T H E I M PA C T O F N S O C O N FA M I LY Q o L

ARTICLE

ABSTRACT The impact of nonsyndromic oral clefts

This study aimed to assess the impact of
nonsyndromic oral cleft (NSOC) on fam-
ilies’ quality of life (QoL) using the
Brazilian version of the Family Impact
Scale (B-FIS). A hospital-based case–
control study was conducted with
NSOC cases and unaffected controls
recruited at Dental Clinic in Federal
University. The mean B-FIS scores were
10.32 (SD 6.53) and 5.04 (SD 4.73),
while the median scores were 9.00 and
3.50 (p < .05 Wilcoxon test), respec-
tively, in case and control group. The
“parental/family activity” subscale had
the highest impact average score in
case (5.62 SD 3.76) and control group
(3.00 SD 3.08) (p < .05 Mann–Whitney
test). The types of cleft with the most
impact were cleft lip (12.00 SD 8.98)
and cleft lip with cleft palate (11.06 SD
6.74). NSOC affects the QoL of families
with children who have this condition;
however, there were no remarkable
­differences between the groups.
KEY WORDS: nonsyndromic cleft lip
with or without cleft palate, oral health,
quality of life
on family quality of life
Leonardo Santos Antunes, DDS, MS, PhD;1 Caroline Pelagio Raick Maués,
DDS;2 Mariana Rocha Nadaes, DDS;2 Marcelo Castro Costa, DDS, MS, PhD;3
Erika Calvano Küchler, DDS, MS, PhD;4 Lívia Azeredo Alves Antunes, DDS,
MS, PhD1*
1Department of Specific Formation, School of Dentistry, Fluminense Federal University, Nova Friburgo, RJ,
Brazil; 2School of Dentistry, Fluminense Federal University, Niterói, RJ, Brazil; 3Department of Pediatric
Dentistry and Orthodontics, School of Dentistry, Federal University of Rio de Janeiro, RJ, Brazil;
4Cell Therapy Center, Unit of Clinical Research, Fluminense Federal University, Niterói, RJ, Brazil.
*Corresponding author e-mail: liviaazeredo@yahoo.com.br; liviaazeredo@gmail.com
Spec Care Dentist 34(3): 138-143, 2014
Int r od uct ion
The most common craniofacial malformation affecting lips and the oral cavity is non-
syndromic oral cleft (NSOC).1 Overall, available findings indicate that NSOC occurs in
approximately 1 out of every 700 live births, with ethnic and geographic variations.2 In
addition, infants with NSOC have an impaired ability to feed, resulting in malnutrition
and weight gain problems in the first weeks of life.3 Individuals with NSOC need surgi-
cal, nutritional, dental, speech, medical, and behavioral interventions from infancy to
adulthood. Clearly, this condition represents a major public health burden in terms of
both medical costs and emotional impact on patients and their families.4

The World Health Organization health-related QoL (OHRQoL) must be

(1948) defines health as not only the
absence of disease but also the presence
of factors that enhance physical, mental,
and social well-being. The Quality of Life
(QoL) group defined QoL as “an individ-
ual’s perception of their position in life in
the context of culture and value systems
in which they live and in relation to their
goals, expectations, standards, and con-
cerns.”5
Thus, when dealing with individuals
with NSOC, it is vital that we consider
the impact of their condition on QoL,
since only treating physical symptoms do
not permit the individual to enjoy his
health fully. This evaluation follows a
new concept of health that takes a holis-
tic view of the individual and considers
the QoL in a multidimensional scope.6
According to contemporary concepts
of child health, the measurement of oral-
from the perspective of the child and the
family for the following reasons7,8: (I) the
family’s perspective of the impact of a
child’s condition is very important due to
the dependence between caregiver and
the child; (II) this caregiver is responsible
for informing the impact of the disease
and its treatment (and also the need for
additional care and treatment,
if applicable) on the functions of the
family; (III) the central role played by the
family in a child’s health; (IV) the likeli-
hood that chronic illness will impact the
child’s family to some degree; (V) the fact
that health care interventions often
address parental needs and concerns as
well as child’s; and (VI) the fact that the
parental reports of a child’s health may be
influenced by the degree to which the
parent is physically or emotionally
affected by the child’s condition. So, the

138 S p e c C a r e D e n t i s t 3 4 ( 3 ) 2 0 1 4 © 2013 Special Care Dentistry Association and Wiley Periodicals, Inc.
DOI: 10.1111/scd.12046

scd12046.indd 138 26/03/14 8:56 AM


family impact data are essential in order
to assess this “caregiver-burden” bias.9
The issue of the impact of children’s
oral conditions on families was first
raised by Sheiham and Croog (1981).10
The development and evaluation of a
Family Impact Scale (FIS) for oral health
research was done by Locker et al.
(2002).9 The FIS is a questionnaire
developed to determine the impact of
children’s oral and oral-facial conditions
on the family. This then serves as out-
come measures in clinical trials or
evaluation research.9
To the best of our knowledge, there
are no reports in the literature on the
effect on a family’s QoL from having chil-
dren with NSOC. So, the purpose of this
study was to evaluate the impact of
NSOC on families’ QoL using the
Brazilian version of FIS (B-FIS) with an
appropriate age-related comparison
group. The null hypothesis states that
the families of children with NSOC
would experience similar emotional and
psychological impacts on their QoL
when compared with the control group.
M ater ials and me tho ds
Ethical approval was obtained from the
Human Ethics Committee of the Health
Department of the city of Rio de Janeiro,
Rio de Janeiro, Brazil (113/09). Informed
consent was obtained from all participat-
ing families.
We used a convenience sample. The
NSOC group consisted of 50 individuals
receiving dental care at a public hospital
that is also a center for oral cleft rehabili-
tation in Rio de Janeiro, Brazil. None of
the subjects were diagnosed with median
cleft. To further reduce possible etiologi-
cal heterogeneity, we excluded those
clefts with additional unspecified multi-
ple malformations. The control group
consisted of 50 healthy, unrelated indi-
viduals, recruited at the Pediatric Dental
Clinic in Federal University of Rio de
Janeiro, Brazil. The control individuals
were selected to ensure that they
matched the cleft group in age (4–17
years), gender, geographic distribution,
and socioeconomic status. Both groups
have similar access to oral care. Both
Antunes et al. S p e c C a r e D e n t i s t 3 4 ( 3 ) 2 0 1 4 139
scd12046.indd 139
T H E I M PA C T O F N S O C O N FA M I LY Q o L
Table 1. FIS questionnaire divided into subscales.
Subscale Questions
Parental/family Have you or the other parent taken time off work?
activity Has your child required more attention from you or the other parent?
Have you or the other parent had less time for yourselves or other
family members?
Has your sleep or that of the other parent been disrupted?
Have family activities been interrupted?
Parental Have you or the other parent been upset?
­emotions Have you or the other parent felt guilty?
Have you or the other parent worried that your child will have fewer life
opportunities?
Have you felt uncomfortable in public places?
Family conflict Has your child argued with you or the other parent?
Has your child been jealous of you or other family members?
Has your child’s condition caused disagreement or conflict in the family?
Has your child blamed you or the other parent?
Financial burden Has your child’s condition caused financial difficulties for your family?
institutions are located in the northern the final score could vary from 0 to 56,
part of the city of Rio de Janeiro. The for which a higher score denoted a
institutions where the subjects were greater degree of the impact of the child’s
recruited are treatment centres and are oral conditions on the functioning of the
located in the Southeast of Brazil, the family as a whole.
most densely populated and industrial- This instrument was adapted
ized region of the country. ­cross-culturally and validated for use on
Individuals born with clefts were Brazilian children’s families, exhibiting
examined clinically and their medical satisfactory psychometric properties.11
records checked to confirm the cleft type The measurement of QoL was
(cleft lip—CL, cleft lip with cleft ­performed by one trained examiner.
palate—CLP, or cleft palate only—CP).
Data management and
QoL assessment ­statistical analysis
QoL was measured using the B-FIS, All data were analyzed using the
which has 14 items distributed into three Statistical Package for the Social Sciences
subscales: parental/family activity, paren- (SPSS 16.0). Data for individuals for
tal emotions, and family conflict. The whom the matching case or control had
financial burden subscale is the only one not responded were dropped from the
that is evaluated separately, since it is analysis. The level of statistical signifi-
comprised by a single item. The ques- cance was set at p < .05.
tions refer only to the frequency of events The relative frequency (%) of sample
in the previous 3 months (Table 1). (children and their families) characteris-
Response options for the four domains tics was obtained. Psychometric
and the respective scores were: “never = properties of B-FIS for the population
0,” “once or twice = 1,” “sometimes = 2,” analyzed were assessed through internal
“often = 3,” “every day or almost every consistency and test-retest reliability.
day = 4.” The FIS scores are calculated by Internal consistency reliability was
summing all of the items scores. Scores assessed by means of Cronbach’s Alpha.
for each of the four domains can also be Test-retest reliability was assessed by
calculated. Since there were 14 questions, means of the intraclass correlation
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 T H E I M PA C T O F N S O C O N FA M I LY Q o L

50 were controls. The characteristics of

Table 2. Sample characteristics.
Mean age (SD)
Gender (%)
Female
Male
Respondents (%)
Father/mother
Grandfather/grandmother
Others
c­ oefficient (ICC), using a new conveni-
ence sample of families with children
born with cleft (n = 12).
The scores of the B-FIS index were
calculated using the additive method—
summing the numeric response codes for
each item. Means and medians were
obtained for items overall and s­ ubscale
scores for the two designations (case and
control) and for type of clefts. Since the
items were scored using the ordinal scale,
nonparametric statistical procedures
(Mann–Whitney’s test) were used.
the population studied are summarized
Case (n = 50) Control (n = 50)
in Table 2.
9.52 (4.02) 8.76 (2.60) The psychometric properties of the
instrument gave a satisfactory reliability
20 (40%) 20 (40%) with Cronbach’s alpha of .71 for case and
30 (60%) 30 (60%) .70 control and good ICC of .84 in
­test-retest.
The mean B-FIS scores were 10.32
34 (68%) 37 (74%) (SD 6.53) and 5.04 (SD 4.73), while the
15 (30%) 11 (22%) median scores were 9.00 and 3.50
1 (2%) 2 (4%) (p < .05, Wilcoxon test), in case and
­control groups, respectively (Table 3).
When the subscales (parental/family
Univariate statistics were calculated activity, parental emotions, family con-
and bivariate analyses were used to flict, and financial burden) were analyzed
explore the data. Odds ratio calculations in the present study, the parental/family
and Chi-square or Fisher’s exact tests at a activity subscale had the highest score
level of significance of .05 were used. For impact average in case (5.62 SD 3.76)
this, instrument scores were d­ ichotomized. and control groups (3.00 SD 3.08)
The impact on OHRQoL was classified as (p < .05, Mann–Whitney test).
absent (FIS = 0) or present(FIS ≥ 1). The types of cleft with high impact
were CL (12.00 SD 8.98) and CLP
(11.06 SD 6.74). Considering all the
R es ul t s ­subscales, all types of cleft presented
Of 100 individuals included in this study, more impact on parent/family activity
50 were individuals born with clefts and (Table 4).

Table 3. Comparison of the mean and median between case and control group.
FIS (variance) Case Control p value*
Mean (SD) Median (Q1–Q3) Mean (SD) Median (Q1–Q3)
Total scale (0–56) 10.32 (6.53) 9.00 (4.75–15.25) 5.04 (4.73) 3.50 (0.00–9.25) p < .05
Subscale
Parent/family activity (0–20) 5.62 (3.76) 5.00 (2.00–8.00) 3.00 (3.08) 2.50 (0.00–6.00) p < .05
Parental emotions (0–16) 3.16 (2.70) 3.00 (0.00–4.25) 0.96 (1.36) 0.00 (0.00–2.00) p < .05
Family conflict (0–16) 0.62 (1.12) 0.00 (0.00–1.00) 0.76 (1.54) 0.00 (0.00–0.25) NS
Financial burden (0–4) 0.92 (1.15) 0.00 (0.00–2.00) 0.32 (0.68) 0.00 (0.00–0.00) NS
Note: *Mann–Whitney test; NS = not significant (p > 0.05); Bold font indicates statistical significance; Q1: 1st quartile/Q3: 3rd quartile.

Table 4. B-FIS mean according to scale and subscales and type of clefts.
Cleft type Control (n = 50)
FIS CL (n = 4) CP (n = 15) CLP (n = 31) All Cleft (n = 50)
Mean (SD) Mean (SD) Mean (SD) Mean (SD)
Total scale 12.00 (8.98) 8.33 (5.31) 11.06 (6.74) 10.32 (6.53) 5.04 (4.73)
Subscale
Parent/family activity 6.50 (4.20) 4.26 (2.98) 6.16 (3.98) 5.62 (3.76) 3.00 (3.08)
Parental emotions 3.50 (2.88) 3.26 (2.48) 3.06 (2.88) 3.16 (2.70) 0.96 (1.36)
Family conflict 1.50 (1.73) 0.26 (0.70) 0.67 (1.16) 0.62 (1.12) 0.76 (1.54)
Financial burden 0.50 (1.00) 0.53 (0.99) 1.16 (1.21) 0.92 (1.15) 0.32 (0.68)

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Table 5. B-Fis impact according to categories between case and control subjects.
B-FIS
Total scale Fis = 0
Fis ≥ 1
Subscale
Parent/family activity Fis = 0
Fis ≥ 1
Parental emotions Fis = 0
Fis ≥ 1
Family conflict Fis = 0
Fis ≥ 1
Financial burden Fis = 0
Fis ≥ 1
Note: *Fisher’s exact test; **Chi-square. Bold font indicates statistical significance.
In Table 5, according to the catego-
ries, the NSOC group was associated
with a greater negative impact on the
families’ QoL in the following subscales:
parent/family activity (p < .01), parental
emotions (p < .01) and financial burden
(p = .03).
D i s cu s s ion
NSOC has great epidemiological impor-
tance in the context of public health,
since it is the most common craniofacial
malformation affecting the lips and oral
cavity.1 Therefore, the detection of the
impact of NSOC on QoL is of paramount
importance not only for the development
of measures to address NSOC, but also to
emphasize the psychological and social
impact this condition has on the family
and the child.
There is no specific instrument to
evaluating HRQoL or OHRQoL in
NSOC patients. In a systematic review
by Klassen et al. (2012),12 HRQoL con-
cepts were measured using 29 different
questionnaires. According to Klassen et
al. (2012),12 to adequately measure the
QoL of oral cleft patients, a scientifically
sound and clinically meaningful patient-
reported outcome (PRO) instrument
that specifically addresses the unique
issues of oral cleft patients is required.
This type of instrument includes con-
tent that is more relevant to a given
Antunes et al. S p e c C a r e D e n t i s t 3 4 ( 3 ) 2 0 1 4 141
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T H E I M PA C T O F N S O C O N FA M I LY Q o L
Case (%) Control (%)
1 15
49 35
3 19
47 31
13 30
37 20
36 38
14 12
29 39
21 11
patient group and is therefore better at
detecting and measuring change in
health.13 The lack of a PRO tool specific
to children with oral-facial clefts was
identified as an important research gap
in the January 2006 workshop entitled
“Prioritizing a Research Agenda for
Orofacial Clefts,” held by the National
Center on Birth Defects and
Developmental Disabilities at the
Centers for Disease Control.14
The FIS was the instrument used in
this study.9 It has been validated in
Portuguese.11 Even though we are
using a questionnaire with proven
­psychometric properties, it is important
to assess it in order to ensure good
methodological quality of the study.15
Therefore, the present instrument was
also tested for validity and reliability,
showing ­satisfactory internal validity
and good understanding of the
­questionnaire, which was detected after
replication of the instrument. The afore-
mentioned data further demonstrate
that the Brazilian version of FIS scores
can satisfactorily be used in detecting
impact on QoL in families with children
suffering from NSOC. In fact, we sug-
gest that this instrument be applied to
other populations of similar families to
detect the impact of this condition on
QoL.
In literature, there are some studies
evaluating the impact of NSOC on the
OR (95%IC) p value
0.05 (0.00–0.37) p < .01*
0.10 (0.02–0.42) p < .01*
0.23 (0.09–0.59) p < .01**
0.81 (0.30–2.18) p = .64**
0.39 (0.15–1.01) p = .03**
OHRQoL of children and adolescents
but not one of them evaluates the
impact of NSOC on their family. Geels
et al. (2008)16 used child oral health
impact profile (COHIP) to evaluate
OHRQoL comparing the children and
the parents’ perception. They found a
high level of agreement. Locker et al.
(2005)17 use CPQ 11–14 to assess the
OHRQoL of children with oral-facial
conditions, comparing them to patients
with dental caries. The oral-facial group
had slightly higher scores on the CPQ
11–14 than the dental group. Ward
et al. (2012)18 used COHIP to evaluate
OHRQoL, comparing the children/car-
egivers’ perception and a control group.
We found that children with NSOC
generate some impact on NSOC families
OHRQoL when compared to control
group (10.32 × 5.04). This fact could
indicate increased dedication by the
family toward their children with cleft,
leaving less time for their own activities.
This devotion, according Topolski et al.
(2005),19 was an attempt to meet the
emotional needs of their children, since
they suffer prejudicial attitudes by their
peers, mainly due to facial aesthetics and
difficulty in speech.
The difference in scores was statisti-
cally significant, however, the OHRQoL
of the families with children who have
oral-facial conditions was not markedly
different from that of the children with
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 T H E I M PA C T O F N S O C O N FA M I LY Q o L
dental conditions in our study. Although
it is appropriate to use those who were
unaffected by NSOC from the same
­geographic distribution and socioeco-
nomic status for the control, the
relatively high variance in both the
NSOC and control groups obviously
reduced the chances of more significant
results. According to Locker et al.
(2005),17 the majority of these children
are well adjusted and able to cope with
the adversity they experience as a result
of their conditions. This leads to the con-
clusion that although the oral-facial
group may encounter more challenges in
daily life, their overall QoL is no differ-
ent from that of children with more
common oral conditions.
There are two ways to explain the
lack of a marked difference in the
­health-related QoL of the dental and
oral-facial groups. Both suggest that the
latter have received high-quality clinical
and psychosocial care at the clinical
­setting where they were recruited. This
clinical setting uses a team approach
involving clinicians, psychologists, and
social workers who provide social and
emotional support to children and their
families from birth, as well as surgical
and orthodontic intervention. Also,
­cultural differences concerning the
­relative importance of perceived
­physical attractiveness of the face
­compared with the body may play a
role. In tropical climates, like Brazil,
where the body is often more visible
than in colder ­climates, less attractive
faces (with or without NSOC) may not
be stigmatized as much as in other
countries.
Questionnaires designed to assess
the impact of OHRQoL are specific
instruments for application in various
areas including political action, research,
and public health clinics, which make
their use extremely important.20–22
Thus, the purpose of this study is to
extrapolate the results to the population
studied in order to generate data to
­facilitate the development of health
­promotion strategies with a holistic view
of the patient’s treatment plan, including
not only the clinical treatment of NSOC,
but also ­covering the psychosocial
142 S p e c C a r e D e n t i s t 3 4 ( 3 ) 2 0 1 4 The impact of NSOC on family QoL
scd12046.indd 142
c­ ontext. This research can be considered
preliminary because it investigates the
impact of NSOC on the QoL of the
­families of c­ hildren with NSOC by means
of a case–control study. For further
research, we suggest a representative
sample with a higher number of partici-
pants in order to make the extrapolation
of our results more reliable, since our
study used a convenience sample with a
small number.
This research showed that the FIS
can be used in Brazilian population with
NSOC to evaluate the impact on family
QoL, since it demonstrated satisfactory
psychometrics properties. The score
impact detected in the present study can
be used as a reference, in comparison
with other future studies.
C oncl us ion
With the results obtained in this
research, we can observe that children
with NSOC generate some impact on
their families’ OHRQoL when compared
to control group; however, we were not
able to observe a strong difference in the
health-related QoL observed between the
groups.
A ck now l ed gem ent s
We are indebted to the participants of the
study. The authors thank Sarah Vinski
for English editing Assistance.
C onf l ict of int er es t
None declared.
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