Psychological and Peer Difficulties of Children with Cleft Lip and/or Palate: A Systematic Review and Meta-Analysis

Abstract

Objectives

A systematic review and meta-analysis were conducted to determine if children born with cleft lip and/or palate are at increased risk of psychological and peer difficulties, and if so, which difficulties they develop.

Methods

EMBASE, MEDLINE, and PsycINFO were searched for English language studies published between January 2005 and January 2022 which investigated the psychological outcomes and peer function of children with nonsyndromic cleft lip and palate. Outcomes included internalizing problems, such as anxiety and depression, externalizing problems, such as hyperactivity, conduct disorders, self-concept including self-image and self-esteem, peer problems, resilience, coping, and overall psychological function. A risk of bias assessment was performed using the Newcastle-Ottawa Scale. Random effects models were used in the meta-analysis to compare the outcomes for children born with a cleft and those without.

Results

In total 41 studies met inclusion criteria, with 9 included in the meta-analysis. Children born with a cleft appear to have similar psychological outcomes compared to normative controls when using the strengths and difficulties questionnaire. There are some minor differences between self-report and parent report, with parents generally reporting that their child with a cleft has increased emotional, conduct, and hyperactivity problems. The small differences between the study cohort and control cohorts are unlikely to imply any differences on a clinical level.

Conclusions

Overall psychological outcomes appear to be similar between children born with a cleft and the nonaffected population, however, some symptoms such as anxiety and depression appear higher in children with cleft lip and/or palate.

Keywords

psychological assessment psychosocial adjustment quality of life

Introduction

Oral clefts are the most common craniofacial diagnosis worldwide, with a prevalence of around 1 in every 700 live births.¹ Being born with a cleft can affect many different aspects of an individual's life, including development of speech and hearing, facial esthetics, dental development, and oral health.² It has been reported that children with a cleft lip and/or palate (CL/P) may experience more psychological and peer difficulties.^3,4 Factors that may contribute to the increased psychological and social adjustment difficulties in this population include an increased incidence of bullying, communication difficulties that may impact social interactions, lower self-esteem due to residual facial scarring following surgery, and increased medical and surgical treatments over several years.^3,5,6 Subjective concern with facial appearance has been shown to be a greater predictor of developing depression in children with a cleft than others’ perceptions of severity.^4,6 Unwanted comments or stares from other children or adults toward children with facial differences have been implicated in poorer psychological functioning.⁷ Children with additional medical diagnoses or comorbid conditions are at a significantly higher risk of developing psychological difficulties.⁶ Support from close family members can be protective against the development of psychological difficulties.⁸ Previous literature and systematic reviews in the area have not found conclusive evidence of a link between a diagnosis of CL/P and increased psychological and peer difficulties.^4,9,10 The lack of uniformity between study designs, including the diversity in psychological domains explored and outcome measures used, results in difficulty evaluating the psychological challenges experienced in this population.

Objectives

The aim of this systematic review and meta-analysis was to consolidate contemporary literature in the area of psychological function and peer relationships of children born with CL/P and update previous reviews. Research questions included:

Are children with CL/P at increased risk of developing psychological and peer difficulties?

What, if any, psychological difficulties do children with CL/P develop?

Methods

The Preferred Reporting Items for Systematic reviews and Meta-Analyses (PRISMA) statement was used to guide this systematic review.¹¹ PsycINFO (Ovid), EMBASE (Ovid), and MEDLINE (Ovid) were searched for literature published between January 2005 and January 2022 for inclusion, following on from the most recent systematic review on the topic, published in 2005.⁴ A medical librarian developed the search strategies following consultation with the research team (see Supplemental Table 1). The search was limited to studies published in English. Titles were reviewed by 1 examiner (EKB) and abstract reviews were conducted by 3 independent examiners (EKB, VCB, and NK), and any discrepancies were discussed and resolved. Texts were reviewed in full by 1 examiner (EKB), with a selection of papers reviewed by 3 examiners to confirm eligibility (EKB, NK, and LMC).

Inclusion criteria required articles to include (1) children up to 17 years of age; (2) with nonsyndromic CL/P (studies that reported syndromic or children with other medical conditions together with children with nonsyndromic CL/P were excluded); and (3) outcomes including psychological symptoms, peer/interpersonal problems, self-image, self-esteem, self-concept, hyperactivity, conduct disorders, resilience and coping, and overall psychological function. Case reports, letters to the editor, randomized control trials, qualitative studies, progress reports, dissertations, and literature reviews were not included. Studies that used a nonvalidated or unpublished measurement tool were also excluded.

Data were extracted from each eligible study and tabulated by 1 author (EKB). Data collected included study type, country of origin, study population, control population (if applicable), the psychosocial tool used, and a summary of results.

In addition, a meta-analysis was conducted on studies that used the strengths and difficulties questionnaire (SDQ) as an outcome measure. The SDQ is a commonly used brief screening tool, which provides an overview of a child's psychological functioning and prosocial behavior and has both self-report and parent report versions.¹² The SDQ has the following domains: emotional problems, conduct problems, hyperactivity, interpersonal problems, and prosocial behavior. It has been shown to have good internal reliability for all outcome domains and satisfactory external validity against clinical diagnoses.^13,14 It has been translated into 40 different languages, originally used in European countries, it is now a commonly utilized psychosocial measure with normative data available from Brazil, Canada, the Middle East, Asia, and Australia.¹⁵ Due to the homogeneity of SDQ scores and its widespread use in measuring psychological outcomes in children born both with and without a cleft, a meta-analysis was conducted on the studies that reported SDQ results. Means and standard deviations (SDs) of SDQ scores were used for the calculation of effect sizes (Hedge's g), confidence intervals, z-scores, and P values. Data were analyzed using the computer software Comprehensive Meta-Analysis, version 3.0 (available from https://www.meta-analysis.com/index.php?cart=B7K36554927). Studies were excluded from the meta-analysis if appropriate data were not provided or duplicate samples were used. The random effects model was used to determine effect sizes due to the inclusion of several different populations as the control population.¹⁶ A risk of bias analysis, The Newcastle-Ottawa Scale (NOS), was conducted for all included studies¹⁷ with the modified NOS seen in Supplemental Table 2. Studies with a higher NOS score reflected a lower risk of bias, with a maximum score of 12. The complete assessment can be seen in Supplemental Table 3.

Results

Study Selection

A total of 1732 studies were identified through database searches, with an additional 4 identified through searches of reference lists of relevant articles. A total of 1634 articles remained following the removal of duplicates, with 68 remaining following screening and 41 studies being included in the systematic review (see Figure 1). Of these 41 studies, 9 were included in the meta-analysis.

Participants from studies included in the qualitative review ranged from 2 to 17 years of age and participants from studies included in the meta-analysis ranged from 5 to 17 years. Studies reported on populations from 16 different countries, most commonly the United States of America, the United Kingdom, and Norway. Total sample sizes ranged between 20 and 1196 participants, with over half of the included studies having <100 participants. A total of 32 different tools were used to measure psychological outcomes, most commonly the SDQ, the child experiences questionnaire (CEQ), and the child behavior checklist (CBCL). A summary of the studies included can be found in Table 1, with further information on results seen in Supplemental Table 4.

Table 1.

Study Characteristics and Risk of Bias Assessment.

Author and year	Study type	Country	Population	Control population	Psychosocial tool	Risk of bias assessment (max = 12)
Aleksieva et al., 2021¹⁸	Prospective cohort	Belgium	91 children aged 10-17 years with CL/P	790 age-matched controls	Harter self-perception profile for children and adolescents	8
Berger and Dalton, 2009⁵	Cross-sectional cohort	The United Kingdom	145 households of children 11-16 years, with either CL/P	Normative data	SDQ SWA CEQ	6
Berger and Dalton 2011¹⁹	Cross-sectional cohort	The United Kingdom	145 households of children 11-16 years, with either CL/P	No control	SDQ KIDCOPE SWA CEQ	6
Bjerke et al., 2018⁶	Cross-sectional cohort	Norway	363 children aged 10 years with CL/P	Normative data	SDQ	8
Boes et al., 2007²⁰	Cross-sectional cohort	The United States	30 boys with CL, CLP, or CP aged 7-12 years	43 otherwise healthy comparison subjects matched on age and gender	Self-description questionnaire 1 Social Adjustment Scale	8
Bous et al., 2020²¹	Cross-sectional cohort	The United States	42 children aged 11-17 years with CL/P	55 otherwise healthy controls from an orthodontic treatment clinic	SDQ	5
Brand et al., 2009²²	Cross-sectional cohort	Switzerland	32 children aged 6-16 years with CLP	34 age- and gender-matched controls	SDQ Participation in early life communication questionnaire	9
Cheung et al., 2007⁸	Cross-sectional cohort	China	94 participants aged 10-40 years with CL/P, CL + A	Normative data	Social avoidance and distress scale Satisfaction with life scale Cultural free self-esteem inventory Chinese Miller behavioral style scale	4
Collett et al., 2012²³	Case-control	The United States	93 children aged 5-9 years with CL/P	124 gender and age-matched controls	CBCL Social competent scale PedsQL HRQoL	9
Crepaldi et al., 2019²⁴	Cross-sectional cohort	Brazil	57 adolescents aged 14-17 years with CL/P	No control	Short form health survey	6
Damiano et al., 2007²⁵	Cross-sectional cohort	The United States	104 mothers of children aged 3-13 years with CL/P	Normative data	PedsQL HRQoL	8
Demir et al., 2011²⁶	Cross-sectional cohort	Turkey	20 children aged 6-16 years with CLP	40 age- and gender-matched controls	Child depression inventory State-trait anxiety inventory for children Piers-Harris children's self-concept scale Children's global assessment scale Beck anxiety inventory Beck depression inventory	6
Fadeyibi et al., 2012²⁷	Cross-sectional cohort	Nigeria	116 children and adults aged 6 and over with CL/P	Normative data	General health questionnaire State-trait anxiety inventory for children Self-rating depression scale	4
Feragen et al., 2009²⁸	Cross-sectional cohort	Norway	268 children aged 10 years with CL/P, CL + A	No control	CEQ Personality inventory for children	7
Feragen et al., 2010²⁹	Retrospective audit	Norway	289 adolescents aged 16 years with CL/P	Normative data	Hopkins symptoms checklist Self-perception profile for adolescents	9
Feragen and Stock, 2014³⁰	Retrospective audit	Norway	132 children aged 10 years with CL/P	Normative data	SDQ CEQ	9
Feragen and Stock, 2016³¹	Retrospective audit	Norway	842 children aged 10-year-olds with CL/P, CL + A	Normative data	Personality inventory for children CEQ SDQ SWA	10
Feragen et al., 2015³²	Retrospective audit	Norway	857 adolescents aged 16 years with CL/P, CL + A	Normative data	CEQ SDQ SWA Self-perception profile for adolescents Hopkins symptoms checklist	10
Feragen et al., 2014³³	Retrospective audit	Norway	754 children aged 10 years with CL/P, CL + A	No control	SDQ	6
Goh et al., 2019³⁴	Cross-sectional cohort	Malaysia	74 children aged 7-17 years with CL/P	No control	SDQ	3
Gussy and Kilpatrick, 2006³⁵	Cross-sectional cohort	Australia	23 adolescents aged 12-16 years with CLP	Normative data	Self-description questionnaire II	5
Ha et al., 2013³⁶	Cross-sectional cohort	China	93 children aged 6-11 years with CL/P	65 age- and gender-matched controls	CBCL	8
Hunt et al., 2007³⁷	Cross-sectional cohort	The United Kingdom	129 children aged 8-18 years with CL/P	96 SES matched controls	CBCL	9
Kelly and Shearer, 2020³⁸	Cross-sectional cohort	The United Kingdom	130 children aged 10 and 15 years with CL/P	Normative data	SDQ	7
Leopoldo-Rodado et al., 2021³⁹	Case-controlled	Spain	171 children aged 4-7 years with CL/P	186 age-matched controls	KINDL-Kiddy HRQoL	7
Lima et al., 2015⁴⁰	Case-controlled	Brazil	61 children aged 7-17 years with CL/P	61 age, gender, and SES matched controls	Child depression inventory	7
Millar et al., 2013⁴¹	Cross-sectional cohort	The United Kingdom	94 children aged 10 years with UCLP or UCL	Normative data	Cultural free self-esteem inventory Child depression inventory State-trait anxiety inventory for children Revised Rutter parent scale	7
Murray et al., 2010⁴²	Cross-sectional cohort	The United Kingdom	103 children aged 18 months to 7 years with CL/P	100 controls	CBCL	6
Nopoulos et al., 2010⁴³	Cross-sectional cohort	The United States	50 boys aged 7-17 years with CL/P	60 age, SES, and ethnicity matched controls	Pediatric behavior scale	7
Petrackova et al., 2015⁴⁴	Cross-sectional cohort	The Czech Republic	32 children aged 2-7 years with CL	No control	Child health questionnaire	5
Potemra et al., 2020⁴⁵	Cross-sectional cohort	The United States	55 children aged 14-17 years with CLP	14 age-matched controls	Patient-reported outcomes Measures information system	8
Ruff et al., 2016⁴⁶	Prospective longitudinal	The United States	1196 children aged 7.5-18.5 years with CP or CLP	No control	Beck youth inventory Resilience scales for children and adolescents	4
Ruiz-Guillen et al., 2021⁴⁷	Cross-sectional cohort	Spain	60 children aged 8-18 years with CL/P	No control	Quality of life adolescents cleft Questionnaire	4
Ruiz-Guillen et al., 2021⁴⁸	Cross-sectional cohort	Spain	60 children aged 8-18 years with CL/P	Normative data	Cognitive emotion regulation questionnaire	4
Sagheri et al., 2009⁴⁹	Cross-sectional cohort	Germany	61 children aged 6-17.5 years with CL/P	Normative data	KINDL-Kiddy HRQoL	6
Slifer et al., 2006⁵⁰	Cross-sectional cohort	The United States	24 children aged 7-16.5 years with CL/P	25 location matched controls	Harter self-perception profile for children and adolescents	5
Snyder and Pope, 2010⁵¹	Retrospective audit	The United States	428 Children aged 2-18 years with CL/P	Normative data	CBCL	5
Soedjana et al., 2021⁵²	Cross-sectional cohort	Indonesia	104 Children with UCL, UCL + A, UCLP	No control	CBCL	3
Thompson et al., 2021⁵³	Cross-sectional cohort	New Zealand	378 Children aged 5-12 years with CL/P	Normative data	SDQ	7
Waylen et al., 2017⁵⁴	Cross-sectional cohort	The United Kingdom	268 Children aged 5-6.5 years with UCLP	Normative data	SDQ	8
Wehby et al., 2012⁵⁵	Cross-sectional cohort	The United States	104 mothers of children aged 2-12 years with CL/P	Normative data	Pediatric behavior scale CBCL	6

Abbreviations: CBCL, childhood behaviour checklist; CEQ, childhood experience questionnaire; CL, cleft lip; CL + A, cleft lip and alveolus CL/P, cleft lip and/or palate; CLP, cleft lip and palate; CP, cleft palate; PedsQL HRQoL, paediatric health-related quality of life questionnaire; SDQ, strengths and difficulties questionnaire; SES, socioeconomic status; SWA, Satisfaction with Appearance Questionnaire; UCL, unilateral cleft lip; UCL + A, unilateral cleft lip and alveolus; UCLP, unilateral cleft lip and palate.

Meta-Analysis of SDQ Results

Of the 41 articles included in the qualitative review, 13 used the SDQ as an outcome measurement tool. To be included in the meta-analysis, studies needed to present findings of the SDQ as continuous data, providing mean, SD, and sample size data for both the cleft cohort and the control cohort. Four articles were excluded as results of the SDQ were either published in other studies already included in this meta-analysis,¹⁹ the data** was presented categorically, as abnormal or normal,^{34, 53} or there was no data provided on a comparison group.³³ As such, a total of 9 studies were included in the meta-analysis, with 8 presenting results of self-reports,^{5,6,21,22,30–32,38} and 7 reporting results of parent reports.^{5,6,21,22,30,31,54}

Some of the studies subcategorized the data, depending on gender,^{6,31,32,38,54} age,^22,38 or cleft visibility.³² In this context, cleft visibility was assigned according to cleft phenotype with clefts of the lip, with or without cleft palate (CP) being defined as “visible” while clefts of the palate only being not visible. These were entered as independent data sets into the Comprehensive Meta-Analysis software. These were then used to determine the overall effect sizes using random effects estimates, as outlined in Table 2.

Table 2.

Overall Effect Sizes for SDQ Subscales Rated by Parents and Self-Reports Using Random Effects Estimates.

	Number of studies	Hedge’s g	95% CI	z-Score	P-value
Emotional symptoms
Self-rated	8	0.04	−0.06, 0.15	0.82	.41
Parent	7	0.25	0.11, 0.38	3.66	<.01
Peer problems
Self-rated	8	0.06	−0.13, 0.09	−0.33	.74
Parent	7	0.06	−0.10, 0.22	0.75	.46
Conduct problems
Self-rated	7	−0.18	−0.31, −0.04	−2.55	.01
Parent	7	0.05	0.00, 0.10	2.03	.04
Hyperactivity
Self-rated	7	0.05	−0.04, 0.14	1.12	.26
Parent	7	0.06	0.00, 0.11	2.15	.03
Prosocial behavior
Self-rated	5	0.11	−0.07, 028	1.18	.24
Parent	6	0.05	−0.11, 0.20	0.60	.55
Total difficulties
Self-rated	6	−0.04	−0.18, 0.10	−0.56	.58
Parent	6	0.14	−0.04, 0.33	1.50	.13

Abbreviations: CI, confidence interval; SDQ, strengths and difficulties questionnaire.

Self-Reports

Children born with a cleft reported similar levels of psychological and peer difficulties when compared to the nonaffected cohorts. Conduct problems were the only outcome domain that was statistically significant on self-reports, reflecting fewer reported concerns with a Hedge’s g of −0.18 (95% confidence interval [CI] −0.31 to −0.04, P = .01). For all other domains, differences between the study group and normative populations were not significant.

Parent Reports

Parents of children born with CL/P reported rates of difficulties differently from their children. Statistically significant results were seen on parent reports for emotional problems with a Hedge’s g of 0.25 (95% CI 0.11-0.38, P < .05), conduct problems 0.05 (95% CI 0.00-0.10, P = .04), and hyperactivity 0.06 (95% CI 0.00-0.11, P = .03), indicating slightly elevated levels of problems in the cleft cohort.

There did not appear to be differences between study subgroup populations (ie, gender, age, or cleft visibility) for any domains of the SDQ. The small differences between the study cohort and control cohort outlined above are unlikely to result in any differences on a clinical level.

Internalizing Problems

Internalizing problems, including depression and anxiety symptoms, were investigated in 15 of the 41 articles. Several different psychological tools were used, with the most commonly employed being the CBCL,⁵⁶ the CEQ,⁵⁷ and the Child Depression Inventory.⁵⁸ Of the 15 studies, 6 reported higher psychological symptoms among children born with a cleft,^{24,26,27,36,39,55} whereas the other 9 studies showed either less or similar levels of psychological symptoms to typically developing children.^{31,32,38,40,41,45,46,49,52} Levels of anxiety and depression in Nigerian children were investigated by Fadeyibi et al,²⁷ who reported higher levels of anxiety and depression in children with a cleft younger than 12 years of age, which they postulated was due to poorer levels of parental acceptance of children born with a craniofacial diagnosis in this culture. Demir et al²⁶ determined that poorer speech and articulation related to the oral cleft was correlated to poorer emotional outcomes and increased incidence of anxiety disorders. However, the small sample size (n = 20) hampers strong conclusions. Although Potemra et al⁴⁵ did not find a significant difference in psychological symptoms in children with cleft lip and palate (CLP) and a control cohort, the number of surgical revisions required between the ages of 8 and 10 years had a positive correlation with anxiety and depressive symptoms. It has also been suggested that children with an isolated cleft lip (CL) or CP had greater psychological difficulties than those born with CLP, however, when the confounders of age and gender were considered, there was no difference associated with cleft type.²⁴ Gender differences have also been investigated, indicating that girls with a cleft have increased psychological symptoms, whereas boys report similar levels to control cohorts.³⁶ Satisfaction with facial appearance was inversely correlated to increased levels of depression and anxiety in children with CL, CP, and CLP in a study by Wehby et al.⁵⁵ Increased incidence of psychological symptoms was noted to occur in children with visible clefts, although this was not clinically significant.⁴¹ Studies investigating psychological and behavioral difficulties in 10-year-old and 16-year-old children with clefts showed similar scores for depressive symptoms compared with the general population.^31,32

Externalizing Problems

Externalizing problems include hyperactivity and conduct problems. Hyperactivity and inattentive behaviors, including difficulties concentrating and completing tasks, among children with a cleft were investigated in 5 of the 41 articles. The SDQ, CEQ, and CBCL were the most used scales for hyperactivity. Three studies investigating the prevalence of hyperactivity in children with a cleft had reported no increased risk for these children when compared to the noncleft population.^26,31,32 Girls born with a cleft had less hyperactive behaviors, whereas boys had similar levels^31,32 or higher levels³⁶ when compared to the normative data, with no reported association with cleft type or cleft visibility.³² Two studies, however, found a correlation between the presence of a cleft and hyperactivity.^36,43 Nopoulos et al⁴³ investigated the relationship between the structure of the right ventromedial prefrontal cortex and hyperactivity, impulsivity, and inattention, concluding that boys born with a cleft had significantly elevated scores for hyperactivity, and 10% of participants having a clinical diagnosis of Attention Deficit Hyperactivity Disorder.³⁶

Conduct problems include aggressive or nonaggressive disobedient or destructive behaviors and rule-breaking.⁵⁹ Only 4 of the 41 articles focused on conduct problems in children with a cleft, most commonly utilizing the SDQ and the Personality Inventory for Children.⁶⁰ Similar levels of conduct problems were reported between girls born with a cleft and control cohorts in 3 of the studies,^31,32 however, boys with a cleft had significantly fewer conduct problems than their peers.³¹ A study of 104 children with unilateral CL/P showed similar levels of conduct problems for children born with a cleft and the typically developing population, with 98.08% of the study population being in the normal range for externalizing problems.⁵² Conversely, a study by Wehby et al⁵⁵ indicated that 12.5% of the study population scored above the 90th percentile for aggressive and oppositional behaviors, indicating an increased risk.

Peer Problems

Peer problems have been defined as psychological distress resulting from interactions with other individuals of a similar age,⁶¹ which may include social difficulties, making and keeping friends, and social skills. Of the 41 articles included in this review, 13 investigated the impact of a cleft on peer problems, primarily using the CBCL and the SDQ.¹² A total of 7 of these studies reported an increase in peer problems in children born with a cleft ^{20,22,29,32,36,37,42} and 4 studies reported no difference.^38,48–50 One study reported more positive interpersonal relationships for children born with a cleft when compared to the normative population,⁵ and 1 specifically investigated the impact of orthodontic treatment on peer problems.⁴⁷ In a study of 32 participants, Brand et al²² reported that children born with CLP had increased social difficulties, with a 6-fold increase in the incidence of peer problems when compared to the control cohort. Cleft visibility was also associated with poorer social acceptance and fewer close friendships in a study involving 16-year-old participants.²⁹ Girls born with a cleft report fewer positive social interactions than boys, and even fewer for girls with visible clefts.³² The opposite was true for boys, however, with more positive social experiences in boys with visible clefts. Older children born with a cleft have fewer peer problems than their younger counterparts.⁵ Boes et al²⁰ further determined with the use of structural magnetic resonance imaging that peer problems were associated with less brain tissue in the straight gyrus in children with nonsyndromic CL/P. Orthodontic treatment and improvement in malocclusion have been shown to have a positive impact on interpersonal problems for children born with a cleft, with moderate effect sizes.⁴⁷

Self-Concept, Self-Image, and Self-Esteem

Self-concept, including self-image and self-esteem, broadly refers to the focus on internal thoughts about oneself, including satisfaction with appearance. These ideas were investigated in 12 of the 41 studies reviewed, with the most commonly used tool being the Satisfaction with Appearance Questionnaire (developed by the Cleft Psychology Special Interest Group, 2007). Of these studies, 6 reported lower levels of self-esteem or satisfaction with appearance^{8,31,32,37,39,5} in children born with a cleft, 5 reported higher levels than control cohorts^{5,18,41,47,49} and 1 reported similar levels among study groups.³⁵ Ruiz-Guillen et al⁴⁷ investigated the impact of orthodontic treatment, revealing that those who had undergone treatment had improved self-concept. Cleft visibility had a significant impact on satisfaction with facial appearance, with poorer self-concept in children with a visible cleft,^5,31,32,41 and results indicating that children with lower satisfaction with facial appearance have increased levels of anxiety and general unhappiness.³⁷ Age also plays a role in this population, with Cheung et al⁸ reporting that while children living with a cleft have poorer self-esteem, this improves as children age to reach levels similar to that reported in the normative adult population. The effect of poor satisfaction with a facial appearance on behavioral problems was investigated by Wehby et al,⁵⁵ with results showing that poorer satisfaction with appearance is directly correlated to increased behavioral problems, except for aggressive behaviors. A study from Australia reported similar levels between children with and without a cleft when investigating both global and total self-concept,³⁵ and improved self-concept when examining opposite sex and parental relations. Timing of cleft repair and associations with self-esteem was investigated, and results showed that those children who received surgery earlier had improved levels of self-esteem.⁴⁴ There was no reported difference between early and late surgical interventions when investigating other forms of self-concept, including social-emotional and self-concept. In half of the studies, self-image and self-esteem appear to be affected by the presence of CL/P, with poor satisfaction with appearance being the most common factor.

Resilience and Coping

Resilience has been defined as “the capacity for positive adaptation in significant adversity.”⁶² Of the 41 articles included, 7 investigated resilience in children born with a cleft,^{5,8,23,28,29,46,48} measured by the KIDCOPE Checklist⁶³ and CEQ. Generally, children and adolescents born with a cleft were found to have good resilience and psychological adaptation,^23,28,29,48 despite the increased incidence of teasing and social stigmatization.²³ Ruiz-Guillen et al⁴⁸ determined that children born with a cleft had more positive coping skills than the general population, with less rumination, catastrophizing, and self-blame, and increased levels of positive focusing. Feragen et al²⁸ suggested that some negative experiences were required to develop the necessary coping strategies to develop resilience. Family support systems, social support, and social avoidance were reported as frequent coping strategies among children with clefts,⁵ and positive friendships and social experiences result in improved resilience among 16-year-olds.²⁹ Coping techniques also differ between age groups, with adolescents reportedly coping better than their adult cleft counterparts.⁸ Frequent incidence of teasing, questions, and staring related to the cleft may lower resilience with a correlation to higher levels of depression and anxiety.²⁸ Resilience and its relationship to cleft surgeries have also been investigated which indicates that resiliency is negatively associated with surgical intervention and the number of surgical revisions required.⁴⁶

Overall Psychological Functioning

Psychological functioning has been defined as a person's ability to achieve goals both personally and within the external environment, including behaviorally, emotionally, socially, and overall mental health.⁶⁴ Of the 41 studies, 15 reported on the overall psychological functioning of children born with a cleft, most commonly utilizing the SDQ, the CBCL, and the CEQ. Of these studies, 7 did not find any difference in overall psychological functioning between the cleft population and the control cohort,^{5,21–23,30,51,53} 5 studies found poorer psychological functioning,^{6,25,33,34,54} and 2 studies found improved psychological functioning in children born with a cleft.^31,32 One study investigating overall psychological functioning did not report on similarities to normative data,¹⁹ however, Berger and Dalton¹⁹ noted that social experiences are the most important predictor for psychosocial functioning, with other less important factors including satisfaction with appearance and speech. Cleft visibility and the impact on psychological functioning were investigated by Feragen and Stock,³⁰ who reported that there were no significant differences between children with a visible cleft and those with a nonvisible cleft. Differences between specific cleft types, including CL, CLP, and CP were also investigated with differing results. Snyder and Pope⁵¹ found that children with CLP had poorer psychological functioning than those with CL or CP, whereas children with CP and CL scored higher than those with CLP in a recent study from New Zealand.⁵³ Boys with a cleft were also at higher risk of developing psychological difficulties when compared to girls.^23,54

Risk of Bias

There was a wide range of scores for the risk of bias analysis, ranging from 3 to 10 out of a possible 12. Most commonly, studies scored poorly in the comparability domain due to not including a control cohort. Several studies also scored poorly on sample selection, with no indication of the cleft cohort being examined or ascertainment of exposure. For studies included in the meta-analysis, generally, the risk of bias was low with scores ranging between 5 and 10. This may be explained by the meta-analysis only including studies that had a reported control cohort.

Discussion

This review of psychological and peer difficulties for children born with a cleft examines 41 studies from 16 different countries. Overall, the results of this review indicated that there is conflicting evidence when determining if children born with a cleft are at increased risk of developing psychological and peer difficulties. Generally, children born with a cleft appear to fare similarly in outcome domains compared to their nonaffected counterparts. Inconsistent evidence for increased psychological symptoms in children with a cleft exists in the literature, with more frequent incidences of teasing, poor speech articulation, and cleft visibility appearing to be associated with increased levels of anxiety and depressive symptoms,^26,55 and occurring more frequently among the female cleft population.^31,32 The increased psychological symptoms in girls are however consistent with findings of normative populations and not unique to this study population.⁶⁵ Communication challenges and an increased incidence of teasing among children born with a cleft may explain the higher rates of peer problems reported in some studies.^36,37,42 Poorer self-concept, self-image, and self-esteem were associated in half of the studies with being born with CL/P, with low satisfaction with facial appearance being the most commonly associated with poorer outcomes.^{8,31,32,35,37,55} Improvements in facial appearance through surgical intervention or orthodontic treatment have been shown to improve self-esteem and peer relationships.^44,47 This suggests that access to revision surgeries and orthodontic treatment for malocclusion associated with oral clefting remains an important component of the care pathway. Hyperactivity problems are an underrepresented psychological domain in the literature involving children born with a cleft. This, along with the conflicting results in a few studies included in this review, makes it hard to draw conclusions regarding hyperactivity in children with a cleft. Further research investigating the link between structural cortex changes in children born with a cleft and the incidence of hyperactivity problems may be of value.³⁶ Similarly the limited number of studies investigating conduct disorders also results in difficulty providing comments on the incidence of these conditions in this population, however, results from the meta-analysis indicate lower levels of conduct problems on self-reports. Children born with clefts show good resilience despite social challenges. Resiliency has previously been demonstrated to have a protective mechanism against psychological difficulties⁶⁶ and appears to have a protective function against the challenges faced by children born with a cleft. This likely contributes to the minimal difference between this group and the normative population for overall psychological functioning.²³ Improved access to preventive and interventional resilience and coping resources has also been shown to improve outcomes in children⁶⁷ and should therefore be part of the overall cleft care pathway.

Findings from the meta-analysis indicated that results of the self-report and parent report of the SDQ vary slightly. This is a common finding when using this tool, with previous studies concluding that self-report scores are more accurate when assessing psychological difficulties in adolescents.⁶⁸ For SDQ domains where it would appear children with a cleft fare poorer than the unaffected population (eg, on parental reports of psychological problems), it is difficult to determine the magnitude of this difference. Results for each domain still fall within the normal range, and therefore do not indicate a clinically significant impairment. Further research is therefore indicated to investigate if there is a clinical difference between groups of children born with a cleft and those without, and if there is, what are the implications for clinical care.

Study Limitations

While this study has attempted to be as comprehensive as possible, the literature in this area is continually updating and keyword selection and exclusion criteria are constantly changing such that it is possible that some articles have not been identified in this review. Not all psychological domains are covered by the SDQ, and therefore could not be included in the meta-analysis, namely self-image, self-esteem, self-concept, and resilience and coping. The majority of included studies used broad questionnaires which investigate multiple psychological domains, which may not provide clear insight into the specific challenges seen in this population. Most studies included in this review are also cross-sectional cohorts in design. It is therefore not possible to determine if there are changes that occur over time and if psychological functioning improves or worsens over time. Studies are also from a wide range of countries, which may impact the interpretation and generalizability of results. Differences in standardized treatment protocols, including the timing of primary surgeries or consideration of subsequent surgeries, may impact study results. Cultural differences between countries may also result in conflicting findings. Few psychological tools used in the review are either country-specific (ie, the Chinese Miller behavioral style scale) or specifically designed to accommodate for cultural differences (ie, the Cultural Free Self-esteem Inventory). However, the majority of studies included are from countries where the commonly used tools, including the SDQ and the CBCL, are validated for use.^15,69

Implications for Future Research and Clinical Practice

This meta-analysis has further highlighted the need for improved consistency in measuring psychosocial outcomes to enable comparison between studies. A recent systematic review and meta-analysis investigating the impact of orofacial clefting on health and oral health-related quality of life similarly concluded the need for more consistent screening tools as this increased understanding would better support changes to clinical care that address areas contributing to the quality of life.⁷⁰ While consensus in the choice of psychological tools used to examine this population would enable greater consistency and analysis, achieving greater uniformity in measures used can be challenging. The SDQ is commonly used to provide a general overview of a child's psychological functioning and is therefore recommended as an initial screening tool in this population. Results from the SDQ are homogeneous and allow for easy comparison between groups, as evidenced in this meta-analysis. It is also frequently used in clinical practice to provide feedback on overall functioning, while also being able to track clinically significant changes over time.⁷¹ Its translation to 40 different languages also aids in its accessibility. Use of Harter's Self-Perception Profile would provide further information regarding the underlying influences on children's self-perception, in particular if physical appearance is a major contributing factor. Although not frequently used in included studies or the cleft population, it does provide a broad overview of self-worth and an understanding of this particular domain could inform targeted clinical care for these children, possibly including more focus on the child's physical appearance and how this may affect their overall psychological functioning. Tools used to measure coping and resilience, including the KIDCOPE,⁶³ may be beneficial to monitor the impact of interventions targeted to improve resilience, and subsequently, improve overall psychological functioning. The CLEFT-Q is a specific questionnaire developed for use in people born with a cleft which measures satisfaction with appearance, health-related quality of life (including psychological and social functioning), and facial function.⁷² It has been shown to have adequate reliability and validity for use within this population.⁷² The CLEFT-Q has been selected to be the most appropriate outcome measure to monitor psychosocial development in this population and is included in the International Consortium for Health Outcomes (ICHOM).⁷³ No single questionnaire adequately investigates all outcomes of interest within this population and therefore multiple questionnaires may need to be implemented to provide an overall understanding of the child's psychological functioning and experiences of peer problems. Studies with larger sample sizes and control groups pooled from similar demographics would help address the methodological problems identified in some of the studies included in this review, and further reduce the risk of bias.

Conclusion

A large amount of work has been and continues to be undertaken in the field of psychological functioning for children born with a cleft. The heterogeneity of reports, including the exploration of a wide range of psychosocial domains and the use of a large number of different outcome measures, results in difficulty determining the true impact being born with a cleft has on overall psychological outcomes. While overall psychological functioning of children with cleft seems to be similar to their typically developing peers, in certain areas children with cleft had a higher incidence of psychological difficulties. Future studies in the area with the use of similar psychological tools may provide a more accurate description of the difficulties faced by this population.

Supplemental Material

sj-docx-1-cpc-10.1177_10556656221125377 - Supplemental material for Psychological and Peer Difficulties of Children with Cleft Lip and/or Palate: A Systematic Review and Meta-Analysis

Supplemental material, sj-docx-1-cpc-10.1177_10556656221125377 for Psychological and Peer Difficulties of Children with Cleft Lip and/or Palate: A Systematic Review and Meta-Analysis by Emma K. Branson and Victoria M. Branson, Roisin McGrath, Vanessa C. Rausa, Nicky Kilpatrick, Louise M. Crowe in The Cleft Palate Craniofacial Journal

Footnotes

Declaration of Conflicting Interests

The authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Funding

The authors received no financial support for the research, authorship, and/or publication of this article

Supplemental material

Supplemental material for this article is available online.

ORCID iD

Emma K. Branson

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