Original Article

The Cleft Palate-Craniofacial Journal

1-8
Treatment Experiences in Adults Born ª 2020, American Cleft Palate-
Craniofacial Association

With Cleft Lip and/or Palate: A Whole of Article reuse guidelines:

sagepub.com/journals-permissions
DOI: 10.1177/1055665620968342
Life Survey in the United Kingdom journals.sagepub.com/home/cpc

Kenny Ardouin, BSLP(Hons)1 , David Drake, BDS, FDS, MbChB, FRCS(OMFS)2,

Sandip Popat, BDS, LDS, RCS, FDS, RCPS, MClinDent(Prostho), MRD,
RCS, FDS(Rest Dent), RCS3, and Nicola Marie Stock, DPhil4

Abstract
Objectives: Adults with cleft lip and/or palate (CL/P) may report dissatisfaction with aesthetic and/or functional outcomes and
express desire for further treatment. Although medical intervention can improve quality of life, surgical procedures require
complex decision-making and can invoke psychological distress. The aim of this study was to gain a better understanding of
ongoing treatment-related support needs by examining self-reported treatment experiences of a sample of adults born with CL/P.
Design: An online, mixed-methods survey was designed by the Cleft Lip and Palate Association in collaboration with the Centre for
Appearance Research at the University of the West of England. A total of 207 eligible responses were received. Qualitative data
were analyzed using inductive content analysis, while quantitative data were analyzed using descriptive statistics.
Results: Although participants were satisfied with surgical and dental/orthodontic outcomes overall, many reported ongoing
difficulties. Some stated they would seek further treatment while others felt conflicted about risks and benefits. Some had refused
treatment, citing treatment fatigue, anxiety, and/or concerns that treatment would be unsuccessful. More than 40% of participants
were unaware of their entitlement to cleft-related treatment provided by the National Health Service, and many others had
experienced difficulties accessing care.
Conclusions: Although advances in health care may offer opportunities for adults to improve their quality of life, treatment
decisions should be weighed with the support of a clinical psychologist where available. Guidance for young adults considering
future treatment is also recommended. Finally, training and resources for local practitioners may improve general awareness of
CL/P services and increase access to specialist care for adults with ongoing CL/P-related concerns.

Keywords
cleft lip and palate, adult, treatment, decision-making, surgery, orthodontics, restorative dentistry

Introduction
For individuals born with a cleft lip and/or palate (CL/P),
long-term multidisciplinary treatment is a part of life. Primary
repairs during infancy and other early interventions are consid-
ered necessary to improve function and optimize aesthetic out-
comes. However, as the child grows older, many procedures
and treatments become optional and the decision-making pro-
cess becomes more complex (Wogden et al., 2019). Several
studies have demonstrated a high level of patient satisfaction
following intervention (Stock and Feragen, 2016), with patients
reporting they would undergo the same treatment again know-
ing the result (Byrne et al., 2014) and that they would recom-
mend the treatment to others (Sharp et al., 2008). Yet, studies
have also highlighted that treatment results may not always be
seen to outweigh the associated burden or discomfort (Alansari
1
Cleft Lip and Palate Association, London, United Kingdom
2
National Cleft Surgical Service for Scotland, Queen Elizabeth University
Hospital, Glasgow, United Kingdom
3
Restorative Dentistry, Spires Cleft Centre, John Radcliffe Hospital, Headley
Way, Oxford, United Kingdom
4
Centre for Appearance Research, University of the West of England,
Coldharbour Lane, Bristol, United Kingdom
Corresponding Author:
Kenny Ardouin, Cleft Lip and Palate Association, The Green House, 244-254
Cambridge Heath Road, London E2 9DA, United Kingdom.
Email: kenny.ardouin@clapa.com
2 The Cleft Palate-Craniofacial Journal XX(X)

Table 1. Survey Overview, With Sections Reported in This Article in Bold (Other Sections Are Reported in Separate Articles).a

Section # Section title Section contents

Section 1 About You Demographic questions (including age, gender, cleft type, etc)
Section 2 Your Health Physical health, surgical outcomes, dental and orthodontic outcomes, speech and hearing
outcomes
Section 3 Emotional Well-being Mental health history, self-worth, fear of negative appearance evaluation, body esteem, and
psychological treatment and support
Section 4 Bullying and Discrimination Experiences of bullying and/or discrimination
Section 5 Family and Friendships Relationships with family and friends, and perceived social competence
Section 6 Dating and Relationships Relationship status, experiences of romantic relationships, perceived competence in intimate
relationships, attachment styles, and relationship satisfaction
Section 7 Genetics and Children Knowledge of the genetic aspects of CL/P, and attitudes toward having children
Section 8 Education Education level, and educational experiences
Section 9 Employment Employment experiences, and perceived job competence
Section 10 Eating and Drinking Eating and drinking abilities and preferences, prevalence of eating disorders, and alcohol and drug
use
Section 11 Additional Demographic Living situation, experiences of crime, and household income
Information
Section 12 Engaging with the Cleft Interactions with others born with CL/P, and experiences and preferences of engaging with
Community charitable organizations
Abbreviation: CL/P, cleft lip and/or palate.
a
Data from these sections contributed to this article.

et al., 2014; Stock et al., 2015). Further, patients may feel
anxious about or intimidated by treatment (Bos & Prahl,
2011; Hall et al., 2012), find treatment information difficult
to understand (Noor & Musa, 2006), and/or feel pressured to
comply with treatment recommendations (Hall et al., 2012).
Some studies have identified an association between a higher
number of surgeries and decreased psychological well-being
and quality of life (Broder et al., 2012; Wehby et al., 2012).
This may reflect cases of greater medical complexity which
necessitate further treatment.
For adults returning to the CL/P service after many years,
treatment advances may offer new opportunities for func-
tional and aesthetic improvements. In particular, adults with
CL/P often report dissatisfaction with the appearance of the
nose, upper lip, facial profile, and teeth (Chuo et al., 2008;
Versnel et al., 2010), as well as a range of functional diffi-
culties with the potential to impact on everyday quality of
life (Gkantidis et al., 2015; Ardouin et al., 2020a). As a
result, around 45% of adults with CL/P express a desire for
further treatment (Marcusson et al., 2002; Sinko et al., 2005;
Kappen et al., 2019). However, there is also potential for
significant psychological distress and disappointment if
adults’ expectations of treatment outcomes are not met
(Stock et al., 2015; Kappen et al., 2019). Calls have there-
fore been made for additional investigation into the ongoing
treatment needs of adults (see Stock and Feragen, 2016) in
order to better understand adults’ motivations for treatment,
to facilitate psychological well-being, and to better support
the complexities of the decision-making process. The aim of
the current study was therefore to examine the self-reported
treatment experiences of a national sample of adults born
with CL/P.
Method
Design
Drawing upon previous literature, an online, mixed-methods
survey was designed by the Cleft Lip and Palate Association
(CLAPA), the UK’s largest charity supporting people and their
families affected by cleft across the lifespan, in collaboration
with the Centre for Appearance Research using the online sur-
vey platform, SurveyMonkey. The survey consisted of quanti-
tative questions including multiple-choice questions and
5-point rating scales, and open-ended qualitative questions
where participants could enter free text. The survey was also
available in paper format contained within a 54-page booklet.
The survey consisted of 220 questions split across 12 sections.
The survey was conducted as part of a larger program of work
aimed at improving the support available to adults born with
CL/P in the United Kingdom (the CLAPA Adults Services
Programme). Given the volume of data collected from the sur-
vey, findings are reported across a series of papers, of which the
current paper is one. This article reports on the data from 32 of
the 56 questions in section 2 titled “Your Health” of the survey,
supported by demographic data from sections 1, 6, 8, and 11.
The remaining data are reported in other papers (see Table 1).
Materials
The survey was designed using current literature (eg, Stock and
Feragen, 2016) and the expertise of the research team. The
question types included questions rated on a 5-point Likert
scale, such as “How much do you agree with the following
statements? (1 ¼ strongly disagree; 5 ¼ strongly agree),” and
open-ended questions, such as “In your own words, please
Ardouin et al
summarize why you feel those relationships ended.” Weighted
averages are also used when reporting some responses.
Weighted averages give an indication of how strongly a respon-
dent feels about a particular statement relative to other state-
ments by assigning each item a “weight” based on the
frequency with which it is selected and therefore reflects the
relative importance of each item. The advantage of this
approach is that rather than results being biased toward the
“middle” if there is wide variation, a weighted average depicts
where the majority of the observations fall. The draft survey
was piloted with the CLAPA Adult Voices Council (AVC), a
group of 8 adults born with CL/P. The AVC examined all
proposed questions to ensure readability and that the results
could be interpreted in a way which would answer the question
that the researchers sought to determine. Questions which were
unclear were either removed or amended with input from the
AVC. The final version was later piloted with 6 self-selecting
volunteers (all adults living in the United Kingdom who were
born with CL/P).
Procedure
Institutional ethical approval was obtained from the Faculty
Ethics Committee at the University of the West of England.
The survey was advertised via direct e-newsletters specifically
promoting the survey to CLAPA’s members; announcements
made during interviews in national and local media; posters
and leaflets; and CLAPA’s social media between July and
October 2018. Prior to survey completion, potential partici-
pants were made aware that their contribution to the survey
was voluntary, that their data would be kept confidential, that
they would not be personally identified during dissemination,
and that they could withdraw their data from the study at any
time prior to publication. Participants were asked to indicate
their consent for their data to be used for research purposes.
According to participant preference, surveys were completed
either online (via the platform SurveyMonkey) or in paper
format contained within a 54-page booklet.
Analysis
Qualitative data were analyzed independently by the first and
fourth authors using inductive content analysis (Elo and
Kingas, 2008). This type of analysis is deemed appropriate
when the aim of a study is to succinctly summarize a large
body of qualitative data, and when existing theory or research
literature on a given phenomenon is limited (Neuendorf, 2017).
First, the data were read and reread, to establish an overall
picture of the data. Initial codes were generated for each com-
ment and notes were kept throughout (Hsieh and Shannon,
2005). Next, codes were inductively grouped together into
broad categories (eg, “Concerns with Surgical Outcomes”;
Dey, 1993). Following this, subcategories were created
(eg, “Breathing/Sinus Issues”; Dey, 1993) and frequency
counts were calculated. Any discrepancies in coding were dis-
cussed between the first and fourth authors until full agreement
3
was reached. The qualitative data presented in the current paper
relates to the main category of “Treatment Experiences.”
Quantitative data were analyzed by the first and fourth
authors. Descriptive statistics were used to analyze data col-
lected from survey questions. Since most questions were
optional, not every participant answered all survey questions.
Reported percentages were therefore adjusted depending on the
number of responses received. In many cases, participants were
able to select more than one answer, and some added totals
therefore exceed 100%. Figures were rounded to one decimal
place.
Results
Participants
A total of 224 responses were received. Seventeen (7.6%) par-
ticipants were excluded as they had been born outside of the
United Kingdom. Of the remaining 207 eligible responses,
94.7% were completed online. A further 5.3% returned paper
versions via post. These data were entered into Survey Monkey
by a student intern and all surveys were checked by the first
author. The most commonly reported recruitment method was
a CLAPA e-newsletter campaign (44.4%). Other successful
recruitment methods included social media (32.8%), direct
contact with CLAPA staff (8.9%), word of mouth (3.9%), pos-
ters and leaflets (3.3%), and the CLAPA website (2.8%).
A total of 207 eligible participants contributed data to the
questions which are reported in the present paper. Participant
demographics are provided in Table 2, alongside National cen-
sus data where available (Cleft Registry and Audit Network,
2018; Office for National Statistics, 2018).
Surgical Experiences
Participants reported having undergone the following surgical
procedures during adulthood: rhinoplasty (n ¼ 107; 53.2%),
orthognathic surgery (n ¼ 47; 23.4%), pharyngoplasty or phar-
yngeal flap (n ¼ 38; 18.9%), fistulae repair (n ¼ 14; 7%), and/
or secondary lip revision (n ¼ 10; 5.0%).
Participants were asked to respond to a series of statements
about their surgical experiences on a scale of 1 (strongly dis-
agree) to 5 (strongly agree). Weighted averages were calcu-
lated for each statement and are presented in order from high
to low: “Overall, I am happy with the surgical outcomes from
my cleft treatment” (4.0); “Overall, I am pleased with the range
of surgeries that I was offered” (3.9); “Overall, I am pleased
with the surgical care and follow-up that I received” (3.9); and
“Overall, I feel that my surgical team listened to me, under-
stood my concerns, and acted upon my concerns” (3.8).
Experiences of General Dental Treatment, Orthodontics,
and Restorative Dentistry
A total of 147 (77.0%) participants had visited a General Dental
Practitioner (GDP) within the last 12 months. Twenty-three
(12.0%) participants had last visited a GDP between 1 and
4 The Cleft Palate-Craniofacial Journal XX(X)

Table 2. Participant Demographics. Table 2. (continued)

% of UK Census
% of UK Census
Demographic sample Data (%)
Demographic sample Data (%)
Student 5.3 6.5
Gender
Stay at home parent 2.9 4.8
Male 26.4 49.1
Unemployed 4.7 7.4
Female 73.6 50.9
Parental leave 0.6 –
Age
Unable to work (illness/disability) 4.1 5.5
16-20 6.9 –
Retired 8.2 18.2
21-24 6.9 –
Other 4.1 –
25-29 10.0 –
Education level
30-34 13.9 –
O-levels/GCSE/Scottish Standard 8.3 14.0
35-39 10.5 –
Grade
40-44 12.4 –
A-Levels/Scottish Higher 11.9 15.0
45-49 11.9 –
NVQ/diploma 5.3 12.0
50-54 10.0 –
Bachelor degree or higher 47.3 27.0
55-59 10.0 –
No qualifications 2.4 23.0
60-64 2.0 –
65þ 5.5 16.5
Ethnicity
White British 96.0 86.0
Other 4.0 14.0
Country of residence 2 years ago. A minority hadn’t visited a GDP for 2 to 5 years
England 83.0 83.9 (n ¼ 9; 4.7%), 5 to 10 years (n ¼ 6; 3.1%), or more than
Wales 5.0 4.9 10 years (n ¼ 1; 0.5%). Five (2.6%) participants couldn’t
Scotland 10.0 8.4 remember the last time they had visited a GDP. A total of
Northern Ireland 2.0 2.8 141 participants reported concerns about visiting the dentist.
Cleft type
These concerns included cost (n ¼ 79; 56.4%), fear/anxiety
Cleft lip only 14.0 19.6
Cleft lip and palate 72.1 30.3 (n ¼ 70; 50.0%), pain/sensitivity (n ¼ 54; 38.6%), a lack of
Cleft palate only 13.4 40.1 understanding of CL/P among GDPs (n ¼ 48; 34.3%), and
Submucous cleft palate 0.5 – having had a prior bad experience (n ¼ 30; 21.4%). Nineteen
Cleft lip type (9.8%) participants reported having ongoing difficulties acces-
Unilateral 65.5 – sing general dental services. Participants reported a weighted
Bilateral 32.8 – average of 3.5 when asked to respond to the following state-
Don’t know 1.7 –
Presence/absence of syndromes ment on a scale of 1 (strongly disagree) to 5 (strongly agree):
No known syndromes 72.6 – “Overall, I feel that my general dental practitioner listened to
Yes 16.4 – me, understood my concerns, and acted upon my concerns.”
Don’t know 11.0 – Participants reported having had a range of general dental/
Religion orthodontic/restorative dentistry procedures in adulthood.
None 48.7 25.1 These most commonly included braces (n ¼ 160; 82.5%), tooth
Agnostic 5.0 –
Christian 38.3 59.3
extractions (non-wisdom teeth, n ¼ 156; 80.4% and wisdom
Other 8.0 8.0 teeth, n ¼ 56; 28.9%), and fillings (n ¼ 143; 73.7%). Other
Sexual orientation procedures included crowns/caps (n ¼ 79; 40.7%), having an
Heterosexual 92.5 93.4 appliance such as an obturator appliance/quad helix (n ¼ 60;
Gay/lesbian 4.5 1.2 30.9%), bridge/implant (n ¼ 58; 29.9%), bonding (n ¼ 49;
Bisexual 2.5 0.8 25.3%), and dentures (n ¼ 48; 24.7%).
Other 0.5 4.6
Participants were asked to respond to a series of statements
Relationship status
Single/never married 25.3 34.6 about their general dental/orthodontic/restorative dentistry
In a relationship (not living together) 9.8 – experiences on a scale of 1 (strongly disagree) to 5 (strongly
In a relationship (cohabiting) 17.2 – agree). Weighted averages were calculated for each statement
Married/civil partnership 40.8 51.1 and are presented in order from high to low: “Overall, I am
Divorced/separated 4.0 8.0 happy with the dental and orthodontic outcomes from my cleft
Widowed 1.7 6.4
treatment” (3.7); “Overall, I am pleased with the range of den-
Other 1.2 –
Employment status tal procedures that I was offered” (3.7); “Overall, I am pleased
Employed 67.7 58.7 with the dental/orthodontic care and follow-up that I received”
Self-employed 2.4 – (3.6); and “Overall, I feel that my dental/orthodontic team
listened to me, understood my concerns, and acted upon my
(continued)
concerns” (3.6).
Ardouin et al 5

Desire for Further Treatment Table 3. Ongoing Concerns.

Most participants were not scheduled for any further operations Area of concern Number of citations
at the time of survey completion (n ¼ 163; 89.1%). However, a Surgical outcomes (n ¼ 108)
total of 108 (53.7%) participants reported ongoing concerns Facial asymmetry 42
with their surgical outcomes. These are presented in Table 3. Lip scarring 23
A minority of participants were waiting to undergo a range of Breathing/sinus issues 22
procedures, including rhinoplasty (n ¼ 13; 7.1%), secondary Difficulties eating and drinking 18
lip revision (n ¼ 11; 6.0%), fistulae repair (n ¼ 6; 3.3%), Pain/numbness 16
pharyngoplasty or pharyngeal flap (n ¼ 3; 1.6%), and/or Hypernasality 9
Skin tightness 7
orthognathic surgery (n ¼ 2; 1.1%). Participants were asked Side profile 5
to respond to 2 statements regarding potential future surgery on Smile 5
a scale of 1 (strongly disagree) to 5 (strongly agree). Weighted General dental/orthodontic/restorative dentistry outcomes (n ¼ 87)
averages were calculated for each statement and are presented Previous dental work is failing 29
in order from high to low: “I would consider having further Teeth are not straight 16
surgery if it would improve function” (3.6); and “I would con- Missing/loose teeth 12
sider having further surgery if it would improve my appear- Fear that future treatment will not be possible 10
Poor treatment coordination/follow-up 9
ance” (3.4). Maintaining oral hygiene 9
A total of 87 (45.3%) participants reported ongoing con- Fear of dental health worsening 8
cerns with their general dental/orthodontic/restorative dentistry Cost of future procedures 8
outcomes. These are presented in Table 3. Participants were Teeth alignment 6
also asked about future general dental/orthodontic/restorative Discoloration 5
dentistry treatment, as follows: “I would consider having fur-
ther dental/orthodontic treatment if it would improve function”
(3.7), and “I would consider having further dental/orthodontic Table 4. Reasons for Refusing a Procedure.a
treatment if it would improve my appearance” (3.7).
However, 63 (35.0%) participants reported having been Number of citations
offered surgery which they later chose not to undergo. A further General dental/
16 (8.3%) participants reported having been offered a general Surgical orthodontic/restorative
dental/orthodontic/restorative dentistry procedure which they Theme procedure dentistry procedure
later refused. Reported reasons for these decisions include, for Content with current 18 5
example, being content with current appearance and/or func- appearance and/or function
tion, the costs outweighing the benefits and having had enough The costs outweigh the 15 8
of surgery. A frequency count of all the given reasons is shown benefits
in Table 4. The desired outcome wasn’t 11 1
guaranteed
Had enough of surgery 8 –
Too many associated risks 6 1
Awareness of Entitlement to NHS Services Traumatic surgical experiences 5 1
Seventy-seven (41.4%) participants were unaware that they as a child
Treatment anxiety 5 –
were eligible to receive CL/P-specific treatment as part of the
Previous operations have been 5 2
National Health Service (NHS). Further, 31 (16.7%) partici- unsuccessful
pants had paid privately for CL/P-related treatment and/or ther- Concern about detrimental 5 2
apy as an adult. This most frequently related to general dental/ impact on speech
orthodontic/restorative dentistry work (n ¼ 23). Seventy-five A family member advised 4 –
(41.0%) participants reported feeling concerned that their elig- against it
ibility to access future services may change in the future. Opted for alternative 4 1
treatment instead
Cleft is part of identity 4 –
Time out of work and study 4 2
Discussion The surgeon was too forceful 2 –
Had just started a new job/ 2 –
The aim of this study was to examine the self-reported treat- relationship
ment experiences of a national sample of adults born with Considered treatment not 2 1
CL/P. The results offer insight into the treatment status of this necessary/possible in
underresearched population. The findings are discussed below later life
in more detail, alongside suggestions for further research, and a
Following analysis to identify recurrent themes, individual quotes were
clinical and community practice. paraphrased and categorized into the reasons cited below.
6 The Cleft Palate-Craniofacial Journal XX(X)
Satisfaction With Treatment Outcomes and Ongoing
Concerns
A considerable proportion of participants had undergone sur-
gery in adulthood. In line with previous research (Sinko et al.,
2005; Chuo et al., 2008), rhinoplasty was the most commonly
sought operation, with just over half of participants reporting
they had undergone this type of surgery. Participants also
reported undergoing a range of general dental/orthodontic/
restorative dentistry procedures. The most commonly cited
procedures included braces, extraction of non-wisdom teeth,
and fillings. On the whole, participants were reasonably satis-
fied with the outcomes of their surgical and general dental/
orthodontic/restorative dentistry treatment. However, many
had ongoing concerns (Table 3). The most frequently reported
concerns included facial asymmetry, prior dental work failing,
and lip scarring. These findings are in line with previous
research stating that while the majority of adults express over-
all satisfaction with treatment outcomes, lower levels of satis-
faction are often found in relation to particular facial features,
including the nose, upper lip, facial profile, and teeth (Chuo
et al., 2008; Versnel et al., 2010).
In addition to aesthetic concerns, participants reported a
number of ongoing functional issues, with the potential to
impact long-term health and everyday quality of life. These
most commonly included breathing/sinus issues, difficulties
eating and drinking, and/or facial pain/numbness. The presence
of these difficulties has been previously reported in the litera-
ture (Chuo et al., 2008; Gkantidis et al., 2015; Ardouin et al.,
2020b), yet further research to identify the specific impact of
these concerns or ways of improving patients’ quality of life is
scarce. Some of these difficulties are challenging to address
through surgery (eg, residual fistulae) and thus may require
other forms of treatment. For example, some adults will have
undergone a general dental/orthodontic/restorative dentistry
procedure (eg, a palatal obturator or speech bulb) for the pur-
poses of improving speech (Ardouin et al., 2020b). Additional
research to identify ways of supporting adults with these issues
is therefore recommended.
Medical Decision-Making in Adulthood
Previous research has suggested that around 45% of adults with
CL/P express a desire for further treatment (Marcusson et al.,
2002; Sinko et al., 2005; Kappen et al., 2019). Unfortunately,
research has also identified a greater prevalence of appearance
dissatisfaction (Marcusson et al., 2002; Chuo et al., 2008), a
higher degree of perceived stigmatization (Bemmels et al.,
2013), and elevated levels of anxiety and depression (Ramstad
et al., 1995; Marcusson et al., 2002; Sinko et al., 2005) in those
adults seeking surgical intervention. Prior research has identi-
fied improvements in psychological well-being following sur-
gical intervention (eg, Hens et al., 2011; Byrne et al., 2014),
suggesting that further treatment may be of benefit to some,
particularly given that outcomes have significantly improved
since today’s adults were originally treated (Al-Ghatam et al.,
2015; Smallridge et al., 2015). However, qualitative literature
has also highlighted the potential for significant distress and
disappointment if adults’ expectations of surgical outcomes are
not met (Stock et al., 2015; Kappen et al., 2019). In the current
study, some participants stated they would opt for further aes-
thetic/functional treatment and/or were already on the surgical
waiting list, while others felt more conflicted about the relative
risks and benefits. In line with previous research (Hall et al.,
2012; Alansari et al., 2014; Krikken et al., 2015; Stock et al.,
2015), a proportion of participants had refused further treat-
ment in adulthood, citing concerns that treatment would be
unsuccessful, treatment fatigue, and/or treatment anxiety stem-
ming from unpleasant childhood experiences. Given that most
participants would have received the majority of their treat-
ment prior to the introduction of clinical psychologists to UK
cleft teams, it is doubtful that they would have had opportunity
to discuss and address any psychological concerns. To avoid
unnecessary treatment-related distress in adulthood, these find-
ings emphasize the need for treatment decisions to be carefully
weighed; balancing what is surgically possible with the
patients’ treatment history, motivations, and expectations. The
potential treatment burden and the patient’s individual circum-
stances should be taken into consideration, with the involve-
ment of a clinical psychologist and the use of psychological
screening tools where available (Aspinall, 2010; Cadogan and
Bennum, 2011; Wogden et al., 2019). Finally, several partici-
pants had refused further treatment citing positive
self-perceptions as a reason for no longer needing corrective
treatment; a finding also identified by previous research (Stock
et al., 2015). Cleft lip and/or palate teams should therefore be
careful not to offer an array of surgical options to adults return-
ing to the service unless this is desired by the patient (Aspinall,
2010) and ensure that the option of not pursuing further treat-
ment is offered alongside other treatment options. In general,
providers should be mindful of how education about treatment
options are described with use of neutral language so patients
are aware of their treatment options with decision making
reflecting each patient’s individual concerns and goals.
Access to Local and Specialist Treatment
More than 40% of participants in the current study were una-
ware of their entitlement to cleft-related treatment on the NHS.
Consequently, some had paid for private treatment. Previous
qualitative research has also highlighted these issues, stating
the need for access to information, treatment, and support in
adulthood, while acknowledging the difficulties for the NHS
and its partners in reaching a population who have previously
been “discharged” from the CL/P service (Stock et al., 2015;
Stock et al., 2018). This is a particular challenge for those
adults who were “discharged” prior to the centralization of cleft
care and who may therefore be entirely unaware of the exis-
tence of specialist CL/P teams. Additionally, they may have
been told previously that no further treatment was available for
them, even though this may no longer be the case. As a result,
they may be less likely to proactively seek care to address any
Ardouin et al
ongoing concerns. Crucially, information should be made
available to all young adults who are about to end routine
treatment to ensure they are aware of the services available
to them and how to access them. Yet, even for those partici-
pants who were aware of their entitlement to NHS care, acces-
sing care via a referral from a local health professional had
proved challenging for some. In some areas of the United
Kingdom, a self-referral made directly to the cleft team is
possible, yet this is not uniform process across the United
Kingdom. Previous research has identified a potential lack of
knowledge among local health professionals (including GPs
and GDPs) regarding CL/P and the corresponding routine treat-
ment pathway (Stock et al., 2015; Stock et al., 2018). Research-
ers have subsequently called for improved training and
resources for local practitioners, closer communication
between local practitioners and specialist CL/P teams, and a
move by the tertiary sector to inform and empower patients to
advocate their eligibility to NHS treatment (Stock et al., 2018).
Just over 40% of participants expressed concerns over their
eligibility to NHS hospital services changing in the future.
Such concerns may reflect the recent decision to centralize
CL/P services in Scotland, wider cuts to NHS services, and
an ever-changing political environment within the United
Kingdom and internationally. Indeed, even in currently com-
missioned services, access to restorative dentistry is variable
across the United Kingdom. Given that such concerns exist
even within the NHS framework, which does ensure access
to care at any age, it is probable that in other countries without
such health care coverage that concerns over eligibility for
treatment would be even more profound. Further research with
participants living in areas which are reliant on private insur-
ance, for example, would offer a valuable comparison.
Methodological Considerations
Limitations of the present study must be acknowledged. First,
the survey was predominantly shared with adults who are exist-
ing members of CLAPA. Although CLAPA’s community is
considerable, it cannot be assumed that this group, nor the
self-selecting subgroup who responded to the survey, are rep-
resentative of the UK population. Individuals with cleft palate
only were particularly underrepresented in the current sample.
How to better represent adults who are less engaged with CL/P
services and/or those who are unaware of the services available
to them remains a significant challenge. Further, not all parti-
cipants answered all the survey questions, and therefore some
data are missing. Second, survey participants predominantly
identified as White and living in England. However, with the
exception of the lower participation rate of men, a challenge
well acknowledged in studies such as this (Johal et al., 2012),
these figures are not considerably different from UK census
data (Office for National Statistics, 2018). Nonetheless, several
previous CL/P studies have been indicative of poorer outcomes
among minority groups (see Stock and Feragen, 2016), and
further efforts are needed to ensure that support services are
applicable and accessible to the population as a whole.
7
Exploration of the psychological well-being and treatment
needs of adults who are currently living in the United Kingdom
but received the majority of their care elsewhere could also be
an important consideration for future studies. Multicenter,
interdisciplinary, and international collaboration is therefore
strongly encouraged to gain a more representative picture of
the population and to move toward a better understanding of
holistic outcomes in CL/P.
Despite some limitations, this comprehensive survey pro-
vides a large amount of quantitative and qualitative data on a
group which has to date received relatively little attention in the
context of CL/P. The findings will be used to inform future
research in this area and are pertinent to the ways in which
psychological support for adults with CL/P is delivered in clin-
ical practice and in the community.
Conclusions
Adults in the United Kingdom with CL/P may experience
ongoing issues related to surgical and general dental/orthodon-
tic/restorative dentistry outcomes. Yet, adults may be unaware
of the NHS hospital services that are available to them and/or
may have difficulties accessing specialist treatment. To address
these gaps, information for young adults who are about to
complete routine treatment is recommended, as is improved
training and resources for local practitioners. The integration
of routine patient reported outcome measures from an early age
is highly recommended to achieve optimal outcomes in the
long term. Comprehensive psychological screening for adults
returning to the service later in life is also strongly advised.
Acknowledgments
The authors would like to thank Miss Nicky Davis, Dr David Stokes,
and Ms Claire Cunniffe for their contributions to the project. The
CLAPA Adult Services Programme was funded by the VTCT Foun-
dation (registered charity: 1155360).
Declaration of Conflicting Interests
The author(s) declared no potential conflicts of interest with respect to
the research, authorship, and/or publication of this article.
Funding
The author(s) disclosed receipt of the following financial support for
the research, authorship, and/or publication of this article: The
CLAPA Adult Services Programme was funded by the VTCT Foun-
dation (Registered Charity: 1155360).
ORCID iD
Kenny Ardouin, BSLP(Hons) https://orcid.org/0000-0002-8105-
3926
Nicola Marie Stock, DPhil https://orcid.org/0000-0002-1285-691X
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