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Zhang et al

Original Paper

Factors Associated With Perception of Stigma Among Parents ofChildren With Cleft Lip and Palate: Cross-Sectional StudyYanan Zhang1, MD; Xinwen Zhang1, BS; Jinzhuo Jiang2, MD; Wanhua Xie3, PhD; Daoman Xiang1, PhD
1Ophthalmology and otorhinolaryngology Department, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, Guangdong
Provincial Clinical Research Center for Child Health, Guangzhou, China
2

College of Engineering, South China Agricultural University, Guangzhou, China

3

Outpatient Department, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, Guangdong Provincial Clinical ResearchCenter for Child Health, Guangzhou, China

Corresponding Author:
Wanhua Xie, PhD
Outpatient Department
Guangzhou Women and Children's Medical Center, Guangzhou Medical University
Guangdong Provincial Clinical Research Center for Child Health
9 Jinsui Road
Guangzhou, 510623
China
Phone: 1 13725370379
Fax: 1 2038076020
Email: xiewanhua1@126.com

Abstract
Background: Parents of children with cleft lip with or without cleft palate (CL/P) often face stigmatization, which has asignificant impact on their quality of life and mental health. However, to date, there is a lack of comprehensive, multicenter
empirical research on parents of children with CL/P in China, particularly those with large-scale samples.
Objective: This study aimed to identify major factors that contribute to the perception of stigma experienced by parents ofchildren with CL/P.
Methods: A cross-sectional survey was conducted. A total of 104 parents of children diagnosed with CL/P in 2 hospitals wereselected by convenience sampling. Demographics and disease information, the Chinese Perception of Stigma Questionnaire, theCenter for Epidemiological Studies Depression Scale, and the Social Anxiety Scale were used in this study. Descriptive statistics,
t tests, and one-way ANOVA were used to compare the differences between participants’ demographic information and perceptionof stigma. Multivariable linear regression was performed to assess associations between demographic factors, social anxiety,depression, and perception of stigma.
Results: The mean scores for the dimensions of perception of stigma, depression, and social anxiety were 22.97 (SD 9.21),
38.34 (SD 8.25), and 22.86 (SD 6.69), respectively. Depression and social anxiety were positively associated with discrimination,
while surgery status was a negatively associated variable. Parents with a college education or higher had significantly lower levelsof perceived stigma compared to parents with a junior high school education (all P values <.05). These 4 factors explained 40.4%of the total model variance (F8=9.726; P<.001; R2=0.450; adjusted R2=0.404).
Conclusions: Our findings highlight a concerning trend of diminished quality of life among parents of children with CL/P.
Factors such as parents’ education level, surgery status, depression, and social anxiety are shown to influence the level of stigma
experienced. Implementing comprehensive nursing care and providing presurgical support are effective strategies for alleviating
parents’ social anxiety, reducing perceived stigma, and preventing depression.
(JMIR Form Res 2024;8:e53353) doi: 10.2196/53353
KEYWORDS
stigma; social anxiety; depression; parents of children with cleft lip and palate; cleft lip; cleft palate; cross-sectional study

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Introduction

Methods

Cleft lip with or without palate (CL/P) is a common congenital
developmental malformation affecting the oral and maxillofacial
region [1], with a global prevalence of about 1.62%-1.82% [2],and the highest prevalence in Asian countries [3]. There are
35,000 babies born with CL/P in China each year [4]. Palate
repair surgery is usually done when babies are 10 to 24 months
old, while lip surgery is generally performed at the age of 6months [3]. The management of CL/P is long-term, beginningfrom birth and continuing into the late teenage years. Children
born with CL/P not only face a visible facial disfigurement but
also have other challenges related to the cleft, such as feeding
difficulties, speech impediments, hearing issues, andpsychological problems, which may compromise their ability
to communicate effectively [5].

Study Design and ParticipantsChildren with CL/P are vulnerable to external discrimination
because of the malformation. A child with CL/P is subjected tobullying, rejection, and social isolation, sometimes even fromtheir own family [6]. The discrimination can have devastating
health and well-being impacts, not only on children affected
but also on people associated with the discriminated person.
Having a baby with CL/P has variable psychological impacts
on the parents [7]. Parents are often concerned about whether
their children will be accepted as they grow up, and parents’
psychological state directly affects their children’s perceptionof the condition [6,8,9]. Some studies [10,11] have indicatedthat the perception of stigma experienced by the mother andchildren is consistent. Numerous studies [2,12,13] have shownthat children with CL/P experience significant stigma, andlimited engagement in social activities suggests that the parentsof these children may experience high levels of stigma.
Furthermore, Parents are troubled by their children’s conditions
from birth to adulthood, while children often experience
discrimination after their cognitive development has matured.
Therefore, exploring ways to prevent these patients and theirparents from feeling stigmatized is urgently necessary.
Currently, research on the perception of stigma among parentsof children with CL/P is predominantly qualitative, emphasizing
the exploration of factors contributing to parental stigma.
However, there is limited reporting on assessments regarding
the level and variation in the perception of stigma. In particular,
there is no research on the perception of stigma in children with
CL/P in China. Previous studies [11,14] have shown thatsuperstition is the main reason for parents’ perception of stigma,
and only 40% of parents believe that genetic problems cause
CL/P. One study [11] found that 72% of mothers felt ashamedof having children with CL/P, and some even abandoned theirbabies. Therefore, it is crucial to examine the perception of
stigma among parents of children with CL/P, pinpoint thoseexperiencing heightened stigma levels, implement tailorednursing interventions based on sensitive factors, and mitigate
the likelihood of public incidents. This study aims to investigate
experiences of stigma in parents of children with CL/P andanalyze the contributing factors.

This cross-sectional study was conducted using a conveniencesample of parents of children with CL/P, recruited from 2
hospitals in southern and western China (from the cities ofGuizhou and Guangzhou) between June 2020 and January 2023.
All parents of children who met the inclusion and exclusion
criteria for surgical treatment in the Department of Stomatology
were invited to complete a questionnaire.
The inclusion criteria were as follows: parents of children (aged<15 years) with CL/P undergoing nasoalveolar molding therapy
at the Department of Stomatology. These parents were invited
to complete an anonymous questionnaire.
Exclusion criteria included parents of infants with additional
birth defects or medical conditions, those with genetic diagnoses(eg, Down syndrome or Trisomy 21 syndrome), and individuals
unable to complete the questionnaires due to critical illness ormental disorders.

Data Collection
After the doctors introduced the operational procedure or thenurses introduced the environment of the wards, the participants
were invited to fill in the questionnaires. Uniform instructions
were used to explain the study aims and their relevance to theparticipants. The participants were informed that the study wasanonymous and voluntary and that they could quit anytime.

Variables and Instruments
The demographic data were collected via a questionnaire. Threequestionnaires with good reliability and validity were used tocollect the data on stigma, social anxiety, and depression.

Demographics Questionnaire
The demographic information questionnaire was designed byresearchers after consulting the literature and mainly included
parents’ age, role (father or mother), education level, andworking status as well as children’s age, sex, type of CL/P,
surgery status, and number of surgeries.

Disability Discrimination Perception Scale
Stigma was assessed using the Disability Discrimination Scale(DDPS), initially developed by Liu and Shen [15]. The
questionnaire comprises 10 items (eg, “When participating in
activities, I feel that people around me would not talk to me andavoid me”), designed to explore the perception of stigma amongindividuals with disabilities in their daily lives. It is also suitablefor children aged 10 to 16 years. Respondents rated each item
on a 5-point scale, ranging from 1 (very inconsistent) to 5(completely consistent). Higher total scores indicate a greater
perception of stigma, with scores ranging from 10 to 50. The
scale demonstrates good internal consistency, as evidenced bya Cronbach α reliability of 0.89. The criterion validity was 0.47,
indicating good validity [15].

The Center for Epidemiological Studies Depression Scale
The level of depression was measured by the Center for
Epidemiological Studies Depression Scale (CES-D) [16]. CES-D

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Zhang et alconsists of 20 items (eg, “I was bothered by things that usually
don’t bother me”), and each item is scored from 0, indicating“rarely or none of the time (less than 1 day)” to 3, indicating“most or all of the time (5-7 days).” The total score of all items
ranges from 12 to 60, with higher scores indicating a greater
likelihood of depression; a total score of 15 points or lessindicates no depression; 16 to 19 points indicate possible
depression; and a score of 20 points or higher indicates definite
depression [16]. The scale is suitable for screening people withdepressive symptoms and can also be used to assess the severityof depressive symptoms. The Cronbach α of CES-D was 0.87,
indicating good internal consistency. The split-half reliability
was 0.85, the test-retest reliability was 0.70, and the criterion
validity was 0.75. Therefore, CES-D had good reliability andvalidity.

samples 2-tailed t test and ANOVA were used for the intergroupcomparisons of parameter changes. Pearson analysis was used
to examine the relationship between perception of stigma, social
anxiety, and depression. Multiple linear regression was
performed to assess the association between demographic
factors, anxiety, depression, and perception of stigma. The
significance level was set at 5%.

The Interaction Anxiousness Scale

Results

The Interaction Anxiousness Scale (IAS) was used to assess
the subjective propensity to experience social anxiety
independent of behavior [17]. The IAS contains 15 self-reported
items (eg, “I want to be confident in social situations more”),
which are answered on a 5-point scale, ranging from 1 (veryinconsistent) to 5 (completely consistent). Total scores range
from 15 to 75, with higher scores indicating higher levels ofsocial concern. The Cronbach α reliability of the scale was 0.87,
the test-retest reliability was 0.80, and the criterion validity was
0.48. In this study, according to the actual situation, 7 items
related to sexual life were deleted, and 8 items were selected
for evaluation. The reliability of the scale was retested, andCronbach α reliability was 0.68, indicating acceptable reliability.

Statistical Analysis
IBM SPSS Statistics (version 25.0; IBM Corp) for Windows,
was used for statistical analysis. Standard descriptive statisticalvalues (means and SDs) were calculated. The independent

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Ethical Considerations
The study was approved by the Ethics Committee for ClinicalStudies at Guangzhou Women and Children's Medical Center,
Guangzhou Medical University (NO.2022268A01). This study
obtained informed consent from participants, who had the option
to withdraw at any time. The research data were collectedanonymously.

In this study, a total of 110 questionnaires were distributed. Of
the collected questionnaires, 104 were complete, resulting in a
94.6% response rate.

Participant Characteristics
Most participants were mothers aged 25-30 years andunemployed, with a high school diploma. Their children had
CL/P, and surgery had not been performed. Unemployed parents
constituted 71% (n=74) of the sample, with nearly half (n=14,47%) of the employed parents being self-employed. Over half
(n=61, 59%) of the respondents had completed high school.
The children’s average age was 3.06 (SD 2.02) years. A totalof 73 (72%) children had not undergone surgery, while most of
those who had done the surgery (n=20, 65%,) had undergone
the procedure twice. A descriptive analysis of the generaldemographics, work-related characteristics, and disease-related
information of the participants is shown in Table 1.

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Table 1. Demographic characteristics of the participants (N=104).
Variable and categoryn (%)

Age (years)
18-25

30 (29)25-30

42 (40)30-35

26 (25)

＞35

6 (6)

Role
Father39 (38)Mother65 (63)

Educational backgroundPrimary school or lower

6 (6)Junior high school

23 (22)

Senior high school

61 (59)College

7 (7)Bachelor’s degree or higher

7 (7)Working status
Unemployed74 (71)

Working

30 (29)

Occupation
Unemployed74 (71)

Independent management

14 (14)Enterprise unit

9 (9)Public service unit

7 (7)Children’s sex
Male

60 (58)

Female44 (43)

Condition type
Cleft lip

36 (35)

Cleft lip and palate

68 (65)

Number of surgeries0

73 (70)

1

7 (7)

2

20 (19)≥3

4 (4)Status of surgery
After surgery

73 (70)

Before surgery

31 (30)Prevalence of Stigma, Social Anxiety, and Depression
The mean scores for the dimensions of perception of stigma,
depression, and social anxiety were 22.97 (SD 9.21), 38.34 (SD8.25), and 22.86 (SD 6.69), respectively. Additionally, 38%(n=40) of the participants exhibited a perception of stigma level
beyond the moderate level, and every participant experienced
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evident depression, with 72% (n=75) displaying average levelsof social anxiety.

Univariate Analyses of Factors Associated With Stigma
The demographic data, including parents’ age, role, educationlevel, and working status as well as the children’s age, sex, CL/P
type, surgery status, and the number of surgeries, were analyzed
using a one-way ANOVA and t tests. The results of the
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Kolmogorov-Smirov test suggested that the continuous variables
were reasonably and normally distributed. The t tests revealed
that the parents whose children had undergone surgery
experienced lower stigma levels compared to those whosechildren had not undergone surgery (P=.01). The perception of

stigma scores differed significantly among parents having
different educational backgrounds (P<.001) and between those
whose children underwent different numbers of surgeries(P=.04). The specific results are detailed in Table 2.

Table 2. Univariate analyses of factors associated with stigma (N=104).
Variable and category

Mean (SD)Age (years)
18-25

25.74 (9.93)

25-30

21.14 (8.46)

30-35

22.24 (8.30)>35

23.50 (12.06)

Role
Father

23.28 (9.62)

Mother

22.69 (8.96)

Education backgroundPrimary school or lower

22.50 (9.89)

Junior high school

24.52 (8.94)

Senior high school

24.21 (9.34)

College

16.14 (2.73)

Bachelor’s degree or higher

13.43 (1.81)

Working status
Unemployed

24.00 (8.84)

Working

20.23 (9.56)

Occupation
Unemployed

24.00 (8.84)

Independent management

19.43 (8.49)

Enterprise unit

21.89 (10.95)

Public service unit

19.71 (10.95)

Children’s sex
Male

23.55 (9.47)

Female22.05 (8.78)

Condition type
Cleft lip

23.33 (8.91)

Cleft lip and palate

22.69 (9.36)

Number of surgeries
024.34 (9.49)

1

17.57 (7.28)

2

18.95 (7.28)≥3

26.00 (7.07)

Status of surgeryaBefore surgery

24.34 (9.49)

After surgery

19.55 (7.47)

F test (df)t test (df)

P value

1.58 (3)

—a

.20

—

0.32 (102).75

3.62 (4)

—

<.001

—

2.02 (102).06

1.36 (3)

—

.26

—

0.83 (102).41—

0.34 (102).74

2.94 (3)

—

.04

—

2.50 (102).01

Not applicable.

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Correlation Analysis With Perception of Stigma, Social
Anxiety, and Depression
The results showed that the perception of stigma was related tosocial anxiety (r=0.54; P<.001) and depression (r=0.39;
P<.001); there was also a correlation between depression andsocial anxiety (r=0.30; P=.002). The specific results are shownin Table 3.

Table 3. Correlation analysis (Pearson r) with stigma, social anxiety, and depression. At P=.01 (2-tailed), the correlation was significant.
Variable

StigmaDepression

Social anxiety

r

1

0.39

0.54

P value

—a

<.001

<.001

r

0.39

1

0.30

P value

<.001

—

.002

r

0.54

0.30

1

P value

<.001

.002

—

StigmaDepression

Social anxietya

Not applicable.

Regression Analyses of Stigma
With the perception of stigma as the dependent variable,
depression, social anxiety, and demographic data withdifferences between groups were included in the model as
independent variables, and multiple linear regression analyses
were performed. In the perception of stigma model, depression,social anxiety, educational background, and status of surgery
were significant correlates explaining 40.4% of the total model
variance (F8=9.726; P<.001; R2=0.450; adjusted R2=0.404).

Depression and anxiety were positively associated withdiscrimination, while the status of surgery was a negatively
associated variable. Parents of children with a college educationor higher had significantly lower levels of perceived stigma
compared to parents with a junior high school education (all P
values <.05). There were no significant differences in theperceived stigma levels between parents with elementary or
high school education and those with junior high school
education (all P values >.05). The specific results are shown in
Table 4.

Table 4. Regression analyses of stigma.
Variables

Unstandardized coefficients (B)SEt test (df)

P valueConstant value

0.10

4.36

0.02 (95).98

Status of surgery

–5.85

2.22

–2.64 (95).01

Number of operations

2.08

1.14

1.82 (95).07

Depression

0.26

0.09

2.93 (95)<.001

Social anxiety

0.52

0.12

4.48 (95)<.001

Primary school or lower

0.23

3.30

0.07 (95).95

Senior high school

–0.29

1.75

–0.16 (95).87College education

–6.53

3.09

–2.11 (95).04

Bachelor’s degree or higher

–7.21

3.15

–2.29 (95).02

Discussion
This study found that parents of children with CL/P experienced
a high level of perception of stigma and obvious social anxiety
and depression. In addition, parents’ education level, children’s
status of surgery, social anxiety, and depression affect parents’levels of perceived stigma. This study complements the gap inthe study of stigma perception in families of children with CL/P
in China. Parents in our study exhibited a higher level of stigma
perception. This could be attributed to the fact that most children
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underwent corrective surgery during infancy, making parents
more attuned to discrimination, as they believe that visual
impairments affect their children’s social lives. Thus, prevailingdata suggest that Chinese clinical nurses should pay attention
to the quality of life of parents of children with CL/P.
Our study showed that children’s sex and the type of CL/P could
not affect parents’ discrimination perception. This is consistent
with the findings of previous studies that reported no significant
difference in the level of discrimination perception between
patients of different sexes and CL/P types [10]. The results of
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our study showed no significant difference in the level of stigma
perception between fathers and mothers. Fathers also had a highlevel of stigma perception. However, previous studies [14,18]
suggested that mothers of children with CL/P experienced severe
stigma or discrimination while ignoring the possibility thatfathers also experienced high levels of discrimination. Twoother studies [19,20] pointed out that the main sources of stressamong fathers were the treatment process, feeding, and social
stigma. Therefore, in the care process of children with CL/P,
we should not only focus on the mother’s psychological state
but also consider the psychological state of the father. The
importance of family nursing should also be emphasized.
Parents of children with CL/P experience obvious depression
and anxiety. It is consistent with the findings of previous studies
reporting that parents of newborns with CL/P had significantly
higher levels of anxiety compared to control parents [20]. The
study findings are consistent with the current literature, as
depression or depressed mood in parents of children with CL/P
is a common phenomenon [21,22]. Kumar [23] reported thehighest incidence of depressive episodes, with 42% of parentsof children with CL/P aged >10 years showing a strongly orvery strongly elevated depressive disorders screening index.
Sommer [24] has pointed out that mothers of children with CL/P
believe their children have a negative impact on them. Their
negative views and emotions are mainly derived from the long
treatment times, the difficulty of treatment, the uncertainty of
the infant’s future condition, and the fear of rejection. Family
members of infants with CL/P experience stigma, anxiety, andworry, which may lead to individual negative emotions andcoping styles, produce adverse intergroup relations, reduce
subjective well-being and life satisfaction, and ultimately affect
the quality of family life. Relieving the stigma perceived byparents of children with CL/P is the first step to relieving
negative emotions like anxiety and depression in these parents.
Therefore, we should pay attention to the physical rehabilitationof children as well as the mental health of children and theirparents.
In this study, parents with junior high school education showeda high level of stigma perception, and the perception of stigma
was lower among parents with a college education or higher.
So far, no study has investigated the relationship between the
education level of parents who have children with CL/P andtheir stigma perception. A previous study [6] showed thatcaregivers of children with attention deficit hyperactivity
disorder, who have higher education levels, experience higher
levels of stigma. However, the results of this study wereinconsistent with a previous study, possibly because parents
with a junior high school education have less knowledge of thecondition and are more likely to focus on the appearance of
their children. The difference could potentially be attributed toZhang et al
the fact that parents with higher education levels are more likely
to access relevant information about the condition and feel more
confident about the later recovery process. Hence, health care
professionals should focus on parents with a high school
education or lower, monitor variations in their perceived stigma
levels, comprehend their psychological well-being acrossdifferent stages of their children’s lives, and offer tailored
psychological support.
Cleft surgery provides hope to those children and their parents,
offering the possibility of re-enrollment in schools, employment
opportunities, social acceptance, and improved prospects formarriage [6]. Undergoing surgery directly affects the level ofthe stigma perceived by parents. Parents of children without
surgery perceive a higher level of stigma, which is consistent
with previous studies [14] suggesting that parents of children
with CL/P show obvious negative emotions in the first 3 months
after the birth of their children without surgery. Therefore, theinitial 3 months emerge as a pivotal phase for health care
professionals to alleviate parental anxiety and deliver emotionaland educational assistance to parents of infants with CL/P.
Through comprehensive nursing and presurgical support,
mothers can enhance their early feeding capabilities, expedite
the bonding process with their infants, and facilitate the infants’swift recovery. Familiarity with CL/P beforehand serves todiminish maternal stress during initial interactions, mitigating
anxiety and perceptions of discrimination. Consequently,
encouraging mothers to engage with CL/P-related charitableorganizations or fostering interaction among children with CL/Pin shared spaces can effectively reduce anxiety and perceptionof stigma levels.
This study has several limitations. All the participants in thisstudy were family members of children who were willing toundergo surgery, and no family members of children who were
unwilling to undergo treatment were included, which may have
led to selective bias. Parents filled out the scale based on recall,which may introduce information bias. The sample size of thisstudy was small, as it was limited to hospitals only from 2
provinces with a large gap in economic levels between the 2places, and fewer fathers were included in the study. Futurestudies should increase the sample size and broaden the scopeof the investigation.
Our findings highlight a concerning trend of diminished qualityof life among parents of children with CL/P. Factors such as
parents’ education level, the status of surgery in children,
depression, and social anxiety are shown to influence the levelof stigma experienced by these parents. Implementing
comprehensive nursing care and providing presurgical support
are effective strategies for alleviating parents’ social anxiety,reducing perceived stigma, and preventing depression.

Acknowledgments
The authors are sincerely grateful to all the participants who responded to the questionnaires and all the nurses and doctors forsupporting this investigation.
This research was funded by the Guangzhou Municipal Science and Technology Bureau (grant 2023A03J0913 and 202103000088).

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Data Availability
The data sets generated and analyzed during this study are available from the corresponding author upon reasonable request.

Conflicts of Interest
None declared.

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Abbreviations
CES-D: Center for Epidemiological Studies Depression Scale
CL/P: cleft lip with or without cleft palate
DDPS: Disability Discrimination Scale
IAS: Interaction Anxiousness Scale

Edited by A Mavragani; submitted 04.10.23; peer-reviewed by M Sarhan; comments to author 29.12.23; revised version received
10.01.24; accepted 06.02.24; published 04.03.24
Please cite as:
Zhang Y, Zhang X, Jiang J, Xie W, Xiang D
Factors Associated With Perception of Stigma Among Parents of Children With Cleft Lip and Palate: Cross-Sectional StudyJMIR Form Res 2024;8:e53353
URL: https://formative.jmir.org/2024/1/e53353
doi: 10.2196/53353
PMID: 38437002©Yanan Zhang, Xinwen Zhang, Jinzhuo Jiang, Wanhua Xie, Daoman Xiang. Originally published in JMIR Formative Research(https://formative.jmir.org), 04.03.2024. This is an open-access article distributed under the terms of the Creative Commons
Attribution License (https://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction
in any medium, provided the original work, first published in JMIR Formative Research, is properly cited. The complete
bibliographic information, a link to the original publication on https://formative.jmir.org, as well as this copyright and license
information must be included.

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