YIJOM-4898; No of Pages 7

Int. J. Oral Maxillofac. Surg. 2021; xx: 1–7

https://doi.org/10.1016/j.ijom.2022.04.003, available online at https://www.sciencedirect.com

Clinical Paper
Cleft Lip and Palate

K. Lentge 1, F. Lentge 1, A.-N. Zeller,

Cleft lip and palate: the N.-C. Gellrich, F. Tavassol, P. Korn,
S. Spalthoff

psychological burden of affected Department of Oral and Maxillofacial

Surgery, Hannover Medical School,

parents during the first three Hannover, Germany

years of their children’s lives

K. Lentge, F. Lentge, A.-N. Zeller, N.-C. Gellrich, F. Tavassol, P. Korn, S.
Spalthoff: Cleft lip and palate: the psychological burden of affected parents during
the first three years of their children’s lives. Int. J. Oral Maxillofac. Surg. 2021;
xx: 1–7. © 2022 International Association of Oral and Maxillofacial Surgeons.
Published by Elsevier Inc. All rights reserved.

Abstract. The surgical treatment of cleft lip and palate (CLP) has been well
described in the literature. Nevertheless, little is known about the psychological
burden of affected parents. The aim of this study was to investigate the
psychological burden in parents of children with CLP within the first 3 years of
the children’s lives. A standardized questionnaire (Parenting Stress Index, PSI)
was administered to 33 parents of children with CLP to evaluate their
psychological burden. The corresponding interview was conducted independent
of any operative procedure during the yearly routine CLP consultation. Each
participant’s stress profile was assessed and compared with the average values of
parents with non-cleft children. Psychological stress was substantially increased
in all participants when compared to the parents of healthy children. This result
was highly significant (P < 0.001). Parent depression (P < 0.001) and child-
related requirements (P < 0.001) were the most critical subscales in the stress
profile. Overall, the child-related burden was greater than the parent-related
Keywords: Cleft lip; Psychological stress;
burden. These results indicate that parents of children with CLP have a higher Parenting; Quality of life; Depression.
level of psychological stress than parents of non-cleft children. This
psychological stress might be reduced if addressed by specialist clinical Accepted for publication 12 April 2022
psychologists in cleft-treating centres. Available online xxxx

Introduction
Facial clefts, especially cleft lip and pa-
late (CLP), are among the most common
congenital malformations; they are re-
lated to other malformations or syn-
dromes, with a prevalence of up to 50%.1
Several therapy regimes for CLP,
including surgical procedures, have been
described and discussed extensively in
the literature2–5. To achieve optimal
aesthetic and functional results, an in-
terdisciplinary, multi-professional treat-
ment team is of the utmost importance,
during both the primary cleft surgery
period and long-term aftercare of
1
Kira Lentge and Fritjof Lentge con-
tributed equally.

0901-5027/xx0001 + 07 © 2022 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Inc. All rights reserved.

Please cite this article as: K. Lentge, F. Lentge, A.-N. Zeller et al., Cleft lip and palate: the psychological burden of affected
parents during the first three years of their children’s lives, Int. J. Oral Maxillofac. Surg, https://doi.org/10.1016/
j.ijom.2022.04.003i
YIJOM-4898; No of Pages 7
2 Lentge et al.
children with CLP.6 The initial parent–-
child relationship is characterized by
numerous operations and feelings of
helplessness frequently reported by the
infants’ parents. In the long term, lan-
guage acquisition and stigmatization,
which may include bullying, will add
more complicated stress factors. How-
ever, the early period from birth until the
age of 3 years is especially important for
parent–child bonding.7 Development of
the child’s attachment system is a critical
phase that interferes with the operative
CLP closures.7 Thus, psychosocial and
psychological factors, such as mental
stressors experienced by the children and
their families, especially parents, should
be considered carefully both at con-
sultations and during the therapy pro-
cess. However, previous studies have
focused almost exclusively on the child’s
psychological stress. To date, few studies
have evaluated the psychological burden
on parents associated with CLP. Studies
on the psychological aspects of CLP
tend to focus on psychosocial stressors
like the individual’s cultural background
and general circumstances.8
Parents of children with CLP have an
increased need for information on the
child’s condition. In addition, they need
guidance and support regarding the in-
fant’s daily care, as demonstrated in
general by Sischo et al.9 and Chua-
charoen et al.10 However, neither of these
previous studies evaluated the resulting
psychological burden on the parents as-
sociated with these increased needs.
The aim of this study was to in-
vestigate the psychological burden ex-
perienced by parents of children with
CLP by evaluating the psychological
and psychosocial factors of parents
with at least one child with CLP.
Materials and methods
Parents of children with any type of
CLP presenting to the interdisciplinary
CLP consultation hour of the
Department of Oral and Maxillofacial
Surgery, Hannover Medical School,
Hannover, Germany, within the first 3
years of the child’s life were eligible for
inclusion. Participation was voluntary,
and all potential participants were in-
formed that declining to participate
would have no bearing on their future
consultations or therapeutic options.
All of the participants were informed
about cleft deformity and the required
surgical procedures before they com- Statistical analyses
pleted the questionnaire. Written in-
The demographic data of the current
formed consent was obtained from all
study group were tested against those
participants prior to their participation.
of the two control groups investigated
The study was approved by the Ethics
by Tröster11 using the two-sample t-test
Committee of Hannover Medical
for independent samples and the χ2 test.
School, Germany (approval number
The questionnaire data were evaluated
8967_BO_K_2020).
using the standardized and calibrated
A face-to-face interview was con-
evaluation concept of the PSI.11 The
ducted after routine CLP consultation
subsections pertaining to parents and
hours in the Department of Oral and
children were evaluated separately. By
Maxillofacial Surgery of the Hanover
assigning raw and stanine values to the
Medical School. The interviews were
responses, scores were determined for
conducted by a psychosocial specialist,
all respondents on each subscale within
independent of and after the medical
different domains of the PSI.
consultation. Due to COVID-19 pan-
Tröster11 not only adapted the EBI for
demic-related contact restrictions
the German population, but also de-
during the study, only one parent per
termined the standard values that are re-
child was allowed to enter the hospital.
presentative of the population. For this
Therefore, no couples were interviewed.
purpose, two studies were conducted with
The interviews were conducted in-
parents of non-cleft toddlers and children
dependent of any surgical interven-
up to the age of 6 years. For a more ac-
tions. In accordance with the study
curate evaluation of the EBI with regard
research question, the children had to
to the normal population, Tröster11 con-
be under the age of 4 years.
verted the values obtained into t-values
Accordingly, the parents of CLP chil-
(parent and child domain and total score)
dren who had already undergone sur-
or stanine values (‘standard of nine’) for
gery and of the CLP children who had
the subscales. Tröster11 also demonstrated
not yet had surgery were interviewed.
that the age of the children and parents
A modified German version of the
did not influence the test results. Further-
Parenting Stress Index (PSI) by Abidin
more, it has been shown that the test
was used for the interview (“Eltern-
works equally well for mothers and fa-
Belastungs-Inventar” (EBI)).11 The
thers. According to Tröster,11 the number
questionnaire contains 48 specific items
of children the parent has is also irrele-
on aspects of parental stress and factors
vant, since the questionnaire must always
related to the child’s upbringing, care,
be answered in relation to the index child.
and support, with responses on a five-
Furthermore, the EBI is robust against
point Likert scale ranging from 1 (not
circumstances such as being a single
applicable) to 5 (fully applicable). The
parent, since there is a special evaluation
items are grouped by four, resulting in
sheet for single parents.12–14 Therefore,
five subscales forming a child-related
valid values exist and no comparison
domain and seven subscales forming a
group had to be examined in this study.
parent-related domain. The child-related
The norm stanine value for each subscale
domain concerns the behaviour and
was 5 (SD = 2), for each domain was 50
feelings of the child, and the demands on
(SD = 10), and for the total score was 50
the parents. The parenting-related do-
(SD = 10). Values were not totalled; each
main concerns restrictions on parental
section had to be evaluated separately.
functions, such as bringing up and caring All further statistical analyses were
for the child. Table 1 presents the char- performed using IBM SPSS Statistics
acteristics and subscales of the ques- version 26.0 (IBM Corp., Armonk,
tionnaire. Furthermore, it has been NY, USA).
shown that the test is robust against The participants’ mean scores (test
variations in the socio-demographic group) for the total score and each do-
characteristics of the parents such as main (parent and child) were compared
gender, age, and level of education.12–14 with the PSI norm values using a two-
The study design was critically re- sample t-test. The 12 individual PSI
viewed with regard to its structure, subscales were also compared to the
taking into consideration the current norm values using a two-sample t-test.
recommendations pertaining to con- Statistical significance was set at
ducting questionnaire studies, in parti- P < 0.05, based on a 95% confidence
cular the CROSS checklist.15,16 interval. For the tested scale, domains,
YIJOM-4898; No of Pages 7

Psychological burden of CLP on parents 3

Table 1. Distribution of the modified Parenting Stress Index (PSI) test subscales, showing the test subscales within the child-related and
the parent-related domains and the content of the subscales.
Child domain
Test subscales Content
Distractibility/hyperactivity Activity and distractibility of the child
Mood Moodiness, easy excitability, and dissatisfaction of the child
Acceptability Disappointment of the parents that the child does not meet their expectations and demands
Demandingness Increased demands in the upbringing and care of the child
Adaptability Difficulties of the child in aligning their behaviour with the demands of everyday life
Parent domain
Test subscales Content
Attachment to child Insecurity about empathizing with the child and assessing their needs, caused by a distant bond with
the child
Social isolation Missing contacts outside of the family and difficult maintenance of social contacts
Competence Worries about not being able to cope with the demands on the upbringing and care of the child
Depression Depressed mood, feelings of guilt and self-doubt about fulfilling the role of mother or father
Health Physical ailments, physical and mental exhaustion
Role restriction Restrictions in personal lifestyle and putting aside personal needs in favour of family responsibilities
and caring for the child
Spouse Impairment of the relationship with the partner due to the requirements in the context of caring for
the child

Table 2. Demographic data of the inter- and subscales showing significant devia- The average number of children the
viewed parents (N = 33). tions from the reference group, the effect parents had was 1.64. Table 2 reports
Average age (years) 34.7 size was then calculated using Cohen’s d, the demographic data. In terms of de-
Sex with values greater than 0.2 indicating a mographic data, the composition of the
Female 26 (78.8%) small effect, values greater than 0.5 in- control groups tested by Tröster11 did
Male 7 (21.2%) dicating a moderate effect, and values not differ significantly from that of the
Educational level greater than 0.8 indicating a large effect. current study group (Table 3).
High school graduation 17 (51.5%) The mean total questionnaire score,
Secondary school 10 (30.3%) assessing total exposure, was
graduation
Secondary school 6 (18.2%)
58.27 ± 9.96 points. The mean PSI
Results
diploma scores for the parent domain were, on
Completed vocational A total of 33 parents with an average average, about 2 points lower, with an
training age of 34.7 years were included in the overall mean for this domain of
Yes 31 (93.9%) study; 26 were female (78.8%) and 56.15 ± 9.84 points. The mean score for
No 2 (6.1%) seven were male (21.2%). There were no the child domain was 58.97 ± 8.13
Employment status single parents in the sample, and all but points. Both the parent and child domain
Working 19 (57.6%) one participant stated that they were in mean scores were significantly higher
On parental leave 12 (36.4%) a stable partnership at the time of the than norm values, with the child domain
Unemployed 2 (6.1%)
survey. Regarding the participants’ showing higher significance (P < 0.001)
Single parent
Yes 1 (3.0%) education level, 51.5% were high school than the parent domain (P = 0.001). The
No 32 (97.0%) graduates, 30.3% were secondary resulting significance for the total scale
Average number of 1.64 school graduates, and 18.2% had a was similar to that of the child domain.
children secondary school leaving certificate. These results are presented in Table 4.

Table 3. Comparison of the demographic data between the current study and the studies performed by Tröster.11
Study a Study b Study a+b Current study P-valuea
Number of parents 245 293 538 33 –
Age (years), mean ± SD 34.7 ± 5.1 35.1 ± 6.8 34.9 ± 6.1 34.7 ± 5.5 0.854b
Number of children, mean ± SD 2.00 ± 0.83 1.59 ± 0.70 1.78 ± 0.58 1.64 ± 0.60 0.183b
Employed 138 (56.3%) 244 (83.3%) 382c (71.0%) 19 (57.6%)
Unemployed 105 (42.9%) 44 (15.0%) 149c (27.7%) 14 (42.4%) 0.077d
High school graduation 92 (37.6%) 194 (66.2%) 286c (53.2%) 17 (51.5%)
Secondary school graduation 87 (35.5%) 68 (23.2%) 155c (28.8%) 10 (30.3%) 0.972d
Secondary school diploma or less 64 (26.1%) 28 (9.6%) 92c (17.1%) 6 (18.2%)
SD, standard deviation.
a
Study a+b compared with the current study.
b
Two-sample t-test for independent samples was applied.
c
Missing values to 100% due to the participants not providing the information in the questionnaire.
d 2
χ test was applied.
YIJOM-4898; No of Pages 7

4 Lentge et al.

Table 4. Statistical evaluation of the Parenting Stress Index (PSI) with respect to the parent domain, child domain, and entire PSI.
PSI, mean ± SDa Difference from the control group PSIa P-value Effect size (Cohen’s d)
Parent domain 56.15 ± 9.84 + 6.15 0.001 * 0.620 Moderate effect
Child domain 58.97 ± 8.13 + 8.97 < 0.001 * 0.984 Large effect
Total PSI 58.27 ± 9.96 + 8.27 < 0.001 * 0.829 Large effect
SD, standard deviation. *Statistically significant.
a
Built by Tröster11 using the t-transformation (mean = 50, SD = 10).

The strongest effect size was found
for the child domain (d = 0.984). The
effect size for the parent domain (d =
0.620) and the total scale (d = 0.829)
also suggested a considerable effect.
The average stress profiles from the
subscale mean scores were compared
and it was found that demandingness
had the highest score (6.91 ± 1.70),
followed by depression (6.76 ± 2.03),
parenting skills (6.15 ± 1.87), and
adaptability (6.15 ± 1.66). Detailed
comparisons with the norm values and
effect sizes are shown in Table 5. All
subscales except distractibility/hyper-
activity (P = 0.073), mood (P = 0.053),
and spouse (P = 0.071) showed sig-
nificantly higher values, representing a
greater burden. The highest significance
was found for acceptability (P = 0.005),
demandingness (P < 0.001), adapt-
ability (P = 0.001), competence
(P = 0.001), and depression
(P < 0.001).
With the exception of the subscales
mood, spouse, and distractibility/hy-
peractivity, which did not show sig-
nificant results, the effect sizes (Cohen’s
d) of all other subscales were evaluated.
Role restriction (d = 0.378) and health
(d = 0.378) had the lowest effect sizes,
corresponding to a minor effect. The
largest effect sizes were for depression
(d = 0.873) and demandingness (d
= 1.029).
Discussion demands on the parents. The elevated
values can be explained by the parti-
From their first day of life, the needs of
cular demands on the affected parents,
children with CLP differ from those of
such as difficulty feeding the child, in-
non-cleft children. They need increased
creased care effort, and recurrent hos-
support with food intake (especially
pital stays, which have been described
breastfeeding) and handling a drinking
in detail in the literature.9,18,19 The
plate, and have a higher frequency of
elevated stress values within the parent
medical consultations and hospital
domain are also understandable, since
stays. This can expose parents to sub-
parents are exposed to social pressure
stantial personal, family, and social
and personal and family expenses, and
stress.17–21 The presence of such stres-
might suffer from certain fears and
sors alone or in combination can lead
uncertainties regarding the special and
to increased psychological stress. The
sometimes challenging situation of
current study findings showed a sig-
caring for a child with CLP.22
nificantly higher level of everyday psy-
The demandingness (child domain)
chological stress compared to the norm.
and depression (parent domain) sub-
Both the parent and child domains of
scales were two of the three most ele-
the PSI showed a significantly increased
vated subscales. The demandingness
psychological sub-load, with all sub-
subscale measures parental stress di-
scale scores except ‘mood’ being sig-
rectly related to their child, and the
nificantly higher than in the control
increased demandingness scores con-
groups. Since the subscales mood and
firm that children with CLP generally
distractibility/hyperactivity represent
place greater or more intensive de-
possible increased activity, excitability,
mands on their parents, such as the
moodiness, and dissatisfaction in the
special aspects of differing drinking and
child (Table 2), the lower scores are not
feeding behavior.23
surprising, because the affected child
The depression subscale registers
does not perceive himself or herself to
parent depressive episodes. Two un-
be sick, and a CLP does not represent
derlying factors may be responsible for
any neurological pathology.
depression. First, their child’s persistent
The highest stress scores were found
condition and the accompanying stress
in the PSI child domain. Three of the
might lead to a psychological disorder,
five highest subscale scores were found
for example in the form of depression.
within this domain, which measures the
Second, the depression subscale is clo-
child’s behaviour, feelings, and
sely linked to the child attachment and

Table 5. Statistical evaluation of the PSI with respect to the subscales.

Subscale Mean ± SD Difference from the control group PSIa P-value Effect size (Cohen’s d)
Child-related subscales
Distractibility/hyperactivity 5.64 ± 1.75 + 0.64 0.073 NE
Mood 5.70 ± 2.22 + 0.70 0.053 NE
Acceptability 6.00 ± 1.15 + 1.00 0.005 * 0.613 Moderate effect
Demandingness 6.91 ± 1.70 + 1.91 < 0.001 * 1.029 Large effect
Adaptability 6.15 ± 1.66 + 1.15 0.001 * 0.626 Moderate effect
Parent-related subscales
Attachment to child 5.85 ± 1.35 + 0.85 0.016 * 0.498 Small effect
Social isolation 5.91 ± 2.08 + 0.91 0.012 * 0.446 Small effect
Competence 6.15 ± 1.87 + 1.15 0.001 * 0.594 Moderate effect
Depression 6.76 ± 2.03 + 1.76 < 0.001 * 0.873 Large effect
Health 5.73 ± 1.86 + 0.73 0.042 * 0.378 Small effect
Role restriction 5.88 ± 2.22 + 0.88 0.015 * 0.378 Small effect
Spouse 5.64 ± 1.39 + 0.64 0.071 * NE
SD, standard deviation; NE, not evaluated. *Statistically significant.
a
Built by Tröster11 using the t-transformation (mean = 5, SD = 2).
YIJOM-4898; No of Pages 7
competence subscales. Therefore, in-
creased stress reflected in these sub-
scales might increase the parents’
psychological stress, leading to mental
illness. Furthermore, difficulty in
breastfeeding or frequent separations
may diminish emotional bonding with
the child, leading to mothers dis-
proportionately suffering due to their
child’s illness.24,25
The study findings are consistent
with the current literature, as depres-
sion or depressed mood in parents of
children with CLP is a common phe-
nomenon.21,26–29 Kumar et al.28 re-
ported the highest incidence of
depressive episodes, with 42% of par-
ents of children with CLP over 10 years
of age showing a strongly or very
strongly elevated depressive disorders
screening index.
Current scientific knowledge does not
provide a clear statement about the
psychological burden of parents caring
for children with CLP. Recent studies
have reported inconsistent findings, with
some demonstrating increased parent
burden or decreased quality of life
(QoL),28,30–34 and others finding no evi-
dence of significantly increased parental
psychological burden.8,34–36 A possible
explanation for this inconsistency is that
these studies have rarely directly ad-
dressed psychological (everyday) stress.
Medical rather than psychological test
procedures are often used to characterize
potential individual stress factors.9 It
appears that only one research group has
used Abidin’s PSI to evaluate the par-
ental CLP-related psychological
burden.26,37 Thus, the current study is
novel in providing an assessment of the
psychological burden of affected par-
ents. However, psychological everyday
life stress is a complex construct: QoL,
coping, depression, parent–child attach-
ment, and sociocultural factors represent
important variables influencing psycho-
logical daily life stress, yet they cannot
be evaluated separately.
Numerous studies on parental stress
associated with children with CLP have
included the parents of children across
a wide age range, sometimes more than
10 years, making them difficult to
compare.26,28,31,38 To overcome this
issue, Zhang et al.39 measured QoL in
several cohorts (child age < 1, 1–3, 3–6,
and > 6 years) to detect such time-lim-
ited phenomena. Although the re-
searchers did not demonstrate
significant differences in psychological
distress between the parents of CLP
children and the comparison group,
Psychological burden of CLP on parents
they reported that parents of younger
children needed more support to
achieve a good QoL.
The phase from birth of the child to
the age of 3 years is a complex and
critical phase with regard to the devel-
opment of the child’s bond.7 Most
surgical interventions for CLP closure,
essential for aesthetic and functional
rehabilitation, are performed within the
first 12–18 months of life.40 The func-
tional success of the surgical measures
becomes particularly evident around
the age of 2 years when the child begins
to develop speech and language cap-
abilities. However, with frequent hos-
pital stays and outpatient
consultations, this phase is followed by
an equally important step for the young
family, namely the transition to daily
normality. This transition to normalcy
can become more difficult for the par-
ents, due to the excessive focus on the
cleft. Additionally, it can be difficult for
the child if, for example, they have
hospitalism consequent to the nu-
merous medical interventions and stays
in hospital.41 Even if the most acute
challenges are faced by parents in the
first month of their child’s life, having a
baby with a CLP means facing pro-
blems in everyday life far longer than
the duration of the surgical treatment
itself.18–20,41
Another factor complicating the
comparability of different studies on
the psychological distress of parents
caring for children with CLP, is that the
research has been conducted in dif-
ferent regions and sociocultural set-
tings.17
The availability and utilization of
healthcare services also influence the
parents’ psychological stress.9,10,19,20,38
These resources are limited in some
regions because of a lack of structural
availability or due to healthcare
costs.9,33,38 Therefore, a supra-re-
gional comparison of studies would
be extremely limited. Differences in
cultural and religious views further
increase the difficulty of comparing
parental psychological stress across
regions. To consider regional differ-
ences, a validated test adapted to the
German population was chosen for
this study.
Certain limitations of the study need
to be noted. Its single-centre design had
a limiting effect in that it introduced
selection bias, since the effects of dif-
ferent CLP treatment protocols could
not be controlled. This also limited the
number of available participants.
5
Thus, local factors, such as active
regional support groups for affected
persons, combined with clinic-specific
aspects within the care of the affected
families might have had a strong but
unquantifiable influence on the results
presented. In addition, because of
COVID-19-related limited contact
during data collection, interviews could
only be conducted with one parent at a
time, resulting in a loss of information
in the case of co-parenting couples.
However, since this restriction was
known in advance, it can be assumed
that the parent more involved in the
child’s care attended the consultation.
The questioning of absent parents was
deliberately omitted since poor re-
sponse rates have already been de-
scribed in the literature, and selection
bias must be assumed accordingly.
This study showed that the parents
of children with CLP experience a sig-
nificant and relevant increased psycho-
logical daily burden within the first 3
years of the child’s life. Special atten-
tion should be paid to depressive
symptoms of the parents. The high
professional, emotional, and psychoso-
cial demands on young parents of CLP
children appear to be the greatest de-
terminant of psychological stress. The
results clearly show a substantial need
for improved psychological care for
these families. There is a need to ad-
dress two factors in the everyday med-
ical routine of cleft-treating centres: (1)
parents of an affected child should be
adequately informed about the condi-
tion and the course of therapy and
should be involved in the treatment
process as much as possible; and (2)
parents should be supported by a spe-
cialist clinical psychologist who pro-
vides advice on psychological problems
and works with the parents to decrease
their everyday burden.
Ethics approval and consent to
participate
The study was approved by the Ethics
Committee of Hannover Medical School
(approval number 8967_BO_K_2020).
Funding
The purchase of the questionnaire li-
censes for this study was supported by
MHH-Plus (grant number 19592104).
Acknowledgements. The authors would
like to thank all of the parents for their
YIJOM-4898; No of Pages 7
6 Lentge et al.
participation in this study and Dr Iris
Steinbach for her valuable contribution
in terms of support and encouragement.
Competing interests
None.
Patient consent
All participants provided written con-
sent to participate in the study.
References
1. Lewis CW, Jacob LS, Lehmann CU, Krol
D, Gereige R, Karp J, Fisher-Owens S,
Braun P, Jacob L, Segura A. The primary
care pediatrician and the care of children
with cleft lip and/or cleft palate.
Pediatrics 2017;139:e20170628https://doi.
org/10.1542/peds.2017-0628
2. Dao AM, Goudy SL. Cleft palate repair,
gingivoperiosteoplasty, and alveolar bone
grafting. Facial Plast Surg Clin North Am
2016;24:467–76. https://doi.org/10.1016/j.
fsc.2016.06.005
3. Woo AS. Evidence-based medicine: cleft
palate. Plast Reconstr Surg 2017;
139:191e–203e. https://doi.org/10.1097/
PRS.0000000000002854
4. Duarte GA, Ramos RB, Cardoso
MCdAF. Feeding methods for children
with cleft lip and/or palate: a systematic
review. Braz J Otorhinolaryngol 2016;
82:602–9.
5. Worley ML, Patel KG, Kilpatrick LA.
Cleft lip and palate. Clin Perinatol 2018;
45:661–78.
6. Cooper DC, Peterson EC, Grellner CG,
Naidoo SD, Skolnick GB, Pfeifauf KD,
Smyth MD, Snyder-Warwick AK, Patel
KB. Cleft and craniofacial multi-
disciplinary team clinic: a look at attrition
rates for patients with complete cleft lip
and palate and nonsyndromic single-su-
ture craniosynostosis. Cleft Palate
Craniofac J 2019;56:1287–94. https://doi.
org/10.1177/1055665619856245
7. Bretherton I, Munholland KA. Internal
working models in attachment relation-
ships: elaborating a central construct in
attachment theory. In: Cassidy J, Shaver
RP, editors. Handbook of attachment:
theory, research, and clinical applications.
Second editionNew York: Guilford Press;
2008. p. 102–27.
8. Nidey N, Moreno Uribe LM, Marazita
MM, Wehby GL. Psychosocial well-being
of parents of children with oral clefts.
Child Care Health Dev 2016;42:42–50.
https://doi.org/10.1111/cch.12276
9. Sischo L, Clouston SA, Phillips C, Broder
HL. Caregiver responses to early cleft
palate care: a mixed method approach.
Health Psychol 2016;35:474–82. https://
doi.org/10.1037/hea0000262
10. Chuacharoen R, Ritthagol W,
Hunsrisakhun J, Nilmanat K. Felt needs
of parents who have a 0- to 3-month-old
child with a cleft lip and palate. Cleft
Palate Craniofac J 2009;46:252–7. https://
doi.org/10.1597/07-099.1
11. Tröster H. EBI. Eltern-Belastungs-
Inventar. Deutsche Version des Parenting
Stress Index (PSI) von R. R. Abidin.
Göttingen: Hogrefe, 2011: 5–21.
12. Gosch A. Maternal stress among mothers
of children with Williams–Beuren syn-
drome, Down’s syndrome and mental
retardation of non-syndromal etiology in
comparison to mothers of non-disabled
children. Z Kinder Jugendpsychiatr
Psychother 2001;29:285–95.
13. Tröster H. Sources of stress in mothers of
young children with visual impairments.
J Vis Impair Blind 2001;95:623–37.
https://doi.org/10.1177/
0145482×0109501005
14. Tröster H, Aktas M. Die Bedeutung in-
dividueller und familiärer Ressourcen für
die Krankheitsbewältigung von Müttern
mit neurodermitiskranken Kindern. Z
Klin Psychol Psychother 2003;32:286–94.
15. Sharma A, Minh Duc NT, Luu Lam Thang
T, Nam NH, Ng SJ, Abbas KS, Huy NT,
Marušić A, Paul CL, Kwok J, Karbwang J,
de Waure C, Drummond FJ, Kizawa Y,
Taal E, Vermeulen J, Lee GHM, Gyedu A,
To KG, Verra ML, Jacqz-Aigrain ÉM,
Leclercq WKG, Salminen ST, Sherbourne
CD, Mintzes B, Lozano S, Tran US, Matsui
M, Karamouzian M. A consensus-based
checklist for reporting of survey studies
(CROSS). J Gen Intern Med 2021;
36:3179–87.
16. Kelley K, Clark B, Brown V, Sitzia J.
Good practice in the conduct and re-
porting of survey research. Int J Qual
Health Care 2003;15:261–6.
17. Chung KY, Sorouri K, Wang L,
Suryavanshi T, Fisher D. The impact of
social stigma for children with cleft lip
and/or palate in low-resource areas: a
systematic review. Plast Reconstr Surg
Glob Open 2019;7:e2487https://doi.org/
10.1097/GOX.0000000000002487
18. Johansson B, Ringsberg KC. Parents’ ex-
periences of having a child with cleft lip and
palate. J Adv Nurs 2004;47:165–73. https://
doi.org/10.1111/j.1365-2648.2004.03075.x
19. Lemacks J, Fowles K, Mateus A,
Thomas K. Insights from parents about
caring for a child with birth defects. Int J
Environ Res Public Health 2013;
10:3465–82. https://doi.org/10.3390/
ijerph10083465
20. Sreejith VP, Arun V, Devarajan AP,
Gopinath A, Sunil M. Psychological ef-
fect of prenatal diagnosis of cleft lip and
palate: a systematic review. Contemp Clin
Dent 2018;9:304–8. https://doi.org/10.
4103/ccd.ccd_673_17
21. Hasanzadeh N, Khoda MO, Jahanbin A,
Vatankhah M. Coping strategies and
psychological distress among mothers of
patients with nonsyndromic cleft lip and
palate and the family impact of this dis-
order. J Craniofac Surg 2014;25:441–5.
https://doi.org/10.1097/SCS.
0000000000000483
22. Scheller K, Urich J, Scheller C, Watzke S.
Psychosocial and socioeconomically as-
pects of mothers having a child with cleft
lip and/or palate (CL/P): a pilot-study
during the first year of life. J Clin Exp
Dent 2020;12:e864–9. https://doi.org/10.
4317/jced.56288
23. Kühn D, Meyer S, Ptok M. Probleme
und Behandlung des Fütterns von
Säuglingen mit Spaltfehlbildungen.
Sprache Stimme Gehör 2012;36:73–5.
24. Christians-Albrecht A, Johannsen U.
Gesichtspunkte einer Elterninitiative bei
der Betreuung von Kindern mit Lippen-
Kiefer-Gaumenspalte. Sprache Stimme
Gehör 2001;25:50–3.
25. Hampel P, von Salisch M. Editorial. Prax
Kinderpsychol Kinderpsychiatr 2017;
66:652–5.
26. Grollemund B, Dissaux C, Gavelle P,
Martínez CP, Mullaert J, Alfaiate T,
Guedeney A, CLIP team (Cleft Lip &
Palate Infant Parent). The impact of
having a baby with cleft lip and palate on
parents and on parent–baby relationship:
the first French prospective multicentre
study. BMC Pediatr 2020;20:230. https://
doi.org/10.21203/rs.2.21239/v2
27. Johns AL, Hershfield JA, Seifu NM,
Haynes KA. Postpartum depression in
mothers of infants with cleft lip and/or
palate. J Craniofac Surg 2018;29:e354–8.
https://doi.org/10.1097/SCS.
0000000000004319
28. Kumar K, Kumar S, Mehrotra D, Gupta
S, Khandpur S, Mishra RK. A psycho-
logic assessment of the parents of patients
with cleft lip and palate. J Craniofac Surg
2020;31:58–61. https://doi.org/10.1097/
SCS.0000000000005835
29. Tsuchiya S, Tsuchiya M, Momma H,
Koseki T, Igarashi K, Nagatomi R,
Arima T, Yaegashi N, Japan
Environment & Children’s Study Group.
Association of cleft lip and palate on
mother-to-infant bonding: a cross-sec-
tional study in the Japan Environment
and Children’s Study (JECS). BMC
Pediatr 2019;19:505. https://doi.org/10.
1186/s12887-019-1877-9
30. Holly LE, Fenley AR, Kritikos TK,
Merson RA, Abidin RR, Langer DA.
Evidence-base update for parenting stress
measures in clinical samples. J Clin Child
Adolesc Psychol 2019;48:685–705. https://
doi.org/10.1080/15374416.2019.1639515
YIJOM-4898; No of Pages 7
31. Aslan BI, Gülşen A, Tirank ŞB,
Findikçioğlu K, Uzuner FD, Tutar H,
Üçüncü N. Family functions and life
quality of parents of children with cleft
lip and palate. J Craniofac Surg 2018;
29:1614–8. https://doi.org/10.1097/SCS.
0000000000004611
32. De Cuyper E, Dochy F, De Leenheer E,
Van Hoecke H. The impact of cleft lip
and/or palate on parental quality of life: a
pilot study. Int J Pediatr
Otorhinolaryngol 2019;126:109598https://
doi.org/10.1016/j.ijporl.2019.109598
33. Emeka CI, Adeyemo WL, Ladeinde AL,
Butali A. A comparative study of quality
of life of families with children born with
cleft lip and/or palate before and after
surgical treatment. J Korean Assoc Oral
Maxillofac Surg 2017;43:247–55. https://
doi.org/10.5125/jkaoms.2017.43.4.247
34. Schuster M, Kummer P, Eysholdt U,
Rosanowski F. Social orientation of
parents of children with cleft lip and pa-
late. HNO 2003;51:507–12. https://doi.
org/10.1007/s00106-003-0843-9
35. Baker SR, Owens J, Stern M, Willmot D.
Coping strategies and social support in
the family impact of cleft lip and palate
Psychological burden of CLP on parents
and parents’ adjustment and psycholo-
gical distress. Cleft Palate Craniofac J
2009;46:229–36. https://doi.org/10.1597/
08-075.1
36. Berger ZE, Dalton LJ. Coping with a cleft:
psychosocial adjustment of adolescents
with a cleft lip and palate and their par-
ents. Cleft Palate Craniofac J 2009;
46:435–43. https://doi.org/10.1597/08-093.1
37. Grollemund B, Guedeney A, Vazquez
MP, Picard A, Soupre V, Pellerin P,
Simon E, Velten M, Dissaux C,
Kauffmann I, Bruant-Rodier C, Danion-
Grilliat A. Relational development in
children with cleft lip and palate: influ-
ence of the waiting period prior to the
first surgical intervention and parental
psychological perceptions of the ab-
normality. BMC Pediatr 2012;12:65.
https://doi.org/10.1186/1471-2431-12-65
38. Hlongwa P, Rispel LC. ‘People look and
ask lots of questions’: caregivers’ per-
ceptions of healthcare provision and
support for children born with cleft lip
and palate. BMC Public Health 2018;
18:506. https://doi.org/10.1186/s12889-
018-5421-x
7
39. Zhang Y, Gong C, Wu H, Chen Y,
Zhang X, Liang Y, Ha P, Shi B. Analysis
of quality of life of 115 parents with cleft
lip and/or palate children. Hua Xi Kou
Qiang Yi Xue Za Zhi 2015;33:169–73.
40. Hemprich A. Die operative Behandlung von
Lippen-Kiefer-Gaumen-Spalten—Domaine
des MKG-Chirurgen. Der MKG-Chirurg
2018;11:221.
41. Sieber M, Ullmann-Bremi A, Baenziger
O, Spirig R. A challenge in spite of ac-
ceptance—how mothers of newborn ba-
bies with a “cleft” experience the
transition from the hospital back home.
Pflege 2008;21:299–307. https://doi.org/
10.1024/1012-5302.21.5.299
Correspondence to: Department of Oral
and Maxillofacial Surgery
Hannover Medical School
Carl-Neuberg-Straße 1
30625 Hannover
Germany.
Tel:+49 (0)511 532 4879. Fax: +49 (0)
511 532 18598.
E-mail: spalthoff.simon@mh-hannover.de