Khe Hở Môi Và Vòm Miệng: Chẩn Đoán Và Quản Lý Điều Trị (Cleft Lip and Palate)

912
N. Rumsey and N.M. Stock
complexities of stability and change in adjust-
ment over time.
The recent growth in the number of qualita-
tive and mixed methods studies has added to
the richness of our understanding of individual
experiences, as have efforts to involve service
users in the research process. More research of
this type is needed. The incorporation of qualita-
tive work into quantitative designs has the poten-
tial to promote a more patient-centred focus in
cleft research (Nelson et al. 2009 ; Rumsey and
Harcourt 2005 ) . In addition, the close concor-
dance in the psychological factors contribut-
ing to adjustment in adults and young people
with a variety of conditions affecting physical
appearance has been demonstrated, increasing
con fi dence in the extrapolation of fi ndings from
cross-condition studies to cleft (Appearance
Research Collaboration, 2009).
45.6
Support and Intervention
The good news is that many of the psychological
factors recently identi fi ed as contributing to
adjustment to cleft are amenable to change. The
major contributors are cognitive factors, which
determine the lens through which a person views
the world and the way people interpret the infor-
mation around them. For adults, the psychologi-
cal
characteristics
differentiating
those
experiencing higher levels of distress from those
who are positively adjusted include levels of
optimism, concern about negative evaluations
by others and feelings of social acceptance
(Rumsey 2012 ) . A great deal of work is neces-
sary to translate these fi ndings into practice and
additionally, the challenge of achieving a con-
sensus on the content and most appropriate tim-
ing of support and intervention remains. Recent
systematic reviews (Bessell and Moss 2007 ;
Jenkinson et al. in preparation ) of interventions
for young people and adults with dis fi gurements
resulting from a variety of causes have high-
lighted that few studies exist, and in those that
do, fi ndings are based on small sample sizes and
study designs are not robust. As the most com-
mon dif fi culties relate to negative self-percep-
tions and dif fi culties in social encounters with
others, approaches based on cognitive behav-
ioural therapy (CBT) and social interaction skills
are favoured by many, and the systematic reviews
indicate these may hold promise. Kapp-Simon
( 1995 ) , for example, showed gains in the fre-
quency of initiation of conversations and the
length of interactions as well as the frequency of
positive interactions between young people with
a cleft and their peers following social interac-
tions skills training.
With their regular contacts with patients and
their families, all HPs involved in cleft care
have a vital role to play in promoting positive
psychological adjustment. Currently, patients
are offered a variety of appearance and/or
function enhancing surgical or medical treat-
ments. While biomedical interventions can
offer bene fi ts, they rarely address all of the
psychosocial needs of patients. Provided in
isolation, they may reinforce the notion that
the way to a better quality of life lies through
improved appearance – yet research has dem-
onstrated clearly that a variety of psychologi-
cal and social attributes are also crucial for
positive adjustment. In their enthusiasm to fur-
ther correct ‘faults’ in appearance resulting
from the cleft, HPs should resist the tempta-
tion to in any way pressurise patients to
undergo further treatment if the patients feel
content with their current appearance – or to
imply that in further correction lies the way to
better adjustment. If the patient and family
have unrealistic expectations of the gains in
quality of life that might be achieved following
aesthetic surgery, the potential for disappoint-
ment is high.
Ideally, a range of interventions based on the
type and intensity of need should be developed.
These should be designed to achieve an agreed
set of outcomes, and a common set of measures
should be used to assess these outcomes. These
interventions should be delivered using a stepped
approach in which all members of the team are
involved in delivering basic psychosocial care,
including routine questions and straightforward
advice for common problems. More intensive,
individually tailored face-to-face interventions

913
45 Living with a Cleft: Psychological Challenges, Support and Intervention
would be delivered by appropriately trained psy-
chosocial specialists (see Table 45.1 ).
Some lay-led organisations have developed
campaigns and interventions designed to reduce
societal pressures on those with a visible and/or
audible ‘difference’ (see, e.g. Changing Faces:
). These have included
‘camps’ and workshops for young people with cleft
and other visible anomalies designed to offer a safe
and positive environment in which self-con fi dence,
self-esteem, peer support and practice in relation-
ship skills can be fostered (see, e.g. Changing
Faces, The Cleft Lip and Palate Association and
About Face; Tiemens et al. 2006 ) . The impact of
these activities remains to be fully evaluated.
Although slow progress towards the comprehen-
sive provision of psychological care is being made,
these resources remain the exception rather than the
rule, particularly for adults and for patients and fam-
ilies who do not have regular access to care teams. In
exploring the potential of other ways of delivering
support, online interventions are showing great
promise (see, e.g. Face IT, designed for adults ( www.
faceitonline.org.uk ) and YP Face IT (
co.uk )). These interventions could be supervised by
a cleft team member with appropriate training (Level
2 or 3) or, in suitable cases, self-administered.
Conclusions
While many people affected by a cleft are well
adjusted, this population has an increased risk
for a range of poorer psychological outcomes
compared to their non-affected peers. Research
efforts should be doubled and redoubled, par-
ticularly in relation to the identi fi cation of fac-
tors contributing to resilience in children and
young people and in order to clarify the ongo-
ing impacts of cleft in adulthood. The detail of
the experience of growing up with and living
in the aftermath of cleft and its treatment
should be better understood through prospec-
tive longitudinal research. Psychosocial inter-
ventions should be designed, implemented
and evaluated, and the routine provision of
psychosocial care across the lifespan should
be promoted in order to optimise outcomes
and to meet changing needs over time.
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Table 45.1 A stepped approach to the delivery of psychosocial care
Level 5
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experience
Coordination and supervision of psychological care
Complex interventions
Level 4
Clinical or health psychologists
Preparation for surgical interventions and post-opera-
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Provision of information and support for families
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All cleft team members
Support and reassurance
Delivery of psychological care agenda
Consideration of psychological factors in treatment
decision-making

914
N. Rumsey and N.M. Stock
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S. Berkowitz (ed.), Cleft Lip and Palate,
DOI 10.1007/978-3-642-30770-6_46, © Springer-Verlag Berlin Heidelberg 2013
46
Within the general population, it is well-
established that teasing and bullying affect a
signi fi cant minority of school-age children and
adolescents; though prevalence study fi ndings
vary, between 15 and 40 % of young people in 21
industrialised nations have been reported to be
affected (UNICEF 2007 ) . As teasing and bully-
ing in childhood are known to have a range of
short- and long-term implications for psychologi-
cal health and well-being (e.g. Herba et al. 2008 ;
Kim et al. 2005 ) , it has been the subject of exten-
sive study over the past 10 years, with research
focusing on de fi ning the phenomena and report-
ing on prevalence, possible causes and implica-
tions for those affected. This chapter will brie fl y
review the literature on teasing and bullying, as it
relates to children and adolescents with cleft lip
and/or palate in particular; its presence within and
impact on the population will be considered and
recommendations for management and intervention
will be made.
46.1
De fi ning Teasing and Bullying
Integral to the meaningful study of teasing and
bullying is a consideration of the de fi nition of
these terms. Commonly, researchers consider
three major components in a de fi nition of bully-
ing: one or more people act negatively with intent
to harm; these actions are repeated over time; and
the actions involve an imbalance of power
between the perpetrator and victim (Olweus
1978 ) . The inclusion of all three de fi nitional
components aims to distinguish typical peer
exchanges from bullying (Leigh 2007 ) , as does
consideration of a lack of reciprocity, intentional-
ity and interpersonal dominance (Crick and
Dodge 1999 ; Marsh et al. 2001 ; Olweus 1978 ) .
Within this guidance, there is no de fi nitive agree-
ment about what frequency of negative incidents
constitutes bullying, though it has been suggested
that children who are bullied once or twice show
evidence of different symptomatology than those
who are bullied more frequently and over longer
periods of time (Solberg and Olweus 2003 ) .
Experienced UK clinicians who have published a
detailed review of this subject as a model to guide
treatment and intervention argue, however, that
teasing and bullying are best de fi ned by the vic-
tim; they argue that the meaning of the event,
whether it is called teasing or bullying, is central
to the experience of, and the impact on, the indi-
vidual (see Hearst et al. 2007 ) . For some, teasing
in the form of banter can be a positive experience
when the context is one of play and can even be
considered a sign of endearment (Keltner et al.
Managing Teasing and Bullying
Sara Shavel-Jessop , Joanna Shearer ,
Elizabeth McDowell, and Daniela Hearst
S. Shavel-Jessop (*) • J. Shearer • E. McDowell
D. Hearst
Psychosocial and Family Services , Great Ormond
Street Hospital for Children NHS Trust and the North
Thames Cleft Lip and Palate Service ,
3rd Floor, Italian Building, Great Ormond Street ,
London WC1N 3JH , UK
e-mail: sara.shavel-jessop@gosh.nhs.uk ;
jo.shearer@gosh.nhs.uk ; elizabeth.mcdowell@gosh.nhs.uk ;
daniela.hearst@gosh.nhs.uk

918
S. Shavel-Jessop et al.
2001 ) . Generally, teasing and bullying are not
found to be more common in either gender,
though girls may be more involved in indirect
and relational bullying (in which peer relation-
ships are purposefully compromised by another;
Theriot et al. 2005 ) . Inclusion of cyberbullying, a
more recent construct, may increase the rates of
bullying in the typically developing population to
close to one in four children (Li 2006 ) .
46.2
Prevalence in the Cleft
Population
It has long been assumed by lay people and pro-
fessionals alike that visible facial differences or
differences in speech make children more vulner-
able to teasing and bullying (Rumsey and
Harcourt 2005 ; Turner et al. 1997 ) and there has
subsequently been much interest in researching
this phenomenon in this population. Though esti-
mates of the prevalence of teasing and bullying in
the cleft population vary in the literature, broadly
it is clear within this group that some children
will be teased and bullied persistently over time;
some will experience teasing and bullying, but no
more so than children without a cleft, and others
not at all (Broder 2001 ; Hearst et al. 2007 ) .
Children with and without a cleft may be classi fi ed
as victims, bullies, bully victims and bystanders,
and there is an extensive literature on how cogni-
tions, emotions and behaviours can differ between
the groups (Pellegrini et al. 1999 ; Toblin et al.
2005 ; Veenstra et al. 2005 ) . There is a robust lit-
erature highlighting that the objective magnitude
of visible difference does not necessarily relate to
the individual’s satisfaction with appearance
(Ong et al. 2007 ) nor to the aggressiveness of
teasing at the hands of others (Carroll and Shute
2005 ) . It is important, therefore, that links are
explored fully before assumptions are made.
Findings from an 8-year clinical audit within a
multidisciplinary UK cleft service reviewing self-
reported teasing and bullying revealed that around
24% of 10-year-olds endorsed that they had been
teased or bullied, while 13% of 15-year-olds indi-
cated the same (Shavel-Jessop et al. 2010 ) . These
estimates are broadly consistent with fi gures
reported by Whitney and Smith ( 1993 ) , who
reviewed rates of teasing and bullying in the non-
cleft population, suggesting that rates of teasing
and bullying were not elevated for children and
young people with a cleft. Other studies (e.g.
Crozier and Dimmock 1999 ) , however, have sug-
gested that playground teasing commonly impli-
cates appearance, and fear of perceived difference
can increase aggressive behaviours (Bull and
Rumsey 1988 ) .
46.3
Understanding Teasing and
Bullying in the Cleft Population
A number of theories have been put forward to
conceptualise and formulate the potential
dif fi culties or challenges facing the individual
with a cleft. These theories are well-summarised
by Hearst and colleagues ( 2007 ) and will not be
discussed in detail here. Broadly, they include (1)
family systems theories (e.g. Haley and Hoffman
1967 ; Minuchin 1974 ) , which emphasise the
family structure, strategies and trans-generational
issues relating to parents’ own experiences of
teasing and bullying; (2) attribution theory (e.g.
Kelley and Michela 1980 ) , which highlights attri-
butions of success or failure to internal or exter-
nal sources; (3) locus of control (Rotter 1966 ) , in
relation to medical services and/or teasing and
bullying; (4) learned helplessness (Seligman
1967 ) , where victims feel they have little or no
control over being teased, which can lead to them
giving up on social experiences necessary to their
developmental stage; (5) social desirability and
social modelling (Bandura 1986 ) , in which chil-
dren learn to respond to others’ teasing through
their parents’ behaviour, with possible impact on
self-worth or self-image; (6) stereotyping, stig-
matisation and scapegoating (Goffman 1963 ) ,
where individuals are rejected and excluded on
the basis of an attribute; (7) moral reasoning
(Kohlberg 1984 ) , which proposes that children
move through stages of moral understanding with
development; (8) shame and moral disgust,
thought to be a consequence of a lack of support
in moments of trauma or insecurity over time
(e.g. Gilbert and Miles 2002 ; Haidt 2003 ; Parlett

919
46 Managing Teasing and Bullying
and Hemming 2002 ; Rozin et al. 1999 ; Taylor
2007 ; Thompson and Kent 2001 ) ; (9) attachment,
loss and adjustment (Bowlby 1969 ; Brazelton
and Sparrow 2007 ) , wherein the birth of a child
with a facial difference may challenge parents’
expectations (Solnit and Stark 1982 ; Tomko
1983 ) , emotional states (including feelings of
competence and self-worth) and roles (e.g. as
buffer between the child and the world); (10)
attractiveness and friendships, where popularity
may be associated with perceived attractiveness
(Boyatzis et al. 1998 ; Dion 1973 ) , with further
impact on social development (Rubin et al. 1999 ) ;
and fi nally, (11) resilience, which though the
integrity of the construct has been questioned in
the literature (e.g. Sarason and Sarason 1993 ) ,
refers to the process of competence in adversity
(Luthar et al. 2000 ) .
46.4
Development over
the Lifespan
As cleft care is a life-long undertaking, and an indi-
vidual’s awareness, appraisals, perceptions and
beliefs change as they age, it is essential that any
model for understanding and intervening in psycho-
logical well-being is developmental in nature.
A useful review and analysis of the development of
teasing itself is by Keltner and colleagues ( 2001 ) .
46.4.1 Birth, Infancy and the Preschool
Years
Teasing and bullying in the earliest years are most
likely to affect parents of a child with a cleft,
rather than the child directly. In addition, it has
been suggested by some clinicians that the reac-
tions of new parents to an antenatal diagnosis or
a diagnosis at birth, and their subsequent process-
ing of these reactions, could have an impact on
their attachment relationship with the child with
a cleft. Findings are in consistent, however, with
some studies fi nding an increased rate of secure
attachment in parent-child dyads where the baby
has a cleft (Collett and Speltz 2007 ) . Additionally,
attachment dif fi culties, if they are present, may
not relate to the cleft alone. Rather, the birth of a
child with a cleft may activate parental childhood
issues, which may become entangled with the
meaning of the cleft. As outlined above,
dif fi culties with attachment may have a range of
implications for adjustment and for the child’s
own coping in the future. It is notable that if
dif fi culties in the attachment relationship do
arise, they are rarely evident when the child is
due to start school around the age of 5 years.
When children enter nursery or preschool, they
are often exposed to groups of (unknown) peers for
the fi rst time, in a context where the protection and
support of a parent is unavailable. Children start to
learn quickly to manage their own emotions, while
empathy emerges. These developing skills are all
important for children who have a cleft, as they
form the foundation of how children will cope with
their social interactions with others. This may also
be the fi rst time that children are asked questions
by other children about their appearance, scar or
speech; how con fi dently children can answer these
questions can have a signi fi cant impact on their
social inclusion and self-concept, and vulnerable
children may interpret curiosity as teasing (Feragen
et al. 2009 ) . Additionally, dif fi culties with hearing
may be attributed to not listening or a social failure
to respond, and children may be misunderstood by
others.
46.4.2 School Age
When children reach school age, dif fi culties
which may have begun to emerge in early child-
hood around others’ misinterpreting hearing
dif fi culties or making inaccurate assumptions
about a child’s learning or emotional needs can
intensify. Given that it has been shown that a
child’s positive self-concept is a protective factor
against the negative effects of teasing and bully-
ing (Egan and Perry 1998 ) , it is important that the
child has every opportunity to develop a positive
sense of self. Group identity also becomes increas-
ingly important at this stage of development.
Research suggests that teasing and bullying may
peak between ages 6 and 9 years (e.g. Finkelhor
et al. 2009 ) . While it has been suggested that this

920
S. Shavel-Jessop et al.
may be an artefact, due to study designs begin-
ning assessment at this age, theories of brain and
psychosocial development support the suggestion
that a peak in bullying at this age is meaningful
and not unexpected. Bullying at this age may be
dominated by individual incidents, less intense
ongoing interactive styles or social exclusion. At
this time, children are learning about the differ-
ence between teasing or bullying and questions
and curiosity and are faced with the dif fi cult task
of needing to respond appropriately to either.
During the school years, the response of an
environment to teasing and bullying within its pop-
ulation is often essential to the impact of these
experiences on the individual; cultural messages
such as ‘bullying is harmless and part of growing
up’, are generally unhelpful (Freeman and Mims
2007 ) . By contrast, school policies and interven-
tions which encourage positive disciplinary action,
high academic standards and strong parental
involvement have been shown to be effective in
reducing bullying within the community (Xin
2002 ) . As children make the transition to second-
ary or middle school between ages 10 and 12 years,
there is the added complication of the possibility of
needing to make new friends and engage with
larger groups of unknown others. It is therefore
possible, at this time, that previous dif fi culties with
peers present anew at this critical point.
46.4.3 Adolescence
Adolescence is known to be a period of transi-
tions and of achieving developmental tasks cen-
tring on individuation, identity and independence.
The social world is especially critical to adoles-
cents, with an adolescent’s peer group featuring
more prominently in the testing and achieving of
developmental tasks than ever before (Sherif and
Sherif 1964, 1965 ) . Appearance is reported to be
rated as the most important characteristic to
young people in this age group (Prokhorov et al.
1993 ) , and for young people with visible differ-
ence, this may feature especially prominently in
their self-evaluation. As it has been repeatedly
demonstrated that degree of physical difference
does not relate to psychological outcome (e.g.
Ong et al. 2007 ; Shute et al. 2007 ) , it is important
not to assume that dissatisfaction with appear-
ance will necessarily relate, however, to cleft-
speci fi c characteristics. According to data from
clinical audit, reported in Rumsey and Harcourt
( 2005 ) , 15-year-olds did not report less satisfac-
tion with appearance or friendships than peers
without a cleft. It is important, therefore, that
these issues are explored in an open-minded way
with young people presenting to services.
A number of studies have revealed risk and pro-
tective factors related to teasing and bullying in
adolescence. For example, authoritarian parenting
and (for boys) overprotective parenting have been
shown to increase vulnerability, while authorita-
tive parenting has been shown to be protective
(Baldry and Farrington 1998 ) . Having a friendship
group is also said to be protective, while social iso-
lation increases risk (Hodges et al. 1997 ) . Schwartz
and colleagues ( 1998 ) report that beliefs and attri-
butions that permit aggression and hostility
increase risk, while being friendly and prosocial is
helpful (Tani et al. 2003 ) . Finally, emotional dys-
regulation has been shown to be potentially prob-
lematic for adolescents exposed to teasing and
bullying (Mahady Wilton et al. 2000 ) .
46.4.4 Adulthood
In adulthood, teasing and bullying can occur for
the fi rst time in the workplace. It can also perpetu-
ate childhood insults, and clinically, the chronicity
of this may worsen the experience. Furthermore,
adults with a cleft may be more vulnerable if they
themselves become parents of children with a
cleft, as this may re-enact dif fi cult childhood expe-
riences. It is notable, however, that in adulthood,
as in childhood, teasing and bullying are not inevi-
table in the presence of a cleft.
46.5
Consequences of Teasing
and Bullying
Teasing and bullying have been shown to have a
potentially far-reaching impact on the well-being of
those experiencing these interpersonal dif fi culties

921
46 Managing Teasing and Bullying
(Hunt et al. 2006 ) . Most directly, teasing and bul-
lying can negatively in fl uence an individual’s self-
esteem and self-concept. It can lead to avoidance
and social withdrawal and, in more extreme cases,
precipitate the development of severe psychologi-
cal distress. Experiences of teasing and bullying
may lead to preoccupation with appearance. Some
individuals may develop mood disorders, includ-
ing anxiety and depression (Storch et al. 2003 ) ,
though it is important to bear in mind that the rela-
tionship between teasing and bullying and psy-
chosocial outcomes is far from linear (Hunt et al.
2006 ) . Furthermore, young people who experience
bullying, and girls in particular, have been shown
to be at an increased risk of suicidal ideation and
self-harm (Kim et al. 2005 ) . A study by Herba and
colleagues ( 2008 ) suggests this risk may be mod-
erated by internalising disorders in parents and
feelings of rejection at home.
Findings from an 8-year clinical audit within a
UK multidisciplinary cleft centre service revealed
that 15-year-olds with a cleft reported more avoid-
ance (e.g. of talking to unknown others, meeting
people, new situations, having photographs taken),
associated with experiencing teasing and bullying,
than did their 10-year-old peers. Fifteen-year-olds
also reported a greater negative impact of the cleft
on their lives than did the 10-year-olds. Though a
proportion of young people reported no avoidance
or ‘bother’ about being teased or bullied, there
was a relationship between the negative impact of
cleft and behavioural avoidance. This suggests
that the more children and young people feel a
cleft has negatively in fl uenced their lives, the
more likely they are to exhibit behavioural avoid-
ance (Shavel-Jessop et al. 2010 ) . Though avoid-
ance may be considered an adaptive coping
mechanism in the face of threat (Kapp-Simon
1992 ) , it may also paradoxically increase vulner-
ability to teasing and bullying (Kapp-Simon et al.
2005 ) , as it can maintain social isolation and poor
peer relations (Robinson et al. 1986 ) .
In adulthood, some individuals who have
experienced teasing and bullying in their child-
hoods may fi nd their self-esteem is compromised
(Arseneault et al. 2008 ) , and adults with anxiety
disorders may endorse a history of teasing and
bullying (McCabe et al. 2003 ) . This may affect
willingness to enter relationships or pursue ambi-
tions, which can have a signi fi cant impact on
long-term quality of life. It is notable, however,
that not all individuals who experience teasing
and bullying will go on to experience psychoso-
cial dif fi culties in later life (Patel and Ross 2003 ) .
For most, these concerns resolve with maturation
into adulthood, with future prospects no less
bright than for individuals unaffected by cleft-
related teasing and bullying.
46.6
Intervention and Management
Recommendations
Given the possible impact of dif fi culties relating
to teasing and bullying on young people with
cleft lip and/or palate, it is essential that concerns
are identi fi ed quickly and effectively, so that
intervention can be timely. Within the UK, clini-
cal psychology services within multidisciplinary
cleft lip and palate services review patients at
nationally agreed audit points (at 3 months,
18 months, 5 years, 10 years, 15 years and
20 years). When developmentally appropriate,
children and their parents are asked about any
concerns or dif fi culties they may be experiencing
and an assessment of cleft-relatedness is made.
Children and young people are then referred for
further assessment and intervention, as needed,
either within cleft services or outside, should the
concern be beyond the remit of the specialist
multidisciplinary team.
It is important to note, however, that the major-
ity of individuals with a cleft have been shown to
be no more likely to present with frank mental
health dif fi culties than their non-cleft peers
(Lockhart 2003 ) . On the other hand, understand-
able concerns relating to dif fi cult experiences are
likely to bene fi t from targeted intervention, at an
individual level, a systemic level, or a combination
of the two.
46.6.1 The Individual
When therapeutic intervention is indicated, it is
well established that there is a range of approaches

922
S. Shavel-Jessop et al.
which have been shown to be effective for the
treatment of psychological distress. These may
include cognitive behavioural therapy (CBT;
Beck 1976 ) , which focuses on the relationship of
thoughts to feelings and behaviour; schema-
focused therapy, which for young people with a
cleft may focus on addressing issues of learned
helplessness, shame, loss of control and misattri-
butions of intent (Stallard 2002 ) ; family therapy
(Hoffman 1981 ) , which focuses on the interac-
tions of and within the family to address unhelp-
ful patterns or interactions; social interaction
skills groups (Kapp-Simon and Simon 1991 ;
Kish and Lansdown 2000 ) ; and narrative therapy
(White 2005 ) and life story work (Carroll 1998 ) ,
which suggest that identity is shaped by narra-
tives/stories which may or may not be problem-
saturated.
Considering the developmental and formula-
tion issues outlined in the previous sections, inter-
vention for the individual who experiences teasing
and bullying may take a range of forms. Parents
may require support in managing anticipatory anx-
iety about their children being bullied and teased,
and this may be particularly so if parents them-
selves have had such experiences. Broadly speak-
ing, in the earliest years, the primary psychological
need for children (with respect to teasing and bul-
lying which may arise in future) is to develop a
healthy and well-consolidated positive sense of
self, to reduce their vulnerability to teasing and
their capacity to cope with it, should it arise.
In the preschool years, children often bene fi t
from behavioural intervention, which can be of
use for children with social or behavioural
dif fi culties. For parents of children at any age,
and the children themselves, interventions which
emphasise proactive support and formulation of
dif fi culties have been shown to be effective
(Maddern et al. 2006 ) . As mentioned, no rela-
tionship has been found between the magnitude
of visible difference and psychosocial outcome,
despite repeated attempts to identify such a link
(Robinson 1997 ) . In the early years, therefore,
offering parents and children reassurance that
they will not necessarily be singled out for teas-
ing can allay fears and help dif fi cult situations be
managed more con fi dently.
For school-age children, exploring their
understanding of the difference between teasing
and bullying and questions and curiosity in a pro-
active way can be bene fi cial, as can supporting
their understanding of incidents as they arise.
This psychoeducation (which may include a
didactic component) can be effective in helping
children to increase their skill repertoire. In addi-
tion to learning about the nature of teasing and
curiosity, the school-age children often bene fi t
from learning about their condition in an age-
and developmentally appropriate way. The better
able children are to respond to questions or curi-
osity, without distress, the more likely they are to
experience success and con fi dence in managing
interactions with others. Parental support to help
children develop strategies to manage concerns
and support their overall con fi dence is key.
For some children, where concerns have devel-
oped into poor self-esteem or low self-worth, mood
dif fi culties, anxiety or social withdrawal, a direct
therapeutic approach such as cognitive behav-
ioural therapy may be advantageous. Though the
evidence in the literature is less well-established,
clinical experience suggests that narrative and
other therapeutic approaches to such dif fi culties
may also be helpful. Additionally, children may
bene fi t from social skills training and from being
equipped with a ‘toolkit’ of behavioural strat-
egies (see Lovegrove and Rumsey 2005 , for
recommendations).
In adolescence, social skills training, as rec-
ommended for younger children, can be helpful
in developing self-awareness and empathy, which
can facilitate popularity, peer integration and use
of effective body language (Kapp-Simon and
Simon 1991 ) . In this older group, however, inter-
ventions may focus more on the self-control and
initiative-taking of young people, taking advan-
tage of their developing regulation, monitoring,
planning and organising (executive function)
skills. Additionally, cognitive behavioural inter-
ventions may be bene fi cial to develop active cop-
ing strategies (e.g. Lovegrove and Rumsey 2005 ) ,
challenge beliefs about others’ thoughts and
impressions and manage concerns around mood.
As it is for adolescents and younger children,
social skills work may be helpful for adults

923
46 Managing Teasing and Bullying
struggling to accommodate challenges to their self-
esteem. They may bene fi t from practising positive
self-statements and assertiveness and from cogni-
tive behavioural therapy or other direct therapeutic
interventions. When more profound dif fi culties
around identity and perceived hopelessness have
arisen as a result of a number of painful experi-
ences (including severe teasing and bullying), in
this adult patient group, offering longer-term psy-
chotherapeutic interventions may be indicated.
46.6.2 The System
Whatever the speci fi c concerns and experiences
of the individual, it is important to recognise that
context will undoubtedly in fl uence the meaning
of those experiences. Expectations, ideals and
relevance of appearance will vary in different
settings and cultural contexts, and attributions
therefore need to be considered in the develop-
ment of a formulation of dif fi culties and plan for
intervention.
In addition to the intervention models sug-
gested above, there is a range of bodies and
organisations dedicated to providing support for
individuals and systems experiencing dif fi culties
with teasing and bullying, and engagement with
these may enhance therapeutic interventions.
Within the UK, organisations such as Changing
Faces, Kidscape, Childline and Bullying UK pro-
vide online support, resources, helplines and
training for children and young people, parents,
carers and other adults. Legal frameworks also
exist within which teasing and bullying can be
positioned (see Fiddy and Hamilton 2004 for a
useful review of these in the UK); guidance from
agencies familiar with these issues (such as the
Anti-Bullying Alliance within the UK) and input
from local, national and international organisa-
tions, school and home support structures can be
facilitative.
For some young people who are attending
school, interventions at this systems level can be
helpful. Programmes within schools which
empower the bystander group to intervene and
support the victim in bullying situations can also
be of bene fi t. Explicit school interventions that
emphasise explicit rules, warmth, positive inter-
est, involvement from adults, fi rm limits to unac-
ceptable behaviour, non-hostile and non-physical
sanctions, authoritative adults, active surveillance
of pupils (including in breaks) and proactive fos-
tering of peer relationships are posited as decreas-
ing bullying within the school context (Olweus
1991, 1993 ) . Though replications have shown
mixed fi ndings, it has been suggested that long-
term approaches to systems change are likely to
be more effective than time-limited systemic
interventions (Hunt 2007 ) . Children and adoles-
cents may bene fi t from strategies such as a pro-
tective posse or a ‘buddy system’, and school or
institutional bullying policies may need to be
reviewed.
Where children or adults alike have additional
needs, such as hearing dif fi culties, it is essential
that schools, workplaces and social systems be
made aware of such challenges, so that incorrect
attributions are not made about the individual and
their social, emotional or cognitive abilities.
46.7
Considerations for the Future
Though many questions remain in the study and
understanding of teasing and bullying in the cleft
population, what is clear is that teasing and bully-
ing remain a signi fi cant issue for many individu-
als affected by a cleft, as for their non-cleft peers.
While having a cleft does not mean that an indi-
vidual will necessarily be a victim of teasing and
bullying, it is evident that those affected by these
challenges are likely to bene fi t from proactive
support and timely intervention, at both the indi-
vidual and systems levels.
Clinically, working with individuals to man-
age teasing and bullying and its effects remains a
signi fi cant part of the role of clinical psycholo-
gists within multidisciplinary cleft lip and palate
services. It is argued that it is not the clinician’s
or team’s decision whether or not an individual
should be classi fi ed as ‘bullied’; rather, this is a
subjective patient-de fi ned experience, and a
judgement cannot be made by the treating team
about the seriousness of the problem on objective
statistics alone (Hearst et al. 2007 ) . Additionally,

924
S. Shavel-Jessop et al.
it is essential that professionals remain curious
about the patient’s experience and cautious about
attributing causality or ‘cleft relatedness’ without
thorough assessment.
Beyond this, there is an opportunity to improve
the quality of interventions, taking into account a
number of issues which may be addressed by fur-
ther research and clinical audit. First, establish-
ing a common language and conceptualisation of
the constructs of teasing and bullying is essential,
to ensure that research and clinical practice are
measuring and discussing the same important
phenomena. Second, the integrity of these con-
structs must be validated-work that is currently
being undertaken by the UK Cleft Special Interest
Group and colleagues. Third, emerging evidence
(e.g. Shavel-Jessop et al. 2010 ) suggests that age
effects relating to the psychosocial impact of
teasing and bullying may be present within the
cleft population, and this requires further longitu-
dinal investigation and attention to elucidate
developmental patterns which may inform more
targeted intervention. Fourth, while a substantial
body of evidence in support of cognitive behav-
ioural therapeutic interventions exists, the evi-
dence for other therapeutic approaches (e.g.
systemic approaches) commonly used in clinical
practice is less readily available. It is important
that these approaches, which are thought to be of
therapeutic bene fi t, are not disregarded or over-
looked due to methodological challenges. Fifth,
it is clinically observed that there is enormous
variability in individuals’ reactions to teasing and
bullying; it remains to be determined how some
children become more resilient to teasing and
bullying than others and how this resilience can
be promoted. This highlights the signi fi cance of
exploration of within-group variation, which may
inform much more than studies of cleft versus
non-cleft populations have yielded to date.
Finally, it has been suggested within the
appearance-related literature that as societal views
and judgements likely underpin these interper-
sonal attitudes and dif fi culties, it may be unfair to
focus therapeutic attention and responsibility for
change on the individual alone (Clarke 1999 ) ; this
is balanced, however, by the unfairness of leaving
an individual suffering for the bene fi t of encour-
aging longer term societal change, and so inter-
ventions often focus on the individual and his or
her needs. Alongside this is a need for the wider
culture to work toward a society which is more
inclusive, less judgemental and more supportive.
With improved public awareness and sensitivity,
individuals with and without a cleft may fi nd
themselves less under threat from teasing and bul-
lying than they are today.
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Part XVI
Multicenter Clinical Reports

929
S. Berkowitz (ed.), Cleft Lip and Palate,
DOI 10.1007/978-3-642-30770-6_47, © Springer-Verlag Berlin Heidelberg 2013
47
47.1
Monitoring and Improving
Outcomes Through Multicenter
Collaboration
47.1.1 Obligations
It goes without saying that professionals involved
in health care have a continuing obligation to
review the quality of care they provide. As stated
in a WHO report ( 2002 ) :
Professionals entrusted with the provision of health
care have an obligation to review the success of
their practices and, where shortcomings are
revealed, to take remedial action. Such efforts
should constitute a continuous cycle, sometimes
known as a ‘clinical audit’…… (which) is divided
into evaluating the process of care (the way in
which care is delivered) and the outcomes of care
(what is achieved) …… Audit of the treatment of
clefts is a considerable challenge, because of the
lengthy follow-up required, the complexity, sub-
tlety and number of relevant outcomes and, above
all, the relatively small number of cases. Inter -
center collaboration still offers signi fi cant advan-
tages, by providing insight into the processes and
outcomes of treatment of comparable services
elsewhere, the establishment of future goals and
the exchange of clearly successful practices. …
Perhaps the greatest bene fi t of inter-center com-
parisons is the cooperative spirit that they foster
and a gradual diminution of rivalry.
Planned recall of patients and collection of
standardized clinical records at prescribed points
in time, not only provide the data for internal audit
of outcomes, but if done identically by a group of
centers, will allow comparisons to be undertaken.
47.1.2 Measuring Outcome
The ultimate goal of cleft care is restoration of
the patient as far as possible to a life, unhindered
by handicap or disability. However, the measure-
ment of well-being is a highly complex proposi-
tion, and there is certainly no index at present that
would allow suf fi ciently sensitive comparison
between alternative treatment protocols. Whether
in internal audit, intercenter comparisons, or clin-
ical trials, emphasis will typically be on “proxi-
mate” outcomes. These will mainly represent
different aspects of form and function in the parts
affected by the cleft, often re fl ecting the particu-
lar interest of individual provider groups. In
essence, most measures will be an indication of
the de fi cits that persist despite (or as a result of)
treatment, such as variations in appearance,
speech, hearing, and dentofacial development.
For outcome measures to be deemed meaning-
ful, they should re fl ect the needs and aspirations
of patients and their families. They should be
Eurocleft and Americleft Studies:
Experiments in Intercenter
and International Collaboration
Ross E. Long Jr., William C. Shaw,
and Gunvor Semb
R. E. Long Jr. , DMD, M.S., Ph.D. (*)
Lancaster Cleft Palate Clinic ,
223 N Lime Street , Lancaster , PA 17602 , USA
e-mail: rlong@supernet.com
W. C. Shaw , BDS, Ph.D. • G. Semb , DDS, Ph.D.
Department of Orthodontics ,
University of Manchester, School of Dentistry ,
Room G.009, Coupland III Building, Coupland Street ,
Manchester M13 9PL , UK
e-mail: bill.shaw@manchester.ac.uk ;
gunvor.semb@manchester.ac.uk

930
R.E. Long Jr. et al.
“patient centered.” The general rules of reproduc-
ibility and validity apply, the latter being espe-
cially important when outcome is assessed before
maturity. Longitudinal archives may be useful to
determine the reliability of prediction for out-
comes that are to be measured in the young (Shaw
and Semb 1996 ; Atack et al. 1997 ) . In relation to
cleft surgery, experience with a number of out-
come measures and scales has been obtained
regarding speech, dentofacial outcomes, and
patient satisfaction (Kuehn and Moller 2000 ; Sell
et al. 2001 ; Williams et al. 2001 ) .
47.1.3 Standardization
The standardization of records necessary to docu-
ment those outcomes has also been established in
the Eurocleft Consensus Recommendations on
Timing of Minimal Records (Shaw et al. 2001 ) and
also adopted by the Americleft Task Force. Further
work is certainly needed to re fi ne these and build
consensus upon international standards. Reliable
rating of appearance is still problematical, and for
speech, linguistic differences represent a signi fi cant
international challenge. However, efforts are under-
way to resolve these (see Sect. 47.3 ).
47.1.4 Scope and Limitations
of Intercenter Comparisons
Provided procedures for entry into the study and
record collection are equivalent in all participat-
ing centers, intercenter comparisons are extremely
valuable in assessing the outcome of surgery,
together with other major components of the treat-
ment program at respective centers. However, for
primary cleft surgery, it is dif fi cult, if not impos-
sible, to establish the key bene fi cial or harmful
features of the speci fi c treatment due to the invari-
ably complex and arbitrary mix of surgical tech-
nique, timing and sequence, ancillary procedures,
and surgical personnel (Shaw et al. 1992b ) .
A recent survey of European cleft services
revealed that, in 201 teams, 194 different surgical
protocols were followed for unilateral cleft lip
and palate alone (Shaw et al. 2001 ) . In the context
of intercenter outcome comparisons, this means
that if two centers differ in the use of presurgical
orthopedics and type of primary lip and palate
surgery, there is no way to determine which of
these procedures might be responsible for any dif-
ference in outcome between centers, nor would a
null result allow the conclusion that individual
aspects of the treatment program are equivalent.
Accordingly, intercenter comparisons are bet-
ter suited to comparative clinical audit and qual-
ity assurance than de fi nitive clinical research.
The existence of signi fi cant disparities in out-
come of the overall treatment process, however,
provides a basis for speculating as to the possible
cause, and intercenter studies should, therefore,
be highly motivating toward the generation of
speci fi c hypotheses for subsequent trials. This
was certainly the case in the Eurocleft cohort
study.
47.2
The Eurocleft Cohort Study
The Eurocleft study (Asher-McDade et al. 1992 ;
Mars et al. 1992 ; Mølsted et al. 1992 ; Shaw et al.
1992a, b ) was the fi rst to utilize retrospective
analysis of clinical outcome records with rigid
methodological control of biases to demonstrate
the ability to detect favorable versus unfavorable
approaches to primary infant management. It
subsequently served as a model for the second
such major intercenter collaborative outcome
comparison, the Americleft Project. These proj-
ects and the results of the efforts will be described
in Sects. 45.2 and 45.3 .
The Eurocleft cohort study began in the late
1980s as an intercenter comparison of the records
of 9-year-old children with complete unilateral
cleft lip and palate. It sought to overcome, at least
in part, some of the limitations and potential
biases associated with the comparison of out-
comes described in single center reports. A full
account of the methodology and fi ndings has
been presented elsewhere (Asher-McDade et al.
1992 ; Mars et al. 1992 ; Mølsted et al. 1992,
1993a, b ; Shaw et al. 1992a, b ) . Five of the origi-
nal six teams agreed to continue a follow-up of
the cohort till age 17.

931
47 Eurocleft and Americleft Studies: Experiments in Intercenter and International Collaboration
47.2.1 Outcomes at Age 9
At age 9, several differences between the centers
were apparent, especially for dental arch relation-
ship (Fig. 47.1 ). Whereas only 7 % of cases for
center E were considered to have a likely future
need for osteotomy, almost half (48 %) did so for
center D. It was not possible however to ascribe
success or failure to particular details of the sur-
gical protocols, but poor outcomes appeared to
be related to decentralized services without con-
sistent protocols.
47.2.2 Follow-Up
The aims of the follow-up were: to quantify the
burden of care imposed by respective protocols,
to see whether the ranking of centers for different
outcomes at age 9 was predictive for equivalent
outcomes at age 17, to assess patient/parent satis-
faction with care, and to explore interrelation-
ships with outcome and burden (Brattström et al.
2005 ; Mølsted and Brattström 2005 ; Semb et al.
2005a, b ; Shaw et al. 2005 ) . A separate compari-
son of speech outcomes was carried out at age
11–14 (Grunwell et al. 2000 ) .
47.2.3 Survey of Treatment Experience
The amount of treatment provided by the fi ve dif-
ferent teams in 1976–1979 was remarkably dif-
ferent (Table 47.1 ). Most notable was the lengthy
hospital stay associated with presurgical orthope-
dics at that time in centers D and F. The subjects
in center D also had more orthodontic visits for
treatment and review and for the overall number
of surgeries compared to the other centers. From
discussion with these centers, it would seem that
the reason for the large differences in the inten-
sity of treatment was not primarily related to
clinical need but rather to differing beliefs and
historical practices that had shaped the clinical
protocols of the period.
+
+
1
B
E
A
F
D
2
3
4
1
2
3
4
5
5
+
+
+
Fig. 47.1 Goslon individual patient scores at age 9 by
center. A Goslon score of 1 represents excellent maxillary
prominence and a score of 5, severe maxillary retrusion.
One way to consider this outcome variable is the likely
future need for subsequent maxillary osteotomy, and cases
falling below 3.5 at this age are likely candidates for
osteotomy in the late teens
A
B
D
E
F
Surgery
Mean number of surgeries
4.8 3.3 6.0
4.4
3.5
Mean days in hospital
33
31
60
24
26
Presurgical orthopedics
Months of treatment
13
0
15
0
5
Number of visits
11
0
8
0
17
Days in hospital
0
0
60
0
146
Orthodontic treatment
Treatment length (years)
5.6 3.3 8.5
3.5
4.0
Number of visits
Treatment
52
41
54
33
47
Follow-up
11
23
42
16
25
Total
63
64
94
49
72
Table 47.1 Amount of
treatment provided by fi ve
different teams from birth to
17 years of age

932
R.E. Long Jr. et al.
47.2.4 Consistency of Outcomes
over Time
The statistical analysis used to compare the fi ve
centers was a general linear mixed model applied
to longitudinal data (Diggle et al. 1994 ) . Variance
terms were included in the model to account for
between subject variation in the intercept as well
as fi xed factor for assessment point (9, 12,
17 years) and center. Full details have been
reported elsewhere (Shaw et al. 2005 ) .
As Fig. 4 7 .2 indicates, the scores for dental
arch relationship tended to improve in centers
A, B, and E, but not in D and F. There was
a consistent relationship over time for most
cephalometric variables, e.g., soft tissue pro fi le
(Fig. 47.3 ), and for nasolabial appearance.
47.2.5 Lack of Association Between
Outcome and the Amount
of Treatment
Not surprisingly, follow-up of these fi ve cohorts
of patients from age 9 to age 17 con fi rmed the
main fi nding of the fi rst report, with some cen-
ters continuing to achieve considerably better
outcome than others, at all age points. Perhaps,
1
Mean score
2
3
4
5
9
12
Age in years
A
B
D
E
F
17
Fig. 47.2 Mean dental
arch relationship scores at
ages 9, 12, and 17 years
for participating centers
6
5
Degrees
4
3
2
1
9
12
Age in years
A
B
D
E
F
17
Fig. 47.3 Mean soft tissue
pro fi le (angle SSS-NS-SMS)
at ages 9, 12, and 17 years
for participating centers

933
47 Eurocleft and Americleft Studies: Experiments in Intercenter and International Collaboration
more surprising is the lack of association
between amount of treatment and fi nal outcome
(Tables 47.2 , 47.3 , and 47.4 ). Especially ironic
is the fi nding that the two centers with the high-
est intensity of early treatment (hospitalization
in order to perform presurgical orthopedics)
achieved the lowest rankings for eventual out-
come (Figs. 4 7 .2 and 47.3 ). Thus, the poorest
ratings for nasal appearance were associated with
the lengthy use of a presurgical device called
T-traction designed not only to reduce the alve-
olar gap but also to straighten the nasal septum
(Nordin et al. 1983 ) . Patients in the center with
the least favorable dentofacial outcomes (center
D) also experienced the longest orthodontic treat-
ment duration and the highest number of orth-
odontic visits. It appears that this was partly due
to the complexity of center D’s orthodontic treat-
ment protocols with almost continuous treatment
from the eruption of the primary dentition and
partly to the unfavorable dentofacial outcomes of
primary surgery.
This lack of association between treatment
outcome and intensity may represent a key lesson
for the development of future protocols. It justi fi es
an emphasis on simplicity, economy, and mini-
mized burden for the patient, rather than adher-
ence to demanding protocols with unsubstantiated
promise.
47.2.6 Lack of Association Between
Outcome and Satisfaction
Perhaps, the most perplexing fi nding of the
Eurocleft series is the inconsistency between
objectively rated outcomes and patient/parent sat-
isfaction. There were instances where the highest
levels of dissatisfaction with treatment outcome
were reported by subjects attending the centers
with the best objective ratings (Table 47.5 ). The
possible reasons for this disparity have been dis-
cussed elsewhere (Semb et al. 2005b ) , and it high-
lights the need for concerted work on the
understanding and measurement of patient/parent
satisfaction and the provision of more holistic
models of cleft care.
Table 47.2 The relationship between outcome assess-
ment (dental arch relationship at 17 years) and amount of
infant orthopedic treatment in the different centers
Objective
ranking
Center
Months of
treatment
No. of
visits
Days in
hospital
Best
E
0
0
0

A
13
11
0
B
0
0
0
F
5
17
146
Worst
D
15
8
60
Table 47.3 The relationship between outcome assess-
ment (dental arch relationship at 17 years) and amount of
orthodontic treatment in the different centers
Objective
ranking
Center Treatment-
length
(years)
No. of visits
Treatment Checkup
Best
E
3.5
33
16

A
5.6
52
11
B
3.3
41
23
F
4.0
47
25
Worst
(D)
8.5
54
42
Table 47.4 The relationship between outcome assess-
ment (dental arch relationship at 17 years) and the mean
number of surgeries per patient in the different centers
Objective ranking
Center
Number of surgeries
Best
E
4.4

A
4.8
B
3.8
F
3.5
Worst
D
6.0
Table 47.5 The relationship between objective ranking
of nasolabial outcome and patient dissatisfaction
Objective
ranking
Percentage of
respondents
dissatis fi ed with
nasal appearance
Objective
ranking
Percentage
of
respondents
dissatis fi ed
with lip
appearance
Best
A
64
Best
B
14

E
32

A
41
B
14
F
6
D
45
E
42
Worst
F
33
Worst
D
16

934
R.E. Long Jr. et al.
47.3
Wider Networks
Following initial reports of the Eurocleft cohort
study, numerous teams from Europe and else-
where approached members of Eurocleft and
arranged to undertake blinded comparisons of their
records with the Eurocleft material or subsections
of it, some of which were published (Flinn et al.
2006 ; Fudalej et al. 2009 ; Gaukroger et al. 2002 ;
MacKay et al. 1994 ; Meazzini et al. 2008, 2010 ;
Nollet et al. 2005 ; Roberts-Harry et al. 1996 ) . Also,
during the Eurocran project that was subsequently
funded by the European Union from 2000 to 2005,
further centers from Europe had the opportunity
to undertake similar comparisons. More recently,
multicenter comparisons were completed in India
and Turkey, and new initiatives are underway in
Thailand and South Africa (Susami et al. 2006 ;
Alex 2011 ; Bellardie 2011 ; Dogan 2011 ) . The
most extensive initiative was carried out in North
America and is described below.
47.4
Americleft
Before 2006, centers in the USA and Canada
had not been as successful as those in Europe in
establishing interest and commitment to inter-
center collaborative outcome studies. In the
2002 WHO report “Addressing the Global
Challenges of Craniofacial Anomalies,” it was
noted that in the USA and elsewhere in North
America, there had been little signi fi cant momen-
tum in the area of intercenter, collaborative,
clinical research, especially compared to the
more successful efforts of Eurocleft and
Eurocran. As a result, there was little useful
information being generated from the ongoing
research which would contribute to the estab-
lishment of sound evidence-based decision making
in clinical care.
47.4.1 The Challenge
The reasons for this failure were complicated.
While the large number of centers and individu-
als providing treatment for CFA in North America
improved patients’ geographical accessibility to
care, it simultaneously created a fractionation of
the study population, thereby reducing the prob-
ability of developing patient samples of adequate
size to enable valid research. The entire landscape
was further complicated by noncomparable patient
populations, noncomparable treatment records,
unquanti fi able differences in operator skills, and
dif fi culties in letting go of biases. Also, while col-
laborative research could be structured without
violating patient privacy laws, the rigors of doing
so were suf fi cient discouragement for many clini-
cians to participate. Finally, there remained a gen-
eral lack of agreement between centers on minimal
standards for reporting and recording outcomes,
as well as cost and ethical concerns over tak-
ing records which cannot be clearly identi fi ed as
essential for diagnosis and treatment purposes.
In summary, although the desire, research tal-
ent, and patient samples were all readily available
in the North America, before 2006, the failure to
get centers to agree on something as basic as stan-
dardization of recording and reporting outcomes,
as well as governmental hurdles and a serious lack
of funding, all resulted in a huge and ongoing
missed opportunity. The WHO ( 2002 ) concluded
by stating that … “it seems most likely, that the
most promising avenue to break out of this inertia,
may still lie in the original Eurocleft approach.
With a core of interested and experienced clini-
cians, operating at high volume centers, and will-
ing to agree on records, outcome measures of
signi fi cance, and research protocols, and addition-
ally with the possible guidance from those involved
in the successful Eurocleft, Scandcleft and Eurocran
programs, it might still be possible to initiate a
major inter-center collaborative research effort.”
47.4.2 Initiation
Based on this report, in 2006, the American Cleft
Palate-Craniofacial Association (ACPA) and the
Cleft Palate Foundation (CPF) approved funds to
organize a pilot project which has become the
“Americleft Project” and ACPA’s Americleft
Task Force. From ACPA’s strategic plan, the pur-
pose of this initiative is “….to continue to develop

935
47 Eurocleft and Americleft Studies: Experiments in Intercenter and International Collaboration
strategies and execute intercenter collaborative
outcomes studies in multiple disciplines for the
purpose of documenting the outcomes and assess-
ing best practices of team care.”
Five centers were identi fi ed to participate in
the pilot project which was modeled exactly after
the Eurocleft study and resulted in completion of
initial comparisons of dental arch relationship
outcomes, cephalometric skeletodental morphol-
ogy outcomes, and nasolabial appearance out-
comes. The results of these, to be discussed
below, con fi rmed the value and bene fi ts of well-
controlled and well-designed intercenter outcome
comparisons. Most importantly, however, was
the experience and insight that was gained in
understanding the requirements, demands, and
possible obstacles that must be overcome in order
to participate successfully in such collaborative
studies.
47.4.3 Participants
The success of Americleft has always been based on
the integrity and intellectual honesty of those choos-
ing to participate and several key requirements for
participation. Participating centers’ team members
had to be experienced and focally interested in CLP
and with an interest in seeking knowledge about the
relative merits of various treatment protocols rather
than having an unquestioning loyalty to particular
procedures. While we as care providers all believe
that the procedures we are doing are the best possi-
ble for our patients, involvement in collaborative
outcome studies implies a degree of uncertainty
about the true effectiveness of our individual proto-
cols, the ability to question our own beliefs and to
accept the possibility that there may be other equally
good or better outcomes with protocols different
from the one(s) used by our own team. In addition,
the Americleft centers had to have a high volume of
patients and well-de fi ned protocols for management
of their patient population. Centers also had to have
the resources to support team representatives in
dedicating the time and absorbing the costs of the
effort as well as the availability of the necessary
records (privacy protected) and ability to secure IRB
approval from the parent institution.
47.4.4 The Original Foundation
Americleft Cohort Studies
The initial studies conducted as part of the
Americleft Project were identical to those carried
out in the original Eurocleft study (Long et al.
2011 ) : 9-year-old, mixed dentition comparison of
dental arch relationship (Hathaway et al. 2011 ) ,
craniofacial form (Daskalogiannakis et al. 2011 ) ,
and nasolabial appearance (Mercado et al. 2011 b ) .
The patients were consecutively treated by high-
volume surgeons in their respective centers. The
outcome measures were also identical to those
used in the Eurocleft study, including dental arch
relationship ratings using dental casts and the
Goslon yardstick (Mars et al. 1987 ) , skeletal and
soft tissue morphology using lateral cephalomet-
ric radiographs and standard cephalometric anal-
yses, and ratings of nasolabial appearance using
cropped lateral and frontal facial photographs
and the Asher-McDade rating system (Asher-
McDade et al. 1991 ) . All measurements and rat-
ings were done by panels of trained and calibrated
examiners using validated and well-established
methods and with all examiners blinded as to the
craniofacial center of origin for each case.
Reliability statistics were carried out on all stud-
ies and indicated acceptable levels of inter- and
intra-rater reliability. The purpose of using
records, outcome measures, and methods that
were identical to those used in the Eurocleft study
was to allow for consistency and valid cross com-
parison of fi ndings from both initiatives.
The conclusions reached also mirrored those
of the original Eurocleft study (Russell et al.
2011a ) . Figure 47.4 illustrates once again, with
the use of the Goslon yardstick to compare den-
tal arch relationships (Mars et al. 1987 ) , the clear
differences between centers with center C hav-
ing only 18 % of patients considered to have a
likely future need for osteotomy (Goslon scores
4 and 5), while over 60 % presumed so for cen-
ter B. Furthermore, the inclusion, in infant man-
agement protocols, of passive presurgical infant
orthopedic appliances to improve the alignment
of cleft maxillary segments before primary sur-
gical repair produced no measurable bene fi ts
in terms of dental arch relationship, skeletal

936
R.E. Long Jr. et al.
morphology, or nasolabial appearance in the
mixed dentition-aged patient. To the contrary, of
the three original Americleft centers which used
infant orthopedic treatment (centers B, D, and E),
all were not signi fi cantly different, or worse than
the Americleft center that used only conserva-
tive primary lip and palate surgery (center C).
Given the additional costs and burdens of care to
families and patients that are invariably incurred
as part of this addition to the treatment protocol,
lack of a signi fi cant difference at best indicates
that the procedure did no harm but at worst that it
also provided no bene fi t to justify the burden. Of
even greater signi fi cance perhaps also is the fact
that in the original Eurocleft study, exactly the
same conclusion was reached, and subsequent
to that, a randomized controlled trial called
Dutchcleft (Prahl et al. 2001, 2003 ; Bongaarts
et al. 2004, 2006 ) fi nally established, with the
greatest strength of evidence, what Eurocleft
and Americleft had suggested based on well-
controlled intercenter comparisons.
Another fi nding from the original Americleft
Project publications was that the inclusion of pri-
mary alveolar repair with primary bone grafting
(center B) was associated with the least favorable
dental arch relationships and skeletal morphol-
ogy in the mixed dentition (Fig. 47.4 ). Speci fi cally,
signi fi cantly more dental and skeletal class III
relationships with maxillary retrusion were found
in center B’s patients. As with the use of infant
passive orthopedics mentioned above, of addi-
tional signi fi cance also is the fact that in the orig-
inal Eurocleft study, exactly the same conclusion
was reached. Furthermore, this conclusion was
consistent with that of a small randomized con-
trolled trial of primary versus secondary bone
grafting by Robertson and Jolleys ( 1968, 1983 ) .
47.4.5 Additional UCLP Comparisons
with Protocols Including
Treatment Features of Interest
Using samples from the foundation Americleft
cohort as a baseline for additional comparisons,
Americleft’s three most recent comparisons
included centers which used nasoalveolar mold-
ing (NAM) as part of an infant management pro-
tocol. In one of these Americleft comparisons
of unilateral clefts, the use of NAM in the infant
management protocol produced no measurable
improvement in mixed dentition skeletal mor-
phology or dental arch relationship (center A vs.
centers B, C, D in Fig. 47.5 ) (Peanchitlertkajorn
et al. 2010, 2011 ) . To the contrary, the aver-
age dental arch relationship score for the NAM
center (center A) was actually signi fi cantly
worse than the best Americleft center (center C)
described previously and nearly identical to the
poorest Americleft result (center B). However,
there was evidence (Fig. 47.6 ) that the nasolabial
Americleft Parcent Goslon Scores
90%
100%
80%
70%
60%
50%
40%
30%
20%
10%
0%
Center C
Center E
Center D
Center A
Center B
5
4
3
2
1
Fig. 47.4 Distribution of
Americleft Goslon scores at
age 9 by center. A Goslon
score of 1 represents
excellent maxillary
prominence and a score of
5 severe maxillary
retrusion. One way to
consider this outcome
variable is the likely future
need for subsequent
maxillary osteotomy, and
cases in categories 4 and 5
at this age are likely
candidates for osteotomy in
the late teens

937
47 Eurocleft and Americleft Studies: Experiments in Intercenter and International Collaboration
appearance of the NAM-treated patients (center
A) was signi fi cantly better than the Americleft
center which used no infant orthopedics nor any
secondary nasolabial surgical revisions (center C)
(Mercado et al. 2011a ). Interestingly, there was
a trend for the NAM center’s ratings to also be
better than the Americleft centers which did not
use NAM but had carried out secondary surgi-
cal revisions of lip and nose (centers B and D).
However, these differences were not statistically
5
4.5
4
3.5
3.13
3.63
3.66
Mean goslon rating
2.45
3.23
3
2.5
2
1.5
1
0.5
0
w/o GPP
w/ GPP
Center B
Center A
Center C
Center D
Fig. 47.5 Means of
Americleft Goslon scores
at age 9 by center.
A Goslon score of 1
represents excellent
maxillary prominence and
a score of 5 severe
maxillary retrusion.
Protocols for center A
included NAM ± GPP,
center B included infant
orthopedics and primary
bone grafting, center D
included infant orthope-
dics, and center C utilized
only lip and palate surgery
5.00
4.00
3.00
2.00
1.00
.00
Nasal form
Nasal
symmetry
Vermillion
border
Nasolabial
profile
A
B
C
D
A
B
C
D
A
B
C
D
A
B
C
D
Fig. 47.6 Means of Americleft nasolabial appearance
scores at age 9 by center. A score of 1 represents excellent
appearance and a score of 5, poorest appearance. Protocols
for center A included NAM ± GPP, center B included
infant orthopedics and primary bone grafting, center D
included infant orthopedics, and center C utilized only lip
and palate surgery

938
R.E. Long Jr. et al.
signi fi cant. Since one of the primary objectives of
NAM is to improve nasolabial esthetics and pre-
sumably thereby to reduce the need for additional
revision surgeries later, these fi ndings suggest that
there could be some merit to further exploring
that possibility. But if it is found that centers using
secondary surgeries instead of NAM are able to
produce similar nasolabial appearance outcomes,
the fi nal decision as to “best practices” for naso-
labial appearance will rely on a comparison of the
burden, cost, and risk of both methods.
To further complicate the picture, however,
in a second comparison of nasolabial appear-
ance in 5-year-old patients from another cen-
ter using NAM (Singer 2012 ) , two of the
original Americleft centers and one Eurocleft
center which had 5-year records available, no
signi fi cant differences were found in frontal
and pro fi le nasal appearance. Of importance
in this most recent comparison was the inclu-
sion of an assessment of the total burden of care
involved in the management of both NAM- and
non-NAM-treated samples. Not unexpectedly,
the group receiving NAM had more clinical
appointments and more total costs involved than
those with no infant orthopedics or no secondary
surgeries. Clearly, in situations in which there
are several approaches to a problem which may
produce equivalent outcomes, the one which
can do so with the least burden, cost, and risk
of care would be considered the most desirable.
These preliminary fi ndings are strongly sugges-
tive of the need for further comparisons which
would include multiple additional centers using
NAM, as well as the initiation of a randomized
controlled trial.
Since many centers using the NAM procedure
include also another form of primary alveolar
repair called gingivoperiosteoplasty, whether the
inclusion of this feature also has a potential nega-
tive effect on growth has yet to be established.
This possibility has been suggested by some pre-
vious retrospective studies, as well as by a small
subset of the patients from the NAM center
described above (see Fig. 47.5 , center A with
GPP), but has also been refuted by other studies.
In support of the purpose of this entire discussion,
the existence of such controversies strongly
suggests the need for intercenter comparative
outcome research and well-controlled clinical tri-
als to provide us with the necessary evidence.
Clearly, both the limitations and the bene fi ts of
these multicenter, intercenter comparisons are
evident in these studies. On the one hand, attempt-
ing to attribute a favorable or unfavorable out-
come to a speci fi c feature within a particular
protocol is an impossibility. However, certain
features of interest within protocols that can be
shown to produce more or less favorable results
can be identi fi ed as worthy targets of future inves-
tigations using methods more capable of isolat-
ing their effects and burdens.
47.4.6 Comparisons of Outcomes in
Bilateral Cleft Lip and Palate
Two additional comparisons of dental arch rela-
tionship and skeletal morphology were completed
by the Americleft Project (Hathaway et al. 2008 ;
Daskalogiannakis et al. 2010 ) . These studies
compared a cohort of 9-year-old mixed dentition
BCLP patients from 3 of the original Americleft
centers, using a dental model yardstick for BCLP,
the Bauru yardstick (Ozawa et al. 2011 ) , and
standard cephalometric analysis. As in the origi-
nal UCLP study, the most favorable results were
seen in the patients from center C, with the least
favorable results in center B which included
infant orthopedics and primary bone grafting in
its protocol.
47.4.7 Comparisons of Outcomes
of Secondary Alveolar Bone
Grafting
In an expansion of the goals of the Americleft
Project, an additional procedure of interest
which has a wide range of protocol and tech-
nique variations is secondary alveolar bone
grafting. Three Americleft centers (A, C, and D
from the original study) and one Eurocleft cen-
ter (E from the original Eurocleft study) pro-
vided a total cohort of 152 consecutively grafted
UCLP and BCLP patients with postgrafting

939
47 Eurocleft and Americleft Studies: Experiments in Intercenter and International Collaboration
follow-up occlusal radiographs. Using a new
6-point bone graft rating scale developed by the
Americleft team (standardized way to assess
grafts or SWAG method) (Russell et al. 2012 ) ,
graft outcomes at a range of postgraft follow-up
times were compared (Russell et al. 2011 b ) . As
with many other intercenter comparisons
described above, a wide and distinct range of
outcomes was seen between centers (Fig. 47.7 ).
With scores of 0–3 indicating failed grafts
de fi nitely requiring additional surgery, and 4–6
indicating more successful grafts, it is clear that
centers 1 and 4 had signi fi cantly higher success
rates than centers 2 and 3. The protocols associ-
ated with the better graft results in this study
included some features of interest such as nasal
fl oor repair at the time of primary lip repair and
the least amount of maxillary expansion presur-
gically. However, there was also a wide range of
ages and length of follow-up, so additional com-
parisons are needed which may provide better
control of those variables.
To initiate this process, the most recent
Americleft study evaluated changes in bone
graft appearance and rating over time, between
two of the four centers in the initial bone graft
outcome study (centers 2 and 4) (Ruppel et al.
2012 ). The intent was to determine if there was
a minimal time of postoperative follow-up nec-
essary to make valid assessments of bone graft
outcomes, as well as a possible optimal time
for the assessments (e.g., before, during, or
after canine eruption). These centers had both
short-term (T1) follow-up radiographs (average
1 year 3 months) and long-term (T2) follow-
up radiographs (average 7 years 9 months). Of
greatest importance was that the group differ-
ences in outcomes from T1 to T2 were identi-
cal in terms of average ratings and distribution
of scores (Ruppel, et al. 2012 ) . Although a
small number of individual patients seemed
to improve or worsen signi fi cantly (more
than 2 categories), in general, the conclusions
reached about each centers’ average bone graft
outcomes were identical whether assessed at
1 year postsurgery or 7 years postsurgery. An
additional fi nding of interest was the sugges-
tions of some trends observed using multiple
regression analysis. Although not reaching sta-
tistical signi fi cance in this study, these included
the fi nding that using canine substitution for
missing lateral incisors was associated with a
greater chance of improving a rating over time
than attempting to utilize present but diminu-
tive lateral incisors or to hold space for a fi xed
bridge replacement. This suggestion that fi nal
bone graft results may be affected by manage-
ment of the lateral incisor in the cleft site cer-
tainly indicates the need for additional outcome
comparisons.
100%
80%
6
5
4
3
2
1
0
60%
40%
20%
Ctr 4
Ctr 1
Ctr 3
0%
Ctr 2
Fig. 47.7 Distribution of
Americleft SWAG scores
by center. A SWAG score
of 0 or 1 represents failed
graft with no bone bridge
and a score of 6 an
excellent graft with
complete bone fi ll-in. One
way to consider this
outcome variable is the
likely need for regrafting
cases in categories 0–3
and less likely or no need
for additional grafting
surgery in cases rated as
4–6

940
R.E. Long Jr. et al.
47.4.8 Comparisons of Speech
Outcomes in UCLP
The most recent expansion of the Americleft
Project has been the development of an Americleft
speech group. Nine speech-language pathologists
(SLPs) from cleft palate clinics across North
America (NA) participated in a 2-day training
session on the Cleft Palate Audit Protocol for
Speech – Augmented (CAPS-A) (John et al.
2006 ) . Similar to the approach taken with the pre-
vious Americleft studies, the use of a speech out-
come assessment tool that had been shown to be
reliable and valid in previous studies was inten-
tional. It saved time in that they did not need to
develop and validate a new rating tool, and it will
facilitate cross comparison of outcomes between
the European centers in the UK and Americleft.
The training, including data collection, record-
ing, and perceptual speech rating procedures, was
provided by three European SLPs who developed
and validated CAPS-A. Inter- and intra-judge
reliability were established by rating 10 samples
(from the UK and Ireland) in three separate lis-
tening sessions (pretraining, posttraining, and
maintenance). Minor modi fi cations were made to
the CAPS-A to improve ease of use and imple-
mentation with North American samples. Based
on these modi fi cations to the CAPS-A protocol, a
follow-up reliability study was done with ten US
samples and the revised CAPS-A scoring proto-
col (CAPS-A Americleft Modi fi cation).
Reliability with the new speech outcome rat-
ing tool was found to be acceptable, and the fi rst
intercenter comparisons of approximately 50
samples collected from four sites is currently
in progress (Cordero et al. 2012 ) . Other accom-
plishments include the development of the pro-
cedures for sending samples electronically to
the University of Utah where they are edited
and posted on a secure server at the university
for remote rating of the samples (Wilson et al.
2012 ) . The procedures to be employed for rat-
ing the samples (e.g., random assignment of rat-
ers, raters not rating their own samples, plans for
inter-judge and intra-judge reliability, posting of
background information on a shared spreadsheet)
have also been fi nalized. This information has
been added to the Americleft Project Study Guide
and is posted and available on the ACPA website
( ).
47.4.9 Future Americleft Plans
While these preliminary fi ndings from the
Americleft Project have produced some very
valuable evidence to help us identify best prac-
tices in cleft palate management, possibly the
most important accomplishment could be the
stimulation of interest and enthusiasm between
cleft-craniofacial centers in participating in such
comparisons. Clearly, much remains to be done.
Current Americleft plans include expansion of
the NAM comparisons to involve other centers
using that technique; comparison of outcomes
from centers using gingivoperiosteoplasty and
active pinned infant orthopedic appliances; fur-
ther comparisons of secondary bone graft out-
comes resulting from protocols varying in timing,
type, and sequencing of the grafts with orthodon-
tic treatment such as expansion, incisor align-
ment, and orthopedics; and comparisons of
outcomes from different management methods
for the lateral incisor in the cleft site such as
crown buildups, bridges, implants, and cuspid
substitution. Beyond the surgical and orthodontic
aspects of the Americleft Project, future plans
also include the expansion of the speech group
comparisons to include many additional centers
with varying protocols for speech management,
as well as the initiation of a psychosocial compo-
nent to the study with emphasis on patient and
parent satisfaction and quality of life issues.
Coincident with all of these outcome compari-
sons, all ratings of outcomes from various
approaches are being compared in the context of
the total burden of care that each carries with it.
A protocol for evaluating the burden of care and
allowing for intercenter comparisons is being
developed similar to that described for the
Eurocleft study in Sect. 47.2 . Once this total,
comprehensive assessment of multiple treatment
outcomes from a wide range of treatment
approaches used by multiple cleft-craniofacial
centers is completed, we will hopefully have at

941
47 Eurocleft and Americleft Studies: Experiments in Intercenter and International Collaboration
our disposal a clearer picture of the treatment
approaches for which we have suf fi cient evi-
dence, with or without actual clinical trials, to
identify best practices in cleft palate care.
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47 Eurocleft and Americleft Studies: Experiments in Intercenter and International Collaboration
Semb G, Brattström V, Mølsted K et al (2005a) The
Eurocleft study: intercenter study of treatment out-
come in patients with complete cleft lip and palate.
Part 1: introduction and treatment experience. Cleft
Palate Craniofac J 42:64–68
Semb G, Brattström V, Mølsted K et al (2005b) The
Eurocleft study: longitudinal follow-up of patients
with complete cleft lip and palate. Part 4: relationship
between treatment outcome, patient/parent satisfac-
tion and the burden of care. Cleft Palate Craniofac J
42:83–92
Shaw WC, Semb G (1996) Facial growth in orofacial
clefting disorders. In: Turvey TA, Vig KWL, Fonseca
RJ (eds) Principles and management of facial clefting
disorders and craniosynostosis. WB Saunders Co.,
Philadelphia, pp 28–56
Shaw WC, Asher-McDade C, Brattström V et al (1992a)
A six-center international study of treatment outcome
in patients with clefts of the lip and palate: part 1.
Principles and study design. Cleft Palate Craniofac J
29:393–397
Shaw WC, Dahl E, Asher-McDade C et al (1992b) A six-
center international study of treatment outcome in
patients with clefts of the lip and palate: part 5. General
discussion and conclusions. Cleft Palate Craniofac
J 29:413–418
Shaw WC, Semb G, Nelson P et al (2001) The Eurocleft
project 1996–2000: overview. J Cranio Maxillofac
Surg 29:131–140
Shaw WC, Brattström V, Mølsted K et al (2005) The
Eurocleft study: longitudinal follow-up of patients
with complete cleft lip and palate. Part 5: discussion
and conclusions. Cleft Palate Craniofac J 42:93–98
Singer E (2012) Burden of care analysis of infant orthope-
dics for improvement of nasolabial aesthetics in com-
plete unilateral cleft lip and palate. Unpublished
Masters thesis, University of Toronto, School of
Dentistry, Department of Orthodontics, Toronto
Susami T, Ogihara Y, Matsuzaki M et al (2006) Assessment
of dental arch relationships in Japanese patients with
unilateral cleft lip and palate using a fi ve-category rat-
ing. Cleft Palate Craniofac J 43:96–102
Williams AC, Bearn DR, Mildinhall S et al (2001) Cleft
lip and palate care in the United Kingdom – the clini-
cal standards advisory group (CSAG) study: part 2 –
dentofacial outcomes, psychosocial status and patient
satisfaction. Cleft Palate Craniofac J 38:24–29
Wilson KD, Thurmes A, Baylis AL, Cordero KN, Dixon AJ,
Dobbelsteyn C, Chapman KL, Sell D, Trost-Cardamone
J (2012) Americleft speech project: overcoming distance
with technology. In: 69th annual meeting, American
Cleft Palate-Craniofacial Association, San Jose, 11–17
Apr 2012
World Health Organization (2001) Global strategies
towards reducing the health-care burden of craniofa-
cial anomalies. Report of WHO meetings on interna-
tional collaborative research on craniofacial anomalies.
WHO Human Genetics Programme, Geneva

Part XVII
Recording Patient Orthodontic/
Surgical Findings

947
S. Berkowitz (ed.), Cleft Lip and Palate,
DOI 10.1007/978-3-642-30770-6_48, © Springer-Verlag Berlin Heidelberg 2013
48
48.1
Cleft Lip and Palate Guide:
Form Used to Record Surgical/
Facial Growth Changes
(To be used for newborns as well as for any other
age group)
A child born with a cleft and/or other craniofa-
cial anomaly is identi fi ed at the obstetric hospital
usually using a simple Birth Defect Registry Form.
The child is then referred to the cleft palate clinic
or the craniofacial anomaly center for a more
complete diagnostic work-up and treatment
planning.
At the cleft palate clinic or craniofacial anom-
aly center, the following entries are completed.
This may be the fi rst record taken:
(a) Physical systems and structures
(b) What else to look for, etc .
(c) Recommended tests
(d) Facial clefts – Tessier’s classi fi cation
(e) Initial evaluation of the cleft lip and / or
palate : This form highlights that although
clefts may be similarly classi fi ed, they can
be very different in physical appearance
and degree of spatial distortion. This dif-
ference helps to explain why the same pro-
cedure can yield different results. For
example, in bilateral clefts, the degree of
premaxillary protrusion at birth may or
may not in fl uence the child’s facial con-
vexity and palatal relationship at a later
age.
(f) The dental , prosthetic, and orthodontic
evaluation : Only a few items need to be
recorded for the next 2–3 years. It will doc-
ument whether presurgical orthodontics or
a speech aid appliance has been utilized.
The prosthetic dental form will record the
patient’s status. The more detailed infor-
mation will be recorded as treatment
progresses.
Summary of Surgical Record : This will be
the surgeon’s choice; however, each specialist
will know why the procedure has been selected
and when it is to be done. Comments are
requested.
*Cannot determine extent of impairment due
to surgery preceding fi rst.
Similarity to known syndromes
_____________________
Form completed by: ___________________
Berkowitz Recording Patient
Information Form
Samuel Berkowitz
S. Berkowitz , DDS, M.S., FICD
Adjunct Professor, Department of Orthodontics ,
College of Dentistry, University of Illinois ,
Chicago , IL , USA
Clinical Professor of Surgery and Pediatrics (Ret),
Director of Research (Ret),
South Florida Cleft Palate Clinic,
University of Miami School of Medicine ,
Miami , FL , USA
Consultant (Ret), Craniofacial Anomalies Program ,
Miami Children’s Hospital , Miami , FL , USA
e-mail: sberk3140@aol.com

948
S. Berkowitz
48.2
Physical Systems and
Structures
Case #: __ __ __ __ (1-4)
Name: ______________ (5-19) ______________
(Last)


(First)
(M)
Date of First Exam: __ __ __ __ __ __ (43-48)
Sex: Male…1
Female…2

Race: Cauc…1
Black…2
Orient…3
Other…4______ (50)
Current: __ __ oz, % __ __; Ht __ __. __ __ cm. % __ __
Systems & Structures
1. Lip: UPPER:
Norm…1
Abnorm…2
No Info…9

(05)
)
6
0
(
9
…
o
f
In
o
N
2
…
tn
e
s
er
P
1
…
e
n
o
N
:
T
F
E
L
C

LOWER:
Norm…
Abnorm…
No Info…9

(07)
Specify: _________________________
2. Hard Palate:
Norm…1
Abnorm…2
No Info…9

9
…
o
f
In
o
N
2
…
tn
e
s
er
P
1
…
e
n
o
N
:tfel
C
3. Soft Palate:
Norm…1
Abnorm…2
No Info…9
4. Uvula:

Norm…1
Abnorm…2
No Info…9





Bifid…1
Cleft…2
Missing…3


Left
Right
Medium
No
Info
Complete
1
1
1
9
Incomplete
2
2
2
9
*Surgery
3
3
3
9
Sub
mucous
4
4
4
9

(22)
(23)
(24)

Other
None Present No Info


Left
Right
Medium
No
Info
Complete
1
1
1
9
Incomplete
2
2
2
9
*Surgery
3
3
3
9
Lateral
1
2


9
(11)
Oblique
1
2


9
(12)
Other
None Present No Info

None
Present No Info

Median Cleft
1
2
9
(15)
Central Lip
Pits
1
2
9
(16)
Commissural
Pits
1
2
9
(17)
*Surgery
1
2
9
(18)
Other
1
2
9
(19)
I.
II.
III.
Specify: _________________________
____

949
48 Berkowitz Recording Patient Information Form
IV.
V.
Specify_______________________
NOTE
_______
In the following systems & structures
you are asked to indicate whether the impairment is suspected or confirmed.
-Suspected means that validation of the observation by other means must be had
-Confirmed means that validation has occurred.
-Age first noticed is only to be used if the age is other than birth.
-Specify means that you are to give a detailed but brief description of the nature of the
impairment.
-Do not leave any individual syst/struct blank, as it will be assumed that it was not
examined.
OCCLUSION
EVALUATION PRIOR TO TREATMENT:
Angle Classification:
(To be recorded per side)



Teeth in crossbite:
I

II

III
Right side________ Left side________
Right: A B C D E
Left:
A B C D E
Right: 1 2 3 4 5 6 7 8
Left: 1 2 3 4 5 6 7 8
Overjet___________mm
Overbite____________mm
TREATMENT RECORD:
Stage I:
Observation and record taking only: yes____
no____
Stage II:

a) Obturator:


yes____
no____



b) Neonatal maxillary orthopedics: appliance
yes____
no____

a. Hotz___ b. NAM___ c. Latham___

date started _______ date completed______

age_____
age______

c) Gingovoperiosteoplasty

yes____ no____ date_____ age_____

d) Retention appliance

yes____ no____ date_____ age____
Cleft:
Complete
1

Incomplete
2

Zona Pellucida
3

*Surgery
4

Other (specify)
)
8
2
(
5

950
S. Berkowitz
VI. Deciduous dentition
a) Treatment:
fixed appliance yes____
no____
removable appliance yes___ no___
date started_____ date completed_____
age____
age____
VII.
Stage III
a) Permanent Dentition: yes___no___
1. Upper Arch: appliance_________date started______
2. Lower Arch: appliance________date started_______
Date treatment completed__________
Still in treatment__________
Stage IV
a) Retention: yes____
no____
fixed appliance
yes____
no____
removable appliance yes____
no____
fixed bridgework_______
none________
b) Date treatment completed________
and in Observation________________
Date treatment discontinued_________
1) Palatal Expansion:

yes____
no____
fixed appliance yes____
no____
removable appliance yes___ no___
date started_____ date completed_____
age____
age____
Surgery
a) Maxilla –
1. Lefort I alv:

yes____
no____
age____
2. Distraction Osteogenesis yes____
no____
age____
b) Mandible –
1. Advancement (DO)
yes____
no____
age____


(surg)
yes____
no____
age____
2. Setback


yes____
no____
age____
3. Symphysis (point PO)

a) advanced
yes____
no____
age____

b) reduced

yes____
no____
age____
c) Alveoloplasty –
1. Primary


yes____
no____
age____
2. Secondary

yes____
no____
age____

a) cortical bone
yes____
no____
age____

b) medular bone
yes____
no____
age____

c) BMP-2

yes____
no____

VIII.
Nasopharyngeal analysis:
Diagnosis :



A. Soft Palate
1. Normal size____ 2. Thin____
3. Short_____ 4. Paralyzed_____
B. Tonsils
1. Absent____2. Large_____
3. Sparse____4. Medium____

951
48 Berkowitz Recording Patient Information Form
IX.
C. Pharyngeal Depth
1. Shallow_____
2. Deep_______
3. Normal_____
D. Pharyngeal Wall Movement
1. Passavants Pad____
2. Lateral wall movement
a. good___ b. poor___ c. cannot determine___
Speech:
E. Cranial Base
1. Normal____ 2. Acute____ 3. Obtuse____
F. Adeniod
1. Sparse____ 2. Medium____
3. Large_____ 4. Absent_____
X.
C. Soft Palate Surgery:
a) None____
b) Pharyngeal____
c) Pushback_____
d) Both b) and c)____
Cervical vertebrae:
A.
B.
1.
2.
3.
Normal____
Abnormal_____
Fusion C2-C3_____
Occipitalization of Anterior Tubercle of Atlas _____
Malposition of Atlas____
B. Evaluation Using Cepths:
A.Velopharyngeal Speech Aid
a. on “U” good___poor___
b. on “S” good___poor___
D. None____________
A.
1. Normal____
2. Hypernasality____
3. Hyponasality____
4. Treatment: yes
no

952
S. Berkowitz
XI.
Recommendations:
A. None____ B. Pharyngoplasty____ C. Pharyngeal Flap____
D. Speech Aid____ E. Adenoidectomy____ F. Tonsillextomy____
G. Speech Therapy_____
H. Other____________
i) Maxillary Distraction Osteogenesis ______
or
Orthognathic Surgery______
ii) Mandibular Distraction Osteogenesis______
or
Mandibular Orthognathic Surgery_______
iii) lip_______nose_______
I. Observation or Recall_______

Part XVIII
The Patient’s World

955
S. Berkowitz (ed.), Cleft Lip and Palate,
DOI 10.1007/978-3-642-30770-6_49, © Springer-Verlag Berlin Heidelberg 2013
49
49.1
Introduction
Over the past decade, major contributions have
been made to the literature regarding social, ethi-
cal, and health policy aspects of craniofacial care
and conditions.
Strides have been made in the measurement of
quality of life and in understandings of health sta-
tus (Chetpakdeechit et al. 2009 ; Damiano et al.
2007 ; Kramer et al. 2008, 2009 ; Mani et al. 2010 ;
Munz et al. 2011 ; Nelson et al. 2011a, b, c ;
Nusbaum et al. 2008 ; Patrick et al. 2007 ; Sagheri
et al. 2009 ; Stone et al. 2010 ; Strauss and Cassell
2009 ; Wehby and Cassell 2010 ) . Health services
research has emerged as a major contributor to
the literature on cleft and craniofacial conditions
(Alkire et al. 2011 ; Austin et al. 2010 ; Blume and
Henson 2011 ; Boulet et al. 2009 ; Cassell et al.
2012 ; Chuo et al. 2008 ; Foo et al. 2011 ; Furr et al. 2011 ;
Knapke et al. 2010 ; Kuttenberger et al. 2010 ;
Mendoza 2009 ; Nelson et al. 2011a, b, c ;
Payakachat et al. 2011 ; van Aalst et al. 2011 ) .
Psychological research has become more sensi-
tive to the challenges, resilience, and health of
children and adults with clefts and craniofacial
conditions (Baker et al. 2009 ; Berger and Dalton
2009, 2011 ; Black et al. 2009 ; Brand et al. 2008 ;
Feragen and Borge 2009, 2010 ; Feragen et al. 2010 ;
Loewenstein et al. 2008 ; Marshman et al. 2009 ;
Meyer-Marcotty et al. 2010 ; Murray et al. 2009 ;
Mzezewa and Muchemwa 2010 ; Nelson et al.
2011a, b, c ; Strauss et al. 2007 ) .
A lively discourse on ethical issues and the
values related to craniofacial care has emerged
(Abbott and Meara 2010 ; Aspinall 2010 ; Bijma
et al. 2008 ; Mossey et al. 2011 ; Nusbaum et al.
2008 ; Strauss et al. 2011 ) . In this same period,
patients and families have been understood to
live within social and health system realms that
de fi ne their experiences with their conditions.
During this period, advances in technology and
informational systems have altered our under-
standings of craniofacial conditions around the
world (Corlew 2010 ; Rodrigues et al. 2009 ) .
The focus of this chapter is on major craniofa-
cial conditions in historical context. The chapter
is a revised version of a 1992 paper which
explored themes that have proven to be both
enduring and salient. Craniofacial surgery and
neonatal intensive care have made it possible for
children with serious craniofacial conditions to
live and often to experience effective habilitation.
Social, Ethical, and Health Policy
Issues in the Care of Children
with Major Craniofacial Conditions
Ronald P. Strauss
Parts of this chapter are reprinted with the permission of
The Cleft Palate-Craniofacial Journal , Allen Press
Publishing Services. The author has assigned all copyright
ownership to the American Cleft Palate-Craniofacial
Association. Parts of this chapter were published in The
Cleft Palate - Craniofacial Journal , November 1992.
Volume 29, No. 6.
R. P. Strauss , DMD, Ph.D.
Departments of Dental Ecology and of Social Medicine,
The University of North Carolina at Chapel Hill,
Schools of Dentistry and Medicine ,
104 South Building; C.B. # 3000 ,
Chapel Hill , NC 27599-3000 , USA
The UNC Craniofacial Center , Chapel Hill , NC , USA
e-mail: ron_strauss@unc.edu

956
R.P. Strauss
These therapeutic interventions also raise impor-
tant social, policy, and ethical issues. This chap-
ter reviews the dilemmas which relate to the
gatekeeper role for physicians, the impact of
prenatal diagnosis, and the allocation of scarce
fi scal and health resources to craniofacial care.
The high degree of cost, the intense investment
of medical resources, and the uncertain out-
comes in the care of children with major cranio-
facial conditions must be considered in the
distribution of resources within a health system.
The rationing of health resources may be a future
determinant of how care for major craniofacial
conditions is delivered.
49.2
Craniofacial Treatment
Decision Making
“I felt shock, hopelessness and an overwhelming
responsibility. With birth defects as bad as hers, it
was hard to believe much could be done.” – a
44-year-old mother of a 12-year-old child with
Apert syndrome
The birth of a child with a major craniofacial
condition is a serious crisis in the life of the fam-
ily affected. Not only is the desire for a healthy
baby unful fi lled, but the family must reframe
their expectations for the child’s needs. This may
mean a reevaluation of who is to provide care to
the infant, how health needs will be met, and how
health costs will be covered. The family may also
need to consider the future health, social, and
developmental limitations associated with their
child’s condition.
In the context of family crisis, clinical deci-
sions must often be made about the extent, tim-
ing, and nature of the treatment which is to be
provided. Family values about the sanctity of
life and norms for parental responsibility will
guide decisions. Religious perspectives and eth-
nic or cultural values often provide the basis for
making treatment decisions within the family
(Botto et al. 2006 ; Florian and Katz 1983 ;
Kleinman 1979 ) . Other social in fl uences may
affect decision making and the available choices
for parents and professionals.
Attitudes about dis fi gured children and about
medical care may guide treatment decisions rela-
tive to children born with major craniofacial con-
ditions. A fascination with dis fi gurement pervades
mythology and literature (Fiedler 1978 ; Shaw
1981 ; Strauss 1985 ) . Combinations of fear, repul-
sion, interest, and sympathy affect attitudes about
the child with a defect. Signi fi cant social ambiva-
lence may be generated by children with obvious
conditions. This ambivalence may fi nd itself
re fl ected in how a family or a clinician decides
about major craniofacial surgery.
The child who is perceived as having a limited
role as a future community participant or worker
may be devalued, and treatment resources may be
directed toward the needs of other children. In
some settings, children seen as hopelessly
af fl icted may be neglected, maltreated, or abused
(Mosher 1983 ) . In other sociocultural settings,
the child with a defect may be nurtured, seen as
an exceptional child with special needs, and pro-
vided with the full range of possible treatment
options. Historical and anthropological studies of
child treatment and maltreatment (Demause
1977 ) suggest that different cultures and different
eras of time are characterized by markedly differ-
ent attitudes and values placed on children and
their health care needs.
Medical and societal resources and wealth
will affect treatment decisions and options. In
settings where treatment facilities are limited, the
options for clinical activism may be severely cur-
tailed. I am reminded of an experience with a
rural mother who had just delivered a very pre-
mature, but otherwise healthy infant, in a regional
hospital in a developing nation. The baby died
soon after birth, there being no neonatal intensive
care unit and limited medical expertise. The
mother raised her head from the bed when I
entered her room and asked me directly “would
my baby have died if she were born in your coun-
try?” I knew the answer was that the baby would
probably have been treated in the neonatal ICU
and might be alive. How you might have answered
her question will provide some insight into the
complexities that surround treatment allocation.
Medical facilities and health resources allow for

957
49 Social, Ethical, and Health Policy Issues in the Care of Children with Major Craniofacial Conditions
care in previously untreatable conditions and cre-
ate options for medical decision making. They
also raise new moral and ethical quandaries about
who is to live and with what quality of life.
This chapter will examine the moral and health
policy basis for making treatment decisions and
will suggest social and ethical issues that may be
raised in the care of children with major craniofa-
cial conditions. It will consider four topics: (1)
the history of care for children with major cranio-
facial conditions, (2) the gatekeeper role for phy-
sicians, (3) the impact of prenatal diagnosis, and
(4) the social justice and resource allocation.
49.2.1 The History of Care for Children
with Major Craniofacial
Conditions
Until the last several decades, many, if not most,
children with major craniofacial conditions did
not survive or experienced a much reduced qual-
ity of life (Kleinman 1979 ; Scheper-Hughes
1987 ) . This occurred primarily because of the
inability to provide nutrition, craniofacial surgi-
cal care, or respiratory assistance. Sometimes,
the lack of willingness to invest caregiving or
resources to the infant with a birth defect was
in fl uential.
Infanticide
(Dickeman
1975 ;
Pertschuk and Whitaker 1982 ) and the with-
drawal of caregiving or sustenance (Langer 1974 )
have been discussed as possible responses to the
infant with a marked defect. There is evidence
that gender selection (Divale and Harris 1976 ;
Scheper-Hughes 1990 ) and the lack of skill in
caring for such a child (Mosher 1983 ) may result
in the infant’s death. Infanticide is now univer-
sally seen as a crime, and most societies have
developed social roles for children with birth
conditions or defects. In spite of this, infanticide
or child maltreatment may still occur hidden
behind closed doors.
The success of neonatal intensive care units
and craniofacial surgery has made it possible to
treat many serious birth defects that may once
have resulted in a child’s death. It is likely that
the application of these advanced forms of
technological assistance have changed the pro fi le
of cases treated by many cleft/craniofacial teams.
While craniofacial teams may once have predom-
inantly seen children born with uncomplicated
clefts of the lip and palate, many teams now
report receiving referrals of children with multi-
ple defects and complicated craniofacial condi-
tions (Gupta 1969 ) .
This epidemiologic change is so substantial
that many centers have changed their names from
“cleft palate team” to “craniofacial team” (Strauss
1992 ) . With this marked shift in focus, clinicians
may fi nd themselves pondering dif fi cult ethical
questions. They may ask: Should all children
who have defects that might be repaired receive
treatment? Should infants with major and handi-
capping craniofacial conditions routinely be
offered treatment in the neonatal intensive care
unit? Should they always get craniofacial surgi-
cal care? What criteria might be used to ration
such care? Who should make the decision to treat
or not to treat? To what extent does the physician
serve as the agent of the society in directing the
use of scarce and costly resources? In the context
of a health care system, how many resources can
be directed toward any individual child?
The advent of major craniofacial surgery, pre-
natal diagnosis, and neonatal intensive care raises
basic social and ethical dilemmas relative to:
(a) Who should be treated and to what degree.
(b) How scarce resources (i.e., medical or surgical
services or expertise) should be allocated.
(c) Whether and how cost/bene fi t calculations
can be used in clinical decision making.
(d) What is the goal for major craniofacial sur-
gery or treatment.
(e) How much does the society value children
and adults with disabilities.
The extremely high cost of correcting a compli-
cated craniofacial condition (Shprintzen 1990 )
suggests that the society must consider the value
of surgical care. Is the cost of repeated surgeries
recouped in the development of human potential?
Does the patient achieve social integration and
productive vocational performance because he/
she was treated? If not, what was the rationale of
treatment? Do surgeons and families seek

958
R.P. Strauss
incremental improvements without regard to the
fi nal status and likely outcome of the process? Do
surgeons have the capacity to manage access to
their services? The role of the gatekeeper to med-
ical and surgical services is a critical one, and the
physician or the health insurer/managed care pro-
vider serves as an agent for the society in making
treatment decisions.
49.2.2 The Gatekeeper Role
for Physicians
The gatekeeper role (Bunker 1970 ; Lapham et al.
1996 ; Strauss 1983 ) involves the professional in
exerting control over the availability of clinical
treatment. Physicians encourage or discourage
treatment based upon their legal responsibilities,
values, and moral codes. Physicians are social-
ized during medical training to meet patients’
needs and to deliver a community service. They
guide patients in accessing care and by making
referrals when specialized expertise is required.
The basic precept is to be of assistance, do no
wrong, and to protect the public and community
health.
Professional time and patient fi nancial
resources are not endless; thus, health profession-
als serve to modulate and control access to their
services, clinical skills, and their time. Surgeons
and other clinicians are constantly deciding
whether it is worthwhile to undertake treatment;
thus, they manage the market for their services.
Physicians offer only treatment that they perceive
as necessary and of bene fi t for the patient or the
community. Often, the surgical gatekeeper role is
apparent when surgical treatment is denied. For
example, a surgeon’s decision not to operate on
the palate of a nonverbal and developmentally
delayed child, until the child begins to speak, is
an application of the gatekeeper role.
The gatekeeper role may also relate to how a
clinician rationalizes the investment of medical
or surgical care. Social and psychological ratio-
nales for care may re fl ect the clinician’s judgment
that nonbiologic bene fi ts may result from struc-
tural change. For example, the surgeon’s decision
to do a rhinoplasty on a 14-year-old girl with a
cleft lip and minor nasal asymmetry may be based
more on expected changes in her social experi-
ence, rather than on her breathing ability. The use
of psychosocial rationales for care is an applica-
tion of the gatekeeper role.
The gatekeeping role allows physicians to
manage many of the health care system’s scarce
resources, including the operating room and the
intensive care unit. Physicians determine who
needs surgery and who requires highly technical
hospital-based management. Insurance compa-
nies, managed care organizations, and other
third-party payment agencies have developed
mechanisms to monitor the use of these costly
resources. The routine use of second surgical
opinions, case managers, and insurance policy
exclusions have been used to control medical
activism and expenditures. As a health care sys-
tem changes and health care, each patient and
family may have a unique experience interacting
with their payer, if covered at all. Physicians and
other health professionals have expressed consid-
erable dissatisfaction with the adversarial rela-
tionship that sometimes exists between the
professional and the payer/insurer of care.
The responsibility of being a gatekeeper
implies a considerable amount of control and
power over one’s work. Controlling the access to
and use of one’s own services may imply a
con fl ict of interest. The gatekeeping role may be
placed under stress when a physician is fi nancially
rewarded for being overly active about care.
When a surgeon who is charged with deciding
about how much care is necessary for a patient is
also to receive fi nancial reward from perfor-
mance of the treatment, ethical roles may be
stressed. This also may occur when outside par-
ties, such as insurance companies or managed
care organizations, are advocates for limiting the
provision of care. They too have a gain from lim-
iting the amount of service realized by a given
patient or client.
In the matter of craniofacial clinical decision
making, the clinician serves as the agent of the
society, making judgments which balance a
variety of inputs. Factors which may play a role
in these judgments include the likely bene fi ts to
the patients, the risks of treatment, the family

959
49 Social, Ethical, and Health Policy Issues in the Care of Children with Major Craniofacial Conditions
capacity to cope with the care of the child, the
child’s long-term prognosis, the cost of care,
and the ability to access fi nancial resources to
pay for care.
The clinician’s judgment may also relate to
career goals and the mission of medicine. The
physician may feel charged by the society with
the mission of “normalizing” children born with
birth defects. Furthermore, the desire to perform
dramatic and highly technical treatments on the
“cutting edge” of medicine may be appealing.
Both goals imply the likelihood of providing a
patient with sophisticated and technical care. The
desire to further the science and the clinical disci-
pline by performing novel treatments may serve
as a stress on the gatekeeper function.
Physicians primarily de fi ne their role in terms
of the individual relationship with the patient and
the family. Many physicians are educated to see
themselves as advocates for the child patient as
well as the parents and have dif fi culties in
moments where the interests of patients and par-
ents may not coincide. For example, in a situation
where a child has a serious defect that would
require intense and costly care and the family
expresses a wish to limit treatment, the clinician
may be placed in a dif fi cult position. Whose
interests does the physician protect? Who advo-
cates for the child’s interests? Does the tendency
to medically intervene generally prevail? Do the
parents always have the fi nal say relative to a
treatment decision? Legislative and judicial deci-
sions have become central in guiding medical
decision makers and in de fi ning the limits of
medical autonomy (Chetpakdeechit et al. 2009 ) .
Medical education socializes Western physi-
cians to be activist in their clinical responses. In
simple words, due to their training, doctors try to
fi x disabilities when possible. The decision not to
offer care, or to withdraw care already started, is
always dif fi cult and places the physician in strug-
gle with well-socialized values to heal. In new or
innovative forms of treatment, few guidelines or
norms may exist for deciding about whether to
intervene. In the case of craniofacial surgery,
activist principles and the desire to fi x disabilities
predispose the physician to providing treatment
whenever possible.
Periodically in the life of a profession, speci fi c
technological innovations markedly alter the
course of medical practice and thinking. This
occurred with the introduction of the stethoscope,
the aseptic technique, and the use of antipsy-
chotic medications. This also occurred with the
introduction of prenatal diagnosis and the intro-
duction of craniofacial surgery. The introduction
of these technologies has markedly changed how
medicine deals with congenital disabilities.
Prenatal diagnosis and ultrasound imaging have
created a situation where parents have the option
about whether to bear a child with a known
defect. Craniofacial surgery has created the
option to correct defects that may exist. Parents
must weigh the pain of bearing a child with a
birth defect and the expectation of surgical cor-
rection against their moral values and may have a
choice about whether to abort the child.
Craniofacial surgery and prenatal diagnosis are
both possibly major components in the family
and parental decisions about the child with a
major craniofacial condition.
49.2.3 The Impact of Prenatal
Diagnosis
Prenatal diagnosis and imaging of major cranio-
facial conditions is increasingly common (Bosk
1992 ; Eng et al. 1997 ; President’s Commission
for the Study of Ethical Problems in Medicine
and Biomedical and Behavioral Research 1983 ) .
The use of ultrasound allows for the early visual-
ization of fetal defects. Techniques such as
amniocentesis, genetic screening, and risk
appraisal may provide the family and physicians
with information about a child’s craniofacial con-
dition prior to birth. The knowledge of a condi-
tion during the prenatal period implies the
possibility of choice relative to the birth of a child
with a defect.
Knowledge about defects prior to birth has
caused medicine to face profound questions about
how we as a society will deal with prenatal infor-
mation about serious craniofacial conditions.
Will there be signi fi cant pressure on mothers to
abort such fetuses in order to avoid the cost and

960
R.P. Strauss
pain of corrections? Will there be rewards for
parents who decide to save the society the cost of
treatment? Will we stigmatize and harass parents
for seeking to terminate a life, regardless of qual-
ity, by abortion?
The realities of the high cost associated with
the survival and treatment of children with major
craniofacial defects may affect how we perceive
a parent decision to abort a fetus with an
identi fi ed condition. A decision to bear such a
child implies major expenditures for the parents
and/or the society.
Advocates against the availability of abortion
will eventually clash with the reality that some
parents of fetuses with signi fi cant conditions may
wish to terminate the pregnancy. Indeed, prenatal
diagnostic technology is based on the premise
that some action may occur in response to an
untoward fi nding on testing. Some parents may
merely be seeking prenatal information to be pre-
pared for the birth of their child. Other parents
may want to arrange for adoption of a special
need child, while still others will seek abortion.
Social policy debates will determine if abortion
remains available or if it becomes illegal. The
debate may consider if society is willing to pay
the costs of maintenance and rehabilitation of
congenitally disabled children and adults.
Social values relative to aesthetic and func-
tional conformity are also challenged by prena-
tal diagnosis of birth defects. Will infants who
are different or have birth defects be aborted or
be accepted? How major a condition must there
be to rationalize an abortion? Will there be less
tolerance of different appearance or identity if
those with different identities are aborted or cor-
rected? To what extent does living with a range
of appearances and disabilities humanize a soci-
ety? Some would argue that medicine should
exert itself to make children look as normal as
possible to meet social expectations. Others
would say that instead of changing the child or
his appearance, the focus should be on changing
social values to encourage the acceptance of
those who appear different. For the moment,
there is little question that those who look differ-
ent are treated in many societies as less than
equal and often are stigmatized.
The introduction of the current array of
sophisticated prenatal screening mechanisms did
not imply that medicine was prepared for the
social and ethical rami fi cations of their use. One
would hope that the introduction of new biomedi-
cal technologies implies the willingness to con-
sider the complex issues these procedures raise
for clinicians and for those engaged in health care
fi nancing and policy.
49.2.4 Social Justice and Resource
Allocation
It is sometimes argued that when limited resources
exist, health dollars are best spent on prevention,
rather than treatment. Some would state that
health and economic resources are most rationally
invested in maternal health care and in the appli-
cation of established preventive regimes. Thus, it
could be argued that prevention of fetal alcohol
syndrome is preferable to focusing resources on
its treatment. Most would agree that when possi-
ble, a preventive approach is preferable.
A health system, however, often functions to
support treatment, as opposed to preventive,
approaches. In the case of cancer and heart dis-
ease, known risk factors might be reduced, yet
the bulk of health dollars are spent on the treat-
ment of existing conditions. In the case of major
craniofacial conditions, resources might best be
spent on research and on the prevention of
defects. This might involve developing programs
that encourage maternal health and nutrition,
genetic counseling and screening, as well as pre-
natal
ultrasound/diagnosis.
A
reasonable
approach to health policy might be built on
research into craniofacial health promotion and
disease prevention.
The major craniofacial disorders are however
rare and currently are often of unknown etiology.
Given the paucity of preventive craniofacial
efforts, the bulk of resources are currently focused
on the clinical treatment of these conditions. In
the face of the reality of a child who is born with
a major craniofacial condition, all attention is
understandably drawn to the clinical treatment
possibilities.

961
49 Social, Ethical, and Health Policy Issues in the Care of Children with Major Craniofacial Conditions
Physicians are not socialized to ask questions
about social justice when encountered with a
speci fi c patient’s needs. Few would fi nd it rea-
sonable for a clinician to worry about whether the
society’s resources are better spent on public
health or immunization, rather than on expensive
surgical reconstruction. Most citizens would
agree that public dollars would be better invested
in the realm of prevention, and yet when faced
with the denial of reconstruction to the individual
patient, due to limited resources, the social will to
persevere diminishes.
Under circumstances of individual patient
need, it is dif fi cult to pose questions about the
appropriate allocation of economic and clinical
resources or social justice. In the context of ethi-
cal discussion, one may consider questions of
fairness and equity.
Values on the sanctity of the individual are
strongly held, and rationing of health care has
proven to be a dif fi cult prospect (Blumenfeld
et al. 1999 ; Churchill 1987 ) , though it happens in
a de facto way regularly. Rationing of health care
occurs, but it often is by limited insurance or pol-
icies, total lack of health coverage, or because of
a scarcity in providers or hospital facilities. For
the individual clinician, these barriers to care
may not be apparent since many patients who
reach his/her door are able to pay for care. The
issues arise when payment is not available and
when care is very costly. If patients cannot them-
selves afford care, and insurers are unable or
unwilling to pay, then the true impact of rationed
clinical care becomes apparent.
Health care is already allocated in many soci-
eties. Poverty and ethnic minority status may
result in lower health status when compared to
more af fl uent or nonminority populations (Aaron
and Schwartz 1984 ) . In a society where marked
disparities in access to health care exist, one must
ask whether the ability to pay is the most just
medium for deciding who gets care. In the case
of major craniofacial conditions, should ability
to pay for expensive services guide their
availability?
Insurers, case managers, or governmental
agencies may determine who receives medical
care. What values guide their decisions? Do they
seek to provide maximum good to the largest
number of citizens? Do they seek to provide a
basic level of good to all? Do they seek to meet
the demands of special groups of needy patients?
In determining bene fi ts, who speaks for the inter-
ests of children? In the case of costly procedures
that bene fi t very few persons, political advocacy
seems unlikely to be a large force in guiding
insurance coverage. Principles of social justice
do come into play when decision makers consider
how much pain would occur if care were denied,
or how much gain can be achieved by funding
treatment. These distinctions are dif fi cult and
cost-bene fi t thinking may be used in making such
funding decisions.
Decisions about making extraordinary expen-
ditures on highly technical craniofacial surgery
and care may be deliberated by health agencies
and insurers. In group policies, the cost of cata-
strophic illness or a major birth defect in a single
member or child may affect the entire group and
its cost of insurance. There are many examples of
health insurance programs that seek to avoid
expensive claims or that withdraw continued cov-
erage from the neediest claimants.
In calculating coverage and policies, insur-
ance companies or systems are not likely to con-
sider how the cost of a craniofacial repair might
compare with the costs of maintaining a person’s
disability. Such companies seek to minimize their
costs and are not responsible for dealing with the
long-term social consequences of a disability.
Governmental agencies, on the other hand, may
ask questions about long-term patient function.
They may consider whether the person will be
self-supporting and self-maintaining, in the long
term, after care. A decision to provide costly care
to a child may re fl ect the perception that treat-
ment will reduce the burden to society of lifelong
assistance or maintenance.
Another approach to cost-bene fi t consider-
ations recognizes the social cost of living around
deformed persons, who may inadvertently chal-
lenge social norms of acceptability. Some condi-
tions elicit negative social responses that provoke
society to cover up the difference or reduce the
deviance. It is in this fashion that signi fi cant
social pressure may actually be placed on the

962
R.P. Strauss
person with a major craniofacial condition to par-
ticipate in and agree to reconstruction. The per-
son is expected to “be a good patient” and seek
amelioration.
Perhaps, the most dif fi cult resource distinc-
tion occurs around the relative worth of social
investment in various diagnoses. Why is a cleft
palate repair seen uniformly as cost effective,
while some question a kleeblattschädel cranio-
synostosis repair? Is it that we expect a better out-
come from the cleft surgery? Or is it that the
frequency of clefts demands their repair, while
the scarcity of major craniofacial syndromes does
not? If we can afford as a society to provide cleft
repairs, why then not afford craniofacial repairs?
Clearly, questions of the magnitude of cost exist.
The costs as calculated cannot be merely the
direct costs of several years of surgical care;
rather, they must also include the costs of paren-
tal work loss, of special education, of rehabilita-
tion services, of mental health assistance, of lost
patient productivity, and of mental anguish. These
costs are generally immeasurable, yet they con-
stitute real components of the losses realized.
In the current context of limiting health dol-
lars and resources, it is predictable that some
rationing decisions will occur. The deciding prin-
ciples to be applied are unclear, but they will
determine who will live, who will live well; who
will be cared for and who will languish; who will
receive bene fi t and who will suffer. These dis-
tinctions will directly affect how care for major
craniofacial conditions will be delivered.
49.3
Justice and the Distribution
of Resources
This chapter has sought to review the social and
ethical rami fi cations of craniofacial surgery for
children born with major craniofacial conditions.
The remarkable tools of craniofacial surgery and
neonatal life support/intensive care have made it
possible for many more children with serious
defects to live. Their survival has raised questions
about how much the society is willing to invest in
their care, especially considering the impact that
even a repaired defect will have on the patient’s
quality of life. The availability of prenatal diagno-
sis and abortion may present additional choices to
parents and clinicians. After a birth of a child with
a major birth defect, the intense expense and the
high degree of clinical activism involved heighten
our perception of the costs of craniofacial surgery.
The desire to utilize new techniques, to advance a
discipline, and to provide patient bene fi ts may
affect clinician decisions. In the context of these
often con fl icting social currents, many questions
about justice and the distribution of resources
arise (National Center For Health Statistics 1990 ) .
Should society invest large amounts of effort and
resources in costly corrections of rare and dis-
abling conditions, or should it spend limited
health dollars on population-based preventive
efforts (Beauchamp and Childress 1994 ; Zuger
2004 ) ? Decisions about rationing care will arise
in the context of limited resources and in societies
where demands on the health system exceed the
system capacity. Craniofacial care poses many
social, ethical, and health policy issues (Demause
1977 ; Feragen et al. 2009 ; Hall 1992 ; Jonsen et al.
1992 ; Murray and Botkin 1995 ; Roberston 1986 ;
Strauss et al. 1995 ; Ward 1995 ; Wertz and Fletcher
1989 ; Wexler 1995 ) for health professionals,
parents, and the community at large.
Acknowledgement Vanessa White is acknowledged for
her assistance in the preparation of this manuscript.
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