the other two principles (24). These are
the basis for setting priorities in health care in Sweden and since July 1st 1997, this is
a general priority rule in found in §2 in the Swedish Health and Medical Services Act
1982:763. However, the result of the commission inquiry were never incorporated in
the Swedish Dental Act (1985:125) (25,26).

1.6 Ethics

Research on human beings and animals requires ethical vetting carried out by an ethic
committee. For this study no ethical vetting was undertaken, because it is a literature
review and partly because it is implemented within the framework of undergraduated
education. The study was performed according to Malmö University's criteria for
master thesis. The scientific papers used in the project are publicly available
materials. Articles have been reported objectively and reproduced without being
altered or defaced (27).

According to “the Convention on the Rights of the Child” the best interests of the
child must be a primary consideration in all decisions affecting the child (28). Further,
it is difficult to know what treatment is preferable according to the principle “best
interest” when there is limited knowledge about treatment outcome. Therefore it is
important to accomplish this project from an ethical perspective.

Aim and issue

The aim of this study was to evaluate the effect of treatment with stimulating palatal
plate on the oral motor function in children with Down syndrome. The study also
aimed to investigate if the treatment is cost-effective.

The following research questions were adressed:

• Does treatment with palatal plates improve oral motor function in children
(<18 years) with Down syndrome?
• Is treatment with palatal plates cost-effective?

12
2. MATERIAL OCH METHODS
A PICO was constructed:
Population
· Children (0-18 years)
· Individuals with the diagnosis Down Syndrome
· All ethnical groups
· Both genders
Intervention
· Treatment with palatal plates
Comparison
· No treatment with palatal plates
Outcome
· Oral motor function

A systematic literature search was conducted in PubMed (30th October 2015),

CINAHL (9th November 2015), Scopus (8th February 2016) and Cochrane Library
(15th February 2016) by the authors with help from scientific information specialists at
Malmö University, Faculty of Odontology. The literature search was performed
according to the PRISMA-statement, to study the quality and relevance of the articles
in a standardized way (29). The search was made using one population block and one
intervention block with as many synonyms in each block as possible to create a wide
search. There were no language restrictions. In PubMed MeSH-terms and keywords
were used and in CINAHL, Scopus and Cochrane Library, relevant keywords were
used. Complete search strategies are presented in Appendix 1, 2, 3 and 4.

The database search resulted in 164 articles in total but after removing the duplicates,
106 unique papers reamined. A hand search was conducted by the authors (HS, IE)
and this search resulted in one new unique article. In all, the literature search resulted
in 107 papers. The papers abstract were individually assessed by the two authors (HS,
IE) according to relevance of the thesis. After the individual assessment, all abstracts
were discussed by the authors (HS, IE) until consensus were reached. A total of 28
articles were decided to be read in full text and were treated according to following
criteria:

Inclusion criteria:
· Children (0-18 years)
· RCTs with control group, observational studies, controlled trials, cohort
studies
· Down syndrome
· Treatment with stimulation palatal plate
Exclusion criteria:
· Adults (>18 years)
· Articulation palatal plates
· Pilot studies and reviews

This process resulted in 14 unique included papers in this study. Any differences were
resolved by consensus discussion and when necessary, a third inspector (GK) was
consulted.

13
A total of 14 full text papers were finally assessed regarding quality using Swedish
Agency for Health Technology Assessment and Assessment of Social Services (SBU)
assessment tool “Mall för kvalitetsgranskning av observationsstudier” (Appendix 5)
(30). With this tool each article was assessed independently by the authors (HS, IE) in
six different sections. If the bias in a particular section was unclear, it was discussed
with a third inspector (GK), and a decision was reached as to whether the section
should be classified as having a low, moderate or high risk of bias.

The following criteria were used to determine the overall risk of bias: To get a low
risk of bias in total, it was required low risk of bias in the majority of the sections
(four sections or more). The article was classified as a high risk of bias in total if half
of the sections were rated “high risk of bias” (3 sections or more). The article was
assessed as moderate risk of bias, if it did not reach the criteria for low or high risk of
bias. Some articles had no control group, therefore they could not be assessed in
section A1 and A2, these articles could only be estimated in four sections.

Figure 8. Flow chart of work process.

The full process is presented in a flow chart (Figure 8). Excluded papers are reported
in Appendix 6.

An additional literature search was made containing the first search along with a new
subject in each of the four previously used databases. The new subject consisted of
terms associated with health economic, economy and cost-effectiveness. This search
resulted in zero articles (N=0) regardless of which database that were used.

14
 3. RESULTS
Screening of 107 unique papers resulted in fourteen eligible publications and the risk
of bias was assessed. The quality of the articles was overall low. No papers met the
criteria for low risk of bias. Nine articles were rated moderate risk of bias and five
articles were rated high risk of bias (Table 1). All 14 included articles reported a
positive effect of treatment (Tables 3 and 4) but there were no consensus regarding
evaluation methods for the treatment with palatal plates, treatment times or which
orofacial outcome variables that should be investigated. No meta-analysis was made
due to this lack of consensus. The 14 publications represented 8 study populations, as
there were several publications based on one or more longitudinal follow up of
treatment in the same study population (18,20,31-38).

Table 1. Risk of bias

Author, year Selection Performance Detection Attrition Reporting Conflict of Risk of

bias bias bias bias bias interest bias
A1 A2 A3 A4 A5 A6 (Summed)

B. Bäckman et al., 2003

B. Bäckman et al., 2007
K. Carlstedt et. al., 1996
K. Carlstedt et. al., 2001
K. Carlstedt et. al., 2003
K. Carlstedt et. al., 2007
H. Fischer-Brandies, 1988

E. Glatz- Noll et. al. 1991

A. Hohoff et. al. 1997

H. M. Korbmacher et. al. 2002

H. M. Korbmacher et. al. 2006

G. J. Limbrock et.al. 1991

G. J. Limbrock et. al. 1993

G. Schuster et. al. 2000

Low risk Moderate risk High risk Not applicable

15
Table 2. Risk of bias according to section

A1 4 4 6

A2 5 3 6

A3 6 8

A4 4 10

A5 1 10 3

A6 13 1

Low risk Moderate risk High risk Not applicable

16
Table 3. Results table of the included articles with a control group (articles
with moderate or high risk of bias).

17
 Table 4. Results table of the included articles without a control group
(articles with moderate or high risk of bias).

In B. Bäckman et al. from 2007 the treatment effect was evaluated by follow up with
a clinical examination and video registration. This article is a follow up study on B.
Bäckman et al. from 2003 and uses the same patient material. Evaluation showed that
treatment with palatal plates had a positive effect of oral motor function. The article
was rated moderate risk of bias because the article report a reasonably low drop out
(41% test group and 32% control groups), but used unstandardized methods and the
groups had large individual variations. The reliability of the result is valued higher
thanks to their control groups and that the dentists who performed the evaluation in
cases of deviance discussed the case until consensus was reached (32).

K. Carlstedt et al. from 1996 found that the test group had significantly longer period
of “closed mouth” (p<0,001) and significantly shorter “inactive protrusion of the
tongue” (p<0,001). The result was evaluated after 12 months by video registration.
The article was rated moderate risk of bias because the patients were randomized into
control and treatment group but used unstandardized methods and the groups had
large individual variations. The reliability of the result is valued higher thanks to their
control group and that the dentists who performed the evaluation were calibrated (20).

K. Carlstedt et al. from 2001 is a longitudinal follow up on the study population from
K. Carlstedt et al. from 1996. Reported that the test group had significantly longer
period of “closed mouth” and significantly shorter “inactive protrusion of the tongue”
(p<0,01). The result was evaluated 49-58 months after treatment by video registration.
The article was rated moderate risk of bias because the patients were randomized into

18
control and treatment group but used unstandardized methods and the groups had
large individual variations. The reliability of the result is valued higher thanks to their
control group and that the dentists who performed the evaluation were calibrated (33).

In K. Carlstedt et al. from 2003 the treatment effect was evaluated by a parental
questionnaire and video recording after 49-58 months of treatment. The test group had
significantly longer period of “closed mouth” and significantly shorter “inactive
protrusion of the tongue” and significantly more “closed mouth” during non speech
time (p<0,05). This article is a follow up study on K. Carlstedt et al. from 1996 and
uses the same patient material. The article was rated moderate risk of bias because the
patients were randomized into control and treatment group but used unstandardized
methods and the groups had large individual variations. The reliability of the result is
valued higher thanks to their control group and that the dentists who performed the
evaluation were calibrated (34).

In K. Carlstedt et al. from 2007 the treatment effect was evaluated by a parental
questionnaire and video recording after 49-58 months of treatment. The study showed
that the treatment group had significantly less “inactive muscle function” even after 4
years. They found no significant difference between the groups at “inactive protrusion
of tongue” and “closed mouth” at follow-up. This article is a follow up study on K.
Carlstedt et al. from 1996 and uses the same patient material. The article was rated
moderate risk of bias because the patients were randomized into control and treatment
group but used unstandardized methods and the groups had large individual
variations. The reliability of the result is valued higher thanks to their control group
and that the dentists who performed the evaluation were calibrated (18).

B. Bäckman et al. from 2003 found that palatal plate therapy in combination with oral
motor and sensory stimulation had a positive effect concerning oral motor
performance. The result was evaluated by clinical examination, video registration and
by interviewing parents. The article was rated high risk of bias partly because the
article does not report any information about drop out, used unstandardized methods
and had large individual variations in the groups. The reliability of the result is
however valued higher thanks to their control groups and that the dentists who
perform the evaluation were calibrated (31).

In E. Glatz-Noll et al. from 1991 the treatment was evaluated by video registration. 12
of 24 children with Down syndrome reached normalization of tongue function, but no
improvement in mouth closure after treatment. The study had a control group with 19
healthy children. At follow up 5 of 7 children in treatment group was assessed to be
stable. The study was rated as high risk of bias because it is unclear how the
recruitment of patients was made, had a large drop out (from originally 24 to 7
patients), the article used unstandardized methods and the groups had large individual
variation. The reliability of the result is valued lower because it has a control group
consisting of healthy children and a wide range of ages (2 months- 12 years) (39).

In G. Schuster et al. study from 2001, the treatment effect was evaluated by follow up
with a clinical examination and parental questionnaire. The article concluded 65%
improved “lip closure” in treatment group compared to 8% improvement in control
group at follow up (unclear significance). 55% of the children in the palatal plate
group showed improved “tongue position” compared to 24% improvement in the

19
control group. The study was rated as high risk of bias partly because the large drop
out (from originally 85 to 33 patients), reported no statistics, used unstandardized
methods and the groups had large individual variations. The reliability of the result is
valued higher partly because it has a control group, even though is a retrospective
study (40).

H. Fischer-Brandies et al. from 1988 found that treatment with palatal plates in most
cases improved orofacial function. A clinical examination and a questionnaire
evaluated the result. Since the article had no control group it was not possible to asses
area A1 and A2 in the quality review. The article was rated moderate risk of bias
partly because there was no conflict of interest but large drop out (from originally 127
to 56 patients), used unstandardized methods and the groups had large individual
variations. The reliability of the result is valued lower because it is an observational
study and it has no control group (41).

H. M. Korbmacher et al. from 2004 showed improved orofacial motor function after
treatment, especially in the children with severe orofacial findings. The result was
evaluated by clinical examination and parental questionnaire. This article and the
article by H. M. Korbmacher et.al. from 2006 use the same patient material but are
two different publications. Since the article had no control group it was not possible to
asses area A1 and A2 in the quality review. The study was assessed moderate risk of
bias partly because there was no declaration of conflict of interest but large drop out
(from originally 102 to 20 patients), used unstandardized methods and the groups had
large individual variations. The reliability of the result is valued lower partly because
it is a retrospective study and because it has no control group (35).

H. M. Korbmacher et al. from 2006 showed improved orofacial motor function after
treatment, the result was statistically significant from first visit to end of stimulating
plate therapy (p=0,038) but not statistically significant from end of stimulating plate
therapy to follow up examination (p=0.283). The result was evaluated by clinical
examination and parental questionnaire. This article and the article by H. M.
Korbmacher et.al. from 2004 use the same patient material but are two different
publications. Since the article had no control group it was not possible to asses area
A1 and A2 in the quality review. The study was assessed moderate risk of bias partly
because the there was no declaration of conflict of interest but large drop out (102 to
27 patients), used unstandardized methods and the groups had large individual
variations. The reliability of the result is valued lower partly because it is a
retrospective study and because it has no control group (36).

G. J. Limbrock et al. from 1991 found a significant positive result in spontaneous

tongue position and mouth closure. Since the article had no control group it was not
possible to asses area A1 and A2 in the quality review. The result was evaluated by
clinical examination. It was rated moderate risk of bias because the article report a
reasonably low drop out (25%), but used unstandardized methods and the groups had
large individual variations. The reliability of the result is valued lower partly because
it is a retrospective study and it has no control group (37).

A. Hohoff et al. from 1999 showed that early treatment with palatal plates improved
mouth closure in children with trisomy 21. The result was evaluated by interviewing
parents and a clinical examination and the result was highly significant. Since the

20
article had no control group it was not possible to asses area A1 and A2 in the quality
review. The study was assessed high risk of bias because it had a large drop out (47 to
18 patients), the article used unstandardized methods and the groups had large
individual variation. The reliability of the result is valued lower partly because it has
no control group (9).

G. J. Limbrock et al. from 1993 uses partly the same patient material as G. J.
Limbrock et al. from 1991. The treatment effect was evaluated by interviewing
parents and a clinical examination. The article concluded improved mouth and tongue
posture after treatment. Since the article had no control group it was not possible to
asses section A1 and A2 in the quality review. The study was rated as high risk of
bias partly because the article does not report any drop out, used unstandardized
methods and large individual variations in the groups. The reliability of the result is
valued lower partly because it is a retrospective study and it has no control group
(38).

There is yet no study published regarding the cost-effectiveness of treatment with

palatal plates on children with Down syndrome.

4. DISCUSSION
This literature review identified a number of studies, which investigated the effect of
treatment with stimulating palatal plates. Due to the unstandardized methods,
different treatment times, and different orofacial measuring variables, no consensus
can be drawn from these studies. More RCT studies with larger groups of children
and standardized methods for evaluation are required.

4.1 Method discussion

The systematic literature review was made according to the PRISMA criteria. Four
databases were used. PubMed and Scopus were used since they are wide and
recognized medical databases. To supplement the search the databases Cochrane
Library and CINAHL were used, to make sure no relevant material were omitted. To
search in multiple databases increases the quality of a systematic review according to
both PRISMA and Amstar (29,30). The search was made using two blocks, one
population block and one intervention block with as many synonyms in each block as
possible to create a wide search. For the same reason no language restrictions were
used.

The age range 0-18 years was selected because it is the age that a person is considered
as a child and the age of 18 you become an adult in several parts of the world. Prior
this project no consensus was found on the optimal treatment age with palatal plates.
By putting the age 0-18 years no studies on children were excluded.

The aim of the systematic review was to answer the question if palatal plate therapy
increased oral motor function. Oral motor function is a relatively comprehensive term
and can be measured in many different ways. Articles that were included in this study,
measured for example: closed mouth, open mouth, tongue position, lip closure and
active- and inactive muscle function. If even stricter inclusion criteria had been used

21
regarding orofacial variables, even fewer articles would have met the inclusion
criteria. Therefor, there were no restrictions concerning type of measurements for
evaluating oral motor function in the inclusion criteria. This was accepted although it
made results from different studies hard to compare due to the use of different
variables.

The articles were quality assessed using SBU’s manual (Appendix 5), which is a
standardized tool for evaluation of scientific literature in order to determine the risk of
bias. Generally, the quality of the articles in this project were not rated particularly
high from a scientifically point of view.

4.2 Material discussion

In the assessment tool from SBU, the sections on selection bias (A1) and performance
bias (A2) could not be used in 6 articles, as these studies had no control groups.
Regarding detection bias (A3), almost all the articles were rated high risk of bias. It
was mainly because there was no standardized method for treatment with palatal
plates or evaluating palatal plate therapy. Unstandardized and unvalidated methods
made it difficult to review if the result was reliable, for the specific study, as well as
when comparing the results from different articles. Also it was rarely defined if the
examiners were blinded or not, which also affected the risk of detection bias.

Among the articles in this scientific material, the risk of attrition bias (A4), was
generally high. Four articles reported no data on drop outs, and the 10 papers which
reported data on this matter had a relatively high loss of 41-81%.

The section regarding conflict of interest (A6) had a particularly important role when
it came to the articles that had no control group in their studies. This because two of
the sections (A1 and A2) concerning selection and treatment, can not be evaluated
without a control group. The remaining four sections thus got an extra significant role,
and since almost all the articles were rated low risk of bias regarding ‘conflict of
interest’ this section acted as a ‘balance of power’. This is a disadvantage of this
study, but also reflects the problems related to the quality of the included papers.

4.3 Result discussion

This systematic review has shown that there is no consensus regarding evaluation
methods for treatment with palatal plates, treatment times or which orofacial variables
that should be standardized as a measuring method. All included articles in this study
indicated that after completion of treatment, the orofacial function had improved, but
if this effect could be attributed to the treatment or just normal growth and
development remains to be further investigated.

The most frequently used methods in the articles in this study were clinical
examination, video registration and parental questionnaire, and as mentioned before,
none of these methods are standardized. Clinical examination and video registration
need to be evaluated to become objective and standardized methods. Those methods
would be even more credible if the examiners were blinded, trained and calibrated

22
and had good agreement between each other. Also a standardized protocol for
parental questionnaires could be developed, but since parents often tend to overrate
the improvements in their own child, parental questionnaires could be quite
subjective. According to Carlstedt et al. questionnaires could be a valuable
complement to clinical examination since they provide information about efforts and
benefits (18). It is important to develop more accurate, reproducible and measurable
methods for examination and evaluation of orofacial function.

Down syndrome is a diagnos with large individual variations, which mean there can
be a wide spectrum of diverse features, aspects or clinical symptoms in different
individuals within the same single diagnosis. A population with Down syndrome
diverge more than a similar group of people without disability. One person with
Down syndrome can have pretty small issues living with the diagnosis, while another
person with the same diagnosis may have multiple illnesses and severe cognitive
problems. This affects individuals both within treatment group and/or control group.
This in combination with small study populations of children with Down syndrome
makes it difficult to transpose the result to other populations with the same diagnosis.
None of the included articles in this review discuss these large individual varions.
None of the included articles in this review discuss these large individual variations
between different children that is characteristic to the diagnosis Down syndrome.

There is no consensus regarding when spontaneous mouth closure generally occurs in

healthy children. According to Glatz-Noll et al. healthy children under the age of
three years and nine months keep their lips closed only ⅙ of registration time (39).
Two of the most common screening tools for orofacial development, ORIS and
NOT-S are created for children over three years (42,43). If spontaneous mouth
closure occurs before the age of three, this is difficult to detect using the assessment
tools that are available today. Since there is no consensus when spontaneous mouth
closure occurs in healthy children, it is difficult to measure the parameter ’increesed
mouth closure’ after treatment with palatal plates in children with Down syndrome.

Six articles (9,35-38,41) which represented 4 different patient materials lacked control
group, eight articles (18,20,31-34,39,40) which represented 4 different patient
materials had a control group. Only one patient material had a randomized control
group (18,20,33,34). Without an untreated control group, it is impossible to
distinguish treatment effects from developmental effects, which is confirmed by
Korbmacher et al (36).

Three of the papers used a healthy untreated population as control group (31,32,39). It
can seem to be a good way to control and evaluate a treatment, but it also has several
disadvantages. Healthy kids may for an exempel have another developmental curve.
Conclusions about the effectiveness of a treatment can not be ensured without a
corresponding control group.

The risk with not using randomization for control group is that the untreated Down
syndrome children in the control group may have different contraindications for
treatment. For example mouth breathing, lack of compliance, coping problems with
the palatal plate or if the child has multiple general diseases. It is a risk that the
children with most pronounced problems related syndrome could be included as
controls. These children should not be in control groups since they can not be directly

23
compared to the treated children. These children may for example not have the
physiological abilities to close the mouth. The consequence of including these
children as controls when investigating treatment outcome could be an unreliable
result with false positive impression of successful treatment.

The results of this systematic review do not answer the complex issue about optimal
time for treatment start or how long the treatment duration should be, further research
of good quality is required also in this area. According to Castillo-Morales an early
treatment is essential for a normal oral motor function in children with Down
syndrome (10). The ages in the articles of this systematic review were widly spread;
from two months to twelve years. Future studies should include more patients but also
be designed to enable comparisons in different age groups for more reliably
conclusions regarding the optimal management and treatment times.

Even though more RCT studies are needed, these are hard to accomplish. From an
ethical perspective it is difficult to conduct randomized treatment studies on humans,
especially children. It is also difficult to perform longitudinal studies on children with
learning disabilities because the studies are time consuming and it is not uncommon
with high out rates in this group of patients (44,45).

Carlstedt concludes that palatal plate therapy has a positive effect on ‘inactive
protrusion of tounge’ and ‘closed mouth’, particularly during the first year of therapy.
At the same time the result indicate no significant difference between the groups at
follow-up four years later. The untreated children had caught up with the treated
children and were now equal regarding ‘inactive protrusion of tongue’ and ‘closed
mouth’ (18). This demonstrates another important aspect. Children with DS may
reach the same results as healthy children; they only develop their oral motor function
later. Treatment with palatal plate would in that case only expedite the normal
development. Therefore it is questionable if the profits of earlier development have
any positive effect for the individual or if the time a palatal plate treatment requires,
could be used for something else important for the child.

Carlstedt debates about the fact that although the duration of ‘closed mouth’ increases
in children in treatment group, the duration of an ‘open mouth’ did not (18). One
possible reason may be that children with Down syndrome have narrow nasal
meatuses and therefore have a high frequency of mouth breathing which leads to an
open mouth. If there is a high frequency of mouth breathers in the study the success
rate will not be particularly high if you look at the variable ‘open mouth in rest’, due
to the person in question does not have the physiological ability to breathe through the
nose.

The support and interest of the parents is a crucial factor throughout treatment. In
Carlstedts study most parents hade no or minor problems with the child’s compliance.
Reported cooperation- and retentions problems often were associated with tooth
eruption. To be in an obstinate stage of development and upper respiratory infections
were other reasons for problem with compliance (18). G. Schuster et al. also mention
that most children learn to remove the plates themselves during tooth eruption when
retention is compromised, which could affect the compliance (40).

24
Korbmacher et al. found that children with more severe orofacial disorders had better
improvement or treatment effect. Several different factors were discussed, for
example more severe orofacial problems could make the physiotherapist more
motivated and the parents spending even more time everyday on the palatal plate
therapy (36).

To determine whether treatment with palatal plate is cost-effective or not, the cost
needs to be assessed in relation to the potential additional benefits. This systematic
review suggests that treatment with palatal plates in children with Down syndrome
may have a positive effect on oral motor function, but more studies in this area are
necessary to ensure the result. Spending time and resources on a treatment with
questionable efficacy, for treating a minor severely condition is not justifiable.

Children with Down syndrome and their parents already have a lot of contact with
health care and dental care. Instead, focus should perhaps be on spending time and
resources on quality of life stressors. Maybe for example, prevention of periodontitis,
which is proven to have a higher incidence in children with Down syndrome and
ultimately can lead to tooth loss. This would be more accurate from both ethical and
health economic perspective and long term would benefit the child, the parents and
the community. This reasoning is supported by the systematic review by Ferreira et al.
which concludes that “early introduction in periodontal care, participation of
parents/caregivers/institutional attendants, frequency of attendance and association
with chemical adjuvants (independently of the periodontal treatment adopted) seems
to improve periodontal outcomes in preventive and periodontal treatment of DS
patients” (46).

Dental care is generally financed by the individual, but since children in Sweden have
free dental care the relevant question of cost-effectiveness is a matter for the society.
Society's resources are limited which means the money must be spent carefully. The
Dental and Pharmaceutical Benefits Agency (TLV) has written a handbook about the
Swedish dental care system. It says that the care that is subsidized should be cost and
socioeconomically efficient (26).

Health economic evaluations aims to provide decision makers with information about
the cost-effectiveness in different treatments, so limited resources can be used
efficiently. It is therefore not primarily about saving resources, but to strive to get as
much health as possible based on limited resources.

Direct health care costs due to dental disease has been estimated to be 4.6 per cent of
global health care costs (47). In Sweden, the turnover of dentistry is about 25 billion
SEK per year (26). Many treatments and preventive programs today are performed
although both evidence for efficacy is weak and studies on cost-effectiveness are
lacking. For example, there is lack of health economic evidence in all of the measures
included in the National Guidelines for Adult Dental Care (48). One reason is that
there is a need for additional method development regarding methods of cost-
effectiveness in dental care. Additionally, there are no National Guidelines regarding
Child and Youth Dental Care, Dental Care for the Elderly or Dental care for persons
with disabilities or chronic illnesses (48).

25
Many therapies in dentistry have long-term consequences for dental health and future
costs. To make relevant health economic evaluations there is need for well-developed
simulation models that can analyze the costs and effects over a long period of time,
combining sources from different areas and take account of uncertainty. Probably, the
requirements for proven cost efficiency of treatments in dentistry will in the future
become increasingly similar to those that is applied to other health care today.
Overall, this will make better use of public resources for dental care, with improved
dental health as a result (49).

4.3 Ethical discussion

According to Nilstun an ethical analysis could be made to assess the ethical principles
of autonomy, beneficence-maleficence and justice for all concerned, i.e. the child, the
parents, the dentist and society. This is not a decision-making model but a tool to
identify and analyze diversity of ethical issues and weigh the ethical gains against
ethical losses. All this requires a good knowledge of the current problem situation, the
possible course of action and their likely consequences (50). In this case, we do not
know with certainty what effect of treatment with palatal plate on oral motor function
in children with Down syndrome. This fact makes it difficult to asses an ethical
analysis. This leads to difficulties for the therapist to know how to respond to parents
desire to perform treatment with uncertain effect or how to meet lack of motivation.

5. IMPORTANCE OF PROJECT
This project is important in the decision making whether or not to treat children with
Down syndrome with palatal plates. It’s important from a health economic
perspective as well as from a treatment efficiency perspective.

Today all children born with Down syndrome in Sweden are offered treatment with
palatal plates. This despite there is currently no scientific consensus when it comes to
the treatment and no systematic reviews have been published in this subject jet.

Treatment with palatal plates requires several healthcare visits and many
professionals are involved. If the treatment lacks efficiency, this is an unjustified
social cost. Moreover, the treatment is an additional burden on the individual and
family that already have a lot of contact with the healthcare system. Therefore it is
very important to evaluate the effect of treatment with palatal plates.

26
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APPENDIX
1. PubMed- search

PubMed
30 October 2015
Search Query Items
#1 Down syndrome 31329
#2 Trisomy 21 32319
#3 Stimulation plate 2662
#4 Stimulation plates 1136
#5 Palatal plate 930
#6 Palatal plates 439
#7 Orthodontic appliances 20917
#8 Orthodontic appliance 22162
#9 Orofacial regulation therapy 78
#10 Dental appliance 5444
#11 Dental appliances 40883
#12 #1 OR #2 32592
#13 #3 OR #4 OR #5 OR #6 OR #7 OR #8 OR #9 OR #10 58752
OR #11
#14 #12 AND #13 59

2. CINAHL – search

CINAHL
9 November 2015
Search Query Items
#1 Downs syndrome 3810
#2 Trisomy 21 258
#3 Stimulation plate 9
#4 Stimulation plates 9
#5 Palatal plate 31
#6 Palatal plates 31
#7 Orthodontic appliances 1558
#8 Orthodontic appliance 1558
#9 Orofacial regulation therapy 6
#10 Dental appliance 49
#11 Dental appliances 49
#12 #1 OR #2 3892
#13 #3 OR #4 OR #5 OR #6 OR #7 OR #8 OR #9 OR #10 1617
OR #11
#14 #12 AND #13 11

31
3. Scopus – search

Scopus
8 February 2016
Search Query Items
#1 Down syndrome 47158
#2 Downs syndrome 285
#3 Trisomy 21 10637
#4 Stimulation plate 5248
#5 Stimulation plates 5248
#6 Palatal plate 812
#7 Palatal plates 812
#8 Orthodontic appliances 18584
#9 Orthodontic appliance 18584
#10 Orofacial regulation therapy 33
#11 Dental appliance 10655
#12 Dental appliances 10655
#13 #1 OR #2 OR 3 51661
#14 #4 OR #5 OR #6 OR #7 OR #8 OR #9 OR #10 OR #11 26288
OR #12
#15 #13 AND #14 65

4. Cochrane – search

Cochrane Library
15 February 2016
Search Query Items
#1 Down syndrome 1795
#2 Downs syndrome 105
#3 Trisomy 21 136
#4 Stimulation plate 69
#5 Stimulation plates 32
#6 Palatal plate 45
#7 Palatal plates 22
#8 Orthodontic appliances 1026
#9 Orthodontic appliance 775
#10 Orofacial regulation therapy 8
#11 Dental appliance 669
#12 Dental appliances 856
#13 #1 OR #2 OR 3 1910
#14 #4 OR #5 OR #6 OR #7 OR #8 OR #9 OR #10 OR #11 1696
OR #12
#15 #13 AND #14 30

32
5. Assessment tool - SBU

33
34
35
6. Excluded articles
Author
Alacam, A. et al., 2007 (1)
Carneiro, V.L et al., 2013 (2)
Chapman, E. et al., 1983 (3)
Fischer-Brandies, H. et al., 1984 (4)
Fischer-Brandies, H. et al., 1985 (5)
Graziani,G. et al., 1988 (6)
Hoyer, H. et al., 1990 (7)
Kelly, G. et al., 2013 (8)
Korbmacher, H. et al., 2003 (9)
Matthews-Brzozowska, T. et al., 2015 (10)
Padró S.M.J. et al., 2010 (11)
Pritchard, M. Kelly. et al., 2013 (12)
Scholz, F. et al., 1983 (13)
Zavaglia, V. et al., 2003 (14)
Reason for exclusion
DS children not specified in the result.
Pilot trial
No experimental study.
Poor described method.
No comparison.
No experimental study.
Poor described method.
No comparison.
No experimental study.
No comparison.
No experimental study.
No comparison.
No experimental study.
Poor described method.
No comparison.
Review
No experimental study.
Poor described method.
No comparison.
Does not response to the aim.
Pilot trial
Review
No experimental study.
Poor described method.
No comparison.
Does not response to the aim.
Poor described method.
36
References excluded articles

(1) Alacam A, Kolcuoǧlu N. Effects of two types of appliances on orofacial

dysfunctions of disabled children. British Journal of Developmental Disabilities
2007;53:111-123.

(2) Carneiro VL, Sullcahuamán JAG, Fraiz FC. Use of palatal plate and orofacial
development in a child with Down's syndrome. Rev Cubana Estomatol 2013;49:305-
311.

(3) Chapman E, Fischer-Brandies H, Stahl A. Preliminary results of functional early

treatment on the improvement of maxillofacial relations in small children with
Down's syndrome. Fortschr Kieferorthop 1983;44:452-456.

(4) Fischer-Brandies H, Avalle C, Renner B, Schmid RG. Early functional

orthodontic treatment of orofacial developmental disorders in children with Down's
syndrome. Monatsschr Kinderheilkd 1984;132:620-621.

(5) Fischer-Brandies H, Junker N. Theory and practice of functional orthodontic

treatment in infants and young children with Down's disease. L" Orthodontie
francaise 1985;56:365-369.

(6) Graziani G, Graziani G. The Castillo-Morales palatal plate. Dent Cadmos

1988;56:103-105.

(7) Hoyer H, Limbrock GJ. Orofacial regulation therapy in children with Down
syndrome, using the methods and appliances of Castillo-Morales. ASDC J Dent Child
1990;57:442-444.

(8) Kelly G, Pritchard M, Thompason S. The use of orofacial regulation therapy,

including palatal plate therapy, in the management of orofacial dysfunction in patients
with Down syndrome. J Disabil Oral Health 2013;14:15-24.

(9) Korbmacher H, Berndsen K, Berndsen S, Kahl-Nieke B. Presentation of a

modified oral stimulation plate system for children with Down syndrome. Forum
Logopadie 2003;17:14-16.

(10) Matthews-Brzozowska T, Cudziło D, Walasz J, Kawala B. Rehabilitation of the

orofacial complex by means of a stimulating plate in children with down syndrome.
Advances in Clinical and Experimental Medicine 2015;24:301-305.

(11) Padró S. MJ, Barraza V. E, Brücher S. C, Concha T. E, Delgado V. Effectiveness

of the use of palate plates and orofacial stimulation in the oral development of
children with down's syndrome. Rev Chil Pediatr 2010;81:46-52.

(12) Pritchard M, Kelly G, Thompson S. A critical appraisal of the literature on

palatal plate therapy for orofacial dysfunction in patients with Down syndrome. J
Disabil Oral Health 2013;14:5-14.

37
(13) Scholz F, Arnold J, Chapman E. Early treatment of tongue and lip dysfunctions
with removable appliances in Down's syndrome. Fortschr Kieferorthop 1983;44:54-
56.

(14) Zavaglia V, Nori A, Mansour NM. Long term effects of the palatal plate therapy
for the orofacial regulation in children with Down syndrome. J Clin Pediatr Dent
2003;28:89-93.

38