Abstract
Objective
Psychosocial adjustment can be challenging for children with cleft lip with or without a cleft palate (CL ± P). Previous studies have linked social integration with self-acceptance and highlighted the impact of the parents’ stress on self-acceptance. Teasing can be linked to children having difficulties explaining their diagnosis to others. This prospective research aimed to gain a better understanding of family communication about cleft, children's oral storytelling about their cleft, and explore their self-image and perceived familial acceptance.
Participants
Parents and their children with CL ± P (N = 54; average age: 5.6 years).
Design
Semistructured interviews with parents and structured interviews with children were conducted regarding their oral storytelling focusing on scars from cleft lip surgery. Children completed a projective test to explore their self-image and perceptions of familial acceptance. Language screening was completed with the French Wechsler Preschool Primary Scale of Intelligence-Fourth Edition.
Results
Only 30% of children explained their cleft in a way appropriate for peers, including presence at birth, having had surgery, and scar location. Children's ability to explain their cleft was not related to language performance, which was in the average range of 84%. Children's cleft explanations were dependent on parents’ narratives and education methods, including the use of verbal explanations and preoperative photographs. Children's storytelling was related to their self-image and perceived parental acceptance.
Conclusion
Cleft teams should assist families in building their cleft story based on a complete explanation with photographs and a positive and accepting approach.
Introduction
Parents of children with cleft lip with or without palate (CL ± P) go through a personal journey involving at first a potential traumatic shock with guilt and grief, 1 followed by progressive acceptance, 2 built on their own adjustment to the diagnosis and leading to the development of early parental relationships. 3 Recent publications reported a significant level of postpartum depression for French mothers of children with CL ± P, 4 as well as the need for psychological support at the time of diagnosis 5 and during the first year after birth. 4 Later, parents often report that their children experience comments or bullying in social situations, with potentially severe consequences if they are left without psychological support.6,7
A positive self-concept has been suggested to result from a positive construction based on parental support and child's parental image.8–10 For children with CL ± P, some authors consider self-concept as an important variable that influences the way children feel about themselves and cope in relationships with others. 11 In fact, the way individuals view themselves influences their responses to others. 12 Self-acceptance results from a positive self-concept and is considered a key component of mental health. 13 Further, research on different dimensions of resilience identified self-acceptance as a pivotal component relating to and protecting mental health throughout the life span. 11 In contrast, it has been shown that peer harassment was correlated with dissatisfaction with appearance. 6 Teasing can eventually lead to depression in adolescence, particularly when taking place after 10 years of age in girls. 7 Poor social and appearance adjustment often demonstrate similar responses, such as depression, shame, anxiety, and associated social avoidance. 14 The perception of the severity of facial differences by others does not seem to be consistently correlated with psychosocial difficulties; rather children's self-image seems to be more strongly related to psychosocial adjustment. 15 Assessing self-image and self-acceptance can assist in implementing appropriate therapeutic strategies at an early age to support socialization and coping with others’ possible reactions to their diagnosis. Parental reports reflect that 39% of children at the age of 5 had already faced comments or teasing about their cleft, 16 this study was carried out around this critical age.
Support for uncomfortable or negative social interactions is important and children need to be prepared for others looking at them in order to respond to questions, be able to explain their difference, avoid repeated questioning, embarrassing stares, and teasing from primary school until late adolescence. Helping children to deal with possible physical differences in their appearance is a key issue in the management of CL ± P. 17 Studies have shown that the attitudes of children without a cleft can be positively influenced by the combination of explanations about the diagnosis (educational information) and a personal contact with a child with a CLP. 18 This ability to be able to explain their difference seems to be crucial for positive social interactions 12 in general situations and especially at school. This social skill, which can be developed even with young children, needs to be scaffolded by an adult, starting with children's parents.10,18
Suggestions to parents from cleft teams and in materials provided by specialized cleft associations, such as American Cleft Palate-Craniofacial Association (ACPA), Cleft Lip and Palate Association (CLAPA), or other associations specialized in visible difference (eg, Changing Faces)19, 20 include to communicate openly with their children about their diagnosis, to take and share photos at birth, and explain why they are going to the hospital frequently. However, little is known about how many parents implement these suggestions to handle this sensitive task. Similarly, more research is needed to understand how children explain their diagnosis in an understandable way to peers and others (ie, what words they use and what is the most difficult for them to understand and explain). Clear diagnosis explanations are key as studies demonstrate that when children do not understand an explanation, they will generally ask the same question again and again. 10 In their study with 10 north European cleft teams, Feragen et al 16 suggested that 93% of parents provided specific information in response to their children's questions, but less is known about parental communication with children who do not ask their parents questions. It is informative for cleft teams in providing support to families to know more precisely how parents cope with explaining a cleft diagnosis to their children as well as how children cope with telling the story of their cleft and answering questions about their diagnosis.
The hypothesis of this study is that children's ability to tell their story about their cleft to others is linked to their self-image and to their parents’ experience in teaching them about their diagnosis. This study aims to better understand how parents and children talk about CL ± P and how their storytelling relates to children's self-acceptance so that efficient tools to help children with CL ± P and their parents can be developed.
Methods
This was a prospective study of 54 consecutive children with CL ± P and their parents in our department. All of them were treated according to the same protocol: (1) primary lip surgery and intravelar veloplasty from 3 to 6 months of age, (2) closure of the hard palate at 18 months of age, and (3) alveolar cleft bone graft using iliac cancellous bone at 4-5 years of age. The study was approved by national ethical authorities.
The patients were aged 4 years, 9 months to 7 years (average age of 5.6 years), and included 16 girls and 38 boys. No parents refused to allow their children to participate in this study. Five patients presented with isolated cleft lip, 10 patients had a cleft lip and alveolar cleft, 27 unilateral cleft lip and palate, and 12 had bilateral cleft lip and palate. Children with hearing impairment or not followed by an ear, nose, and throat (ENT) specialist were excluded. Children with language trouble diagnosed by a speech therapist were also excluded as well as parents and children who did not speak French fluently. In our sample, 88.8% of parents were in a relationship and 91.7% of fathers and 72.7% of mothers were employed. Children's ethnic backgrounds included European (81.4%), African (9.2%), Asian (5.5%), and Caribbean (3.7%). No children were adopted. At school, 59.2% of the children of the study sample were in a nursery school full time, 37% were in first grade, and 3.7% were in second grade. This study took place before COVID-19.
The authors first completed semistructured interviews with parents and children, followed by interviews with children alone. Children were asked to explain their scars and were administered a projective test named “The not-the-same baby bear.” 21 Children were also administered a verbal assessment in order to identify any language difficulties that might impact the results.
Semistructured Interviews of Parents and Children
Semistructured interviews with parents and children followed an interview guide. Parents were asked to explain if they had already told their child the story of his or her cleft and, if so, how they explained it. Responses were classified into 4 categories: (1) only sharing easily accessible pictures at birth with no explanations regarding the cleft story, (2) explanations regarding the cleft alone, without showing the child pictures at birth, (3) using both pictures and explanations, or (4) neither showing pictures nor giving explanations about their CL ± P.
Children were surveyed using semistructured interviews on their own. Children were first asked if peers in school asked questions about their cleft lip scar or about their physical difference and, if so, how often questions were asked and how they dealt with these questions. They were also asked if they faced repeated questions. Children were then asked to tell the story of their scar with the following question: “Could you explain to me why you have a little scar?” Their answers were evaluated according to the presence or absence of the following elements, indicating whether their narrative was clear to other children (all 3 elements present) or not (<3 elements present): (1) time awareness—presence since birth, (2) space awareness—localization on the lip/mouth, and (3) awareness of surgery—the concept of initial cleft and subsequent closure. In recommendations given by specialized associations such as CLAPA 19 in the UK and ACPA's Family Services “Help with Social Situations,” 20 children are encouraged to use these 3 elements in their narrative when they explain their diagnosis to others. It should be noted that most of the time when children are answering a question about their difference, the wording of this question already contains a localization like “lip” or “mouth,” so time awareness and awareness of the surgery should be considered keys to clear answers.
Projective Tests of Children
After their semistructured interview, children were administered a projective test. This projective test consisted of a story that needed to be completed by the child. Projective tests generally present ambiguous stimuli for individuals to respond to with an unstructured format that allows for a more subtle way to measure psychological variables than more structured formats. 22 For instance, when children are invited to build a story based on an image of a family, the story they create is thought to reflect their representation of their parents, their relationship with them, and their family relationships. The use of a standardized projective measure for storytelling helps elicit children's narratives and learn about their internal world. This method also assesses children's ability to organize their feelings into coherent stories. This tool can be used with children as young as 3 years old. 23
The verbal projective tool used, named “The not the same baby bear,” was selected for the age group of the participants, and for the theme explored of “not being the same” or “feeling different.” As the experience of being different is often part of social interactions for children with a cleft,6,7 a general sense of being different was the focus of the projective measure and allowed for comparison with a norm group. This projective test was developed by Jacqueline Royer in 1978 21 and validated by C. de Tychey in 1993 24 and 2010 25 after being tested on a population of French children. De Tychey suggested that this projective test could assess their positive or negative self-perception of being/feeling differently from their parents. 25 Golse and Missonnier 26 supported the hypothesis that the quality of storytelling or “narrativity” strongly depends on the quality of attachment relationships.
The projective measure of developing stories is presented as a game. This projective tool refers to the main character who is a baby bear defined as different from the others. It starts with, “In the mountains, a mother bear had cubs. One was not the same as the others.” First, children were invited to describe in which manner this cub feels different: “What was ‘not the same’ with him? Why was he not the same?” This first question focuses on children's self-identification with the bear by questioning the nature of the difference between the baby bear. 25
The next questions were designed to explore the way children had internalized their mother's, father's, and sibling's image of them (ie, mother/father/sibling acceptance). According to C. de Tychey, 25 the second question provides information about the loving acceptance of parents towards their child: “What did the mother bear think of her baby bear who was not the same? Did she like him? What about the daddy bear? What about the other cubs, his brothers and sisters?” This question is evaluated as “Positive/negative maternal/paternal/sibling acceptance.” The test then explores the way children see themselves as different from others: “How does the little bear himself feel about being different? Does he like himself?” This question is evaluated as “positive/negative self-image.” C. de Tychey suggests that this third question provides an indication of children's self-perception in positive or negative terms. 25
Finally, the projective test assesses the perception children have of their future. According to C. de Tychey, “this last question informs about the capacity of the child to project himself in the future in a positive or negative way, which reflects a sense of value, a child's confidence or not in his or her possibilities.” 25 Children are asked: “What will happen to the baby bear at the end of this story? Is the story ending badly or happily?” Recommended evaluations for this question are “positive/negative/mixed ending.”
Assessment of the Children's Language Ability
In order to describe children's language development as a key skill needed for developing their story, all children included in this study had their language ability level assessed. The language scales (Verbal Comprehension: Information and Similarities and Vocabulary Acquisition: Receptive vocabulary and Picture naming) of the Wechsler Preschool Primary Scale of Intelligence, Fourth Edition (WPPSI-IV)26,27 were used, as they are already normed with French children of the same age. Three children were not tested with the WPPSI-IV and 2 children only had the Verbal Acquisition scale because their parents did not have time to participate in that portion of the study visit.
Statistical Assessment and Control Group
Two types of tests were performed: Hi-square (K*) test when no variable in the results table was <5 and the Fisher test (F*) when at least 1 variable was <5. Study results were compared with results obtained in the general population (control population). The control group for the projective test has been previously published by C. de Tychey in 2016 and includes French-speaking children (N = 40) with no specific condition, aged from 4 to 6 years (results summarized in Table 1).
Projective Test Results of the Cleft Group Compared to French Population (Control Group: 4-6 years old, 40 Children).
Results
Language Screening
The majority of children (83.6%) had at least a normal Verbal Comprehension Index (VCI) compared to the French population (VCI≥80), while 12.2% of children had a borderline score (70≤VCI≥79) and 4% had an extremely low score (VCI<70). Nearly all children (96%) had at least a normal Verbal Acquisition Index (VAI) compared to the French population (VAI≥80) and 3.7% had an extremely low score (VAI<70).
Semistructured Interviews with Parents and Child
There were 27 families (50%) who provided explanations to their children about the story of their cleft and showed pictures dating from birth. There were 12 families (22.2%) who showed preoperative pictures to their children without any specific explanation. Six families (11.1%) never showed pictures, but explained the story of the cleft to their child. Nine families (16.6%) neither showed any pictures nor provided any information to their children. Three families out of these 9 (5.5%) did not have any pictures of their child before primary surgery (either they had not taken pictures of their child for several months before surgery or the only pictures they had from this period had been lost).
Parents spontaneously reported that the interview had been useful and contributed to helping them in building the story of the cleft with their child. Many parents said they learned during the interview that children were facing questions about their diagnosis.
Semistructured Interviews with Children Alone and Assessment of Storytelling Regarding their Diagnosis
There were 21 children (38.9%) who reported never having been questioned about their scar or facial difference. The remaining 33 children (61.1%) reported that they had been questioned about their facial appearance and 5 of these 33 children (15.1%) children reported repeated or/and embarrassing questions or teasing.
Only 18 children (33.3%) were able to provide complete answers to questions on the origin of their scar based on 3 key elements: time awareness (birth), space awareness (the child says “lip” or points his lip), and awareness of surgery (lip closed). Examples of answers showing such awareness were: “I was operated on when I was a baby. There was a hole on my lip and it was repaired.” or “I had a hole here [the child then points to his/her lip] when I was born and the doctor stitched it.” “Hole” was often used by children to designate the nonoperated cleft. No children forgot to tell or show space awareness in the responses. Time awareness or awareness of surgery were sometimes not included in children's responses, as these concepts may have not been as clear for them. Complete storytelling about their cleft was not related to children's age, gender, cleft type, or language abilities.
Storytelling regarding the scar was strongly influenced by the way the parents told the story of the diagnosis to their child (Table 2). Depending on the method used, the narrative was more or less complete (p = .0004). Children who had both seen pictures of themselves as babies before surgery and were told a narrative by their parents regarding the history of their cleft accounted for 89% of children who provided a complete narrative. However, it should be noted that 20% of the children with less complete narratives had also been given an explanation and shown preoperative photographs.
Storytelling and Parental Method Used to Tell the Story of the Cleft to Their Child.
F* = 0.0004, p ≤ 0.01.
Projective Test
Children's self-image: There were 23 children (42.5%) whose stories indicated that they had a positive image of their difference and themselves in their projective response about the “not-the-same baby bear” story (eg, “He was ok with him being different, it was not a big deal.”), while 29 children (55.7%) gave answers suggesting a negative image of themselves (eg, “It was not pretty.”). When comparing all children with CL ± P to the control group, there was a significant difference (F* = 0.0031, p ≤ .01) with 70% of the control group classified as having a positive self-image (Table 1).
Parental acceptance: There were 22 children (43%) who constructed a response indicative of positive maternal acceptance of difference while 30 children (57%) spoke of a lack of maternal acceptance or negative maternal judgment (“The mother wanted to sell him. He bothered everyone.”). It was significantly more common (F* = 0.0255, p ≤ 0.05) to find responses that suggested a lack of maternal acceptance of differences in children with CL ± P than in the control group (Table 1). For paternal acceptance, there were 39 children (75%) who constructed a response suggesting the same level of acceptance for the father and the mother while 12 children (23%) spoke of a better acceptance of the father compared to the mother, and only 1 child (1.8%) indicated less acceptance by the father. There was no significant difference compared to the control group.
Siblings’ perception and judgment about the difference: Most children in our group (61.1%) expressed a negative perception of siblings toward their difference. This result in Table 1 stood out strongly compared to controls (F* = 0.0424, p ≤ .05).
Positive or negative ending of the story: In our study, 25 children (48%) responded with a positive ending to their story (“Then it grows up and all ends well” or “Then he liked not being the same as the others”). There were 12 children (23%) who gave a negative ending to their projective story (“He was always annoyed and angry. He was sent to stand in the corner. The mother spanked him and the dad kicked him in the backside.” or “He gets eaten by a wolf”). Finally, 15 children (29%) chose a mixed or neutral ending (“After that it will be the same thing again, nothing is going to change.”). Besides an interest in raw data exploring a cleft population, it is worth noting that these figures show a significant difference with controls (Table 1), particularly regarding the proportion of negative and mixed endings, which may suggest a greater difficulty for children with a cleft to see themselves positively in the future (F* = 0.0367, p ≤ 0.05). Finally, there was a recurrent theme in the constructed stories that require further investigation as 8 children (15.8%) told their stories where the question of biological parentage was called into question. (“He was pretty, but in fact they were not his real dad and his real mom.” or “In the end he found his real dad and his real mom.”)
Interactions with storytelling: Complete storytelling was directly influenced by a positive or a negative self-image, as evaluated through the projective test, with 44% of children with CL ± P who had incomplete stories about their cleft and negative self-image (K* = 0.0178; p ≤ .05) (Table 3). Complete storytelling was influenced by the maternal acceptance of the difference, with 48% of children who had incomplete storytelling relating stories with a lack of maternal acceptance in their projective test (K* = 0.0015, p ≤ .01) (Table 3). Similarly, complete storytelling was influenced by paternal acceptance, with only 10.5% of children with a lack of paternal acceptance constructing complete storytelling (F* = 0.0091, p ≤ .01) (Table 3). Language ability level had no influence on the completeness of children's diagnosis storytelling (F* = 0.72, p > .1).
Interaction Between Storytelling and Projective Test Results.
Maternal acceptance and storytelling: (K* = 0.0015; p ≤ .01). Self-image and storytelling: (K* = 0.0178; .01 < p ≤ .05). Paternal acceptance: (F* = 0.0091; p ≤ 001).
Discussion
As a factor facilitating psychosocial adjustment, the aim of this study was to assess children's storytelling about their cleft diagnosis, their self-image, and perceptions of familial acceptance. In this sample, 61% of children reported having been asked questions about their diagnosis by the age of 7, yet only 30% of children between 4.10 and 7 years of age provided what was considered a complete explanation of their cleft diagnosis. The literature indicates that the first questions asked directly to children by their peers when their parents are not present to help them usually occur around 4 or 5 years of age. 16 A peak frequency of when children find themselves with peers they do not know occurs at the time of entry into elementary school (around 6 years old). From children's perspective, this often represents the first time that their “narrative identity” (ie, their ability to tell something about themselves) is challenged. This function is coconstructed in the parent–child interaction, in what the parent communicates to their child about themselves, like a reflection in the mirror, but also in the words the parent uses to talk to and about them. 26 Indeed, P. Ricoeur stated that the ability to understand oneself is based on the stories individuals tell themselves, which need to be clear and acceptable.28 The literature suggests that the answer given by children about their diagnosis can influence the response of their peers. If the child cannot answer, if the answer is unclear, or if the child tries to ignore the question, peers are likely to come back to ask the same question repeatedly. Sometimes other children's curiosity is awakened by this half-perceived secret or they can use this easy target as part of typical children’s quarrels. 10 Finally, this age of 4 to 6 years is also the age in France at which bone grafting is indicated for labio-alveolar or labio-palatine clefts. This surgical procedure is often the first time that children are developmentally able to fully participate in and remember. It could be therefore an opportunity for the multidisciplinary team not only to reassess how aware children are of their cleft, but to also include them in the treatment process by speaking to them directly during the preoperative visits and by ensuring that they understand and agree with the treatment plan. This step is often crucial for children to adhere to the follow-up of the treatment, to take part in decisions, and to understand their meaning and benefits.29
Children's ability to tell their cleft story was directly dependent on their parents’ explanations to them and this study also highlighted that even good explanations were sometimes not enough to help children answer questions adequately about their cleft (as 20% of children with less complete storytelling had seen preoperative pictures and had explanations about their cleft).
The standardized projective test offered the opportunity to see how children with a cleft lip with or without a palate feel about “being different” in comparison to a general population at the same age. While 42.5% of children with CL ± P had a positive self-image, the results suggested that 55.7% of them had a negative image of themselves. This result corresponds to clinical experience with children at this young age describing the difference in a negative way and expressing that they wish to be the same as their siblings. For example, this was illustrated in a response of a child with a cleft while telling the story about the “not the same” baby bear, saying “He didn't like not being the same as others,” explaining that the feeling of being different was hard to be positive about.
Having a positive or negative self-image about being different in their projective responses was also related to their ability to explain their cleft. While this study cannot answer if this relationship is causal or correlational, the ability to talk about one's diagnosis is thought to be a foundation for children's self-confidence. Complete storytelling was also influenced by the parental acceptance of their child being different in the projective test. This result emphasizes the parental need for psychological support after a cleft diagnosis is made. Parents’ interviews in this study consistent with the literature9,30 showed that parents may frequently feel helpless to support their child in social interactions when the diagnosis has strongly affected them, when they feel shame, or have not developed narrativity about their child's cleft. For those children who have parents experiencing distress and may not be able to address the topic of their cleft with them,31 little is known regarding the strategies those children use to talk to their peers about their cleft. Parents in this study were generally appreciative of the opportunity they were given to think about their family's cleft story and of the support they received in the course of the study to find their own way of talking about their cleft story with their child, which was still sensitive for some parents. Indeed, several parents were anxious that their child would feel the same distress that they themselves experienced as parents when the cleft was diagnosed. Despite the distress expressed by some parents, none of the parents spontaneously requested to meet with the psychologist to help them explain the cleft diagnosis to their child. However, when offered as part of the study, no parents rejected psychological assistance. This suggests that cleft teams should uniformly inform parents early on about the importance of the ability for children to be able to explain their cleft. Additionally, cleft teams may consider an offer for a psychological consultation to all children in the team between 5 and 7 years old as it seems that parents do not perceive forming cleft explanations as a reason to ask for psychological support.
Finally, this study highlights a possible area of difficulty encountered by this population of children in their relationship with peers in needing to respond to questions about their cleft, particularly as some of them did not talk about it with their parents. It is also important to note that 16% of children created a story in their projective responses questioning the biological link between the protagonist and his or her parents, which may be indicative of children with a cleft not seeing themselves reflected in the appearance of their parents without a cleft. While some questions from peers may reflect age-appropriate curiosity without any negative intention, about 10% of the sample reported being embarrassed or having questions asked of them repeatedly. This suggests that asking about children's experiences with questions from peers about their cleft is crucial to understanding the difficulties encountered by some children with socialization. It is important that members of cleft teams support parents prenatally and after birth in their own coping and adaptive parenting skills. Teams should also contribute to children with a cleft form of a positive self-image and self-acceptance, learn ways to explain their diagnosis to peers that they are comfortable with, and general social skills.32 Multiple ways to support forming an explanation are crucial; as the study results illustrated that 20% of children had difficulties explaining their cleft even with parental explanation and sharing preoperative photographs.
In this sample, children's ability to answer questions about their cleft was not related to their language ability. For example, 32 (65.3%) children in our sample who had at least an average VAI constructed an incomplete story. In addition, 10 children out of 15 (66.6%) who had at least a high normal score (VAI≥110) did not construct a complete story. This result highlights the fact that it is not only a question of verbal development for children to be able to form a complete explanation about their cleft.
Clinical Recommendations
Regarding parents, the essential psychological support offered to them by the team's psychologist should not be the only resource available because some parents, quite often those encountering the most severe difficulties, may reject or avoid this help due to stigma related to psychological support. Other types of support at the time of diagnosis and after birth should be considered by the team, such as antenatal parent support groups led by a nurse, breastfeeding support led by a nursery nurse, parent and baby massage sessions led by a childcare assistant and childcare worker, and psychoeducation materials for parents. All these types of care can help reduce parental isolation and allow them to build relationships with different professionals to whom they can address their questions and talk about their possible difficulties. Finally, professionals should also be informed about the online social networks and discussion forums dedicated to clefts, as well as accurate and well-vetted websites, in order to be able to guide parents who would like to meet other families or gather additional information. Furthermore, reading the online exchanges that are publically accessible can also inform professionals about the gaps in care and the needs of parents.
Regarding children, their understanding of their own story is crucial. Support should be offered to the parents and to the child through a psychologist within the cleft center. Group workshops of therapeutic education for children on the subject of others’ reactions to a cleft can complement this support. These workshops can help the child to build relationships with others, learn coping skills for possible remarks and questions, and develop an adaptive attitude toward dealing with teasing.33 This support helps guide children so that they can construct a typical “short sentence” that they can use whenever others question them about the visible marks of the cleft, speech, or other aspects of their diagnosis. In online booklets for families,19,20 for this short sentence to work and for it to be easily understood by other children, it should contain these 3 target elements: (1) When I was born (concept of time awareness), […]. (2) My lip (and/or my palate) […] (space awareness) was not closed (concept of open/closed) […]. (3) A doctor sewed it/I had surgery (concept of surgery), that is why I have a scar. Children's booklets about cleft stories are also important to promote and support parenting and storytelling. This first narrative should go beyond the family circle and the hospital environment to include the school environment. Some teachers, at the request of the child, use these literature materials to explain the child's diagnosis and extend the message to accept differences in general.
Limits of This Study
While both the projective test and WPPSI-IV have been validated in French, the projective test was not validated on a large number of subjects. The self-image variable used in the interpretation of the projective measure may refer to issues beyond being born with a cleft and can be further measured with questionnaires or other tools. However, the projective test was an age-appropriate method to effectively gather a wide range of information in a young sample. We did not address how sociodemographic variables could impact language development or family composition, such as having older siblings, may have impacted results. For coding interviews, the authors did not assess the interrater reliability of the categories. Finally, it would be worthwhile to extend this research to a larger population of patients from several other French centers and internationally.
Conclusion
We showed that about a third of children were able to clearly explain the origin of their cleft lip scar, which was linked to their self-concept and how parents explained the story of their cleft. Supporting parents so that they can appropriately discuss CL ± P with their children can include meeting with the team psychologist during antenatal care or within the first months following birth for prevention purposes. It is important to design therapeutic tools intended to support parents from diagnosis throughout their child's development, to help parents in developing a narrative to tell their child, and to guide children in social skills, including a set response to questions and how to deal with comments and teasing. The study results also show that it is important for cleft professionals to address with patients ages 4 to 7 years their own understanding of their diagnosis with questions like: “Have you ever had questions about your scar or anything else related to your cleft?” and “Are you able to explain why you have a small scar on your lip or answer other questions?” These simple questions and the answers provided allow for screening in order to discuss responses during consultations, connect families to resources, and refer families as needed to a psychologist.
Footnotes
Authors’ Note
This study was approved by National ethical authorities (CPP Ouest 6, CHU Cavale Blanche, Avenue Tanguy Prigent, 29609 BREST Cedex cpp.ouest6@chu-brest.fr ID RCB 2017-A02495-48).
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.
