Examining the Psychosocial Needs of Adolescents With Craniofacial Conditions: A Mixed-Methods Approach

Abstract

Objective:

To determine, for intervention development, the psychosocial needs of adolescents diagnosed with a craniofacial condition who attended focus group sessions.

Design:

A mixed-methods design combining qualitative focus groups with quantitative measures.

Setting:

An outpatient clinic at a major medical center in Manhattan, New York.

Participants:

Fourteen adolescents, aged 14 to 18, with craniofacial conditions.

Main Measures:

Participants completed measures assessing a range of psychological constructs. Average scores were compared to clinical cutoff scores and normative data for adolescents. The 2 focus groups were coded using an inductive approach to assess pertinent themes. Additionally, the acceptability and feasibility of a proposed intervention was measured.

Results:

Adolescents with craniofacial conditions were within normal ranges for quality of life, self-esteem, and body image and they reported higher resiliency. They were above cutoff scores for perceived stress and post-traumatic stress disorder symptoms and below cutoff scores for mindfulness. When compared to normative samples, they displayed higher perceived social support, but lower coping. Based on qualitative analyses, 6 themes emerged: stress, bullying, coping, resiliency, mindfulness, and social support. Both qualitative and quantitative analyses revealed most participants were supportive of a future intervention for this population.

Conclusions:

The present study identified several factors associated with psychological well-being of adolescents with craniofacial diagnoses and demonstrates the importance of creating interventions to target specific psychosocial needs. Findings from this study may guide researchers in developing and refining a specific program for this population and provide information to help those with craniofacial conditions who are experiencing psychosocial challenges.

Keywords

pediatrics psychosocial adjustment quality of life social support psychological assessment

It is estimated that 2% to 3% of all babies are born with a craniofacial condition (CFC) that significantly alters their life trajectory (World Health Organization, 2003). There are several multidisciplinary interventions in place for the treatment of CFCs, including surgical interventions (eg, plastic surgery, facial advancements, neurosurgery, orthodontic work, speech surgery, etc), speech therapy, and mental health treatment, among others (Bemmels et al., 2013; Susanu et al., 2018). These approaches not only can improve an individual’s physical functioning but they have also been found to impact their psychological functioning (Susanu et al., 2018). However, treatment gains can vary by age, type of intervention, and patient experience (Johns et al., 2018). Although surgical interventions are viewed as standard of care, research has shown that some young adults experience emotional and social distress after surgeries, possibly due to unmet or unrealistic expectations regarding their appearance and self-concept (Topolski et al., 2005; Bemmels et al., 2013). It has been found that their quality of life (QoL), resiliency, and self-esteem can decline when there are persistent feelings of inadequacy or unattractiveness after these procedures (Edwards et al., 2005). Further, those who have multiple plastic surgeries tend to have more negative perceptions of themselves at an early age (Patrick et al., 2007), which may be exacerbated during adolescence. Given these suggested long-lasting effects, some have characterized CFCs as a lifetime stressor that “exacerbates the pressures associated with various developmental stages,” and thus, appropriate support is needed in relation to these stress-related and potentially traumatic responses (Rumsey and Harcourt, 2007, p. 114). Adolescence is a difficult transitional period, and more research is needed to understand the specific challenges faced by those with CFCs. Understanding the baseline psychological functioning of adolescents with CFCs may help develop appropriate interventions for this population.

Some prior research has demonstrated lower self-esteem and QoL, significantly more distress, and worse psychological functioning among individuals with CFCs compared to the general population (Patrick et al., 2007; Feragen et al., 2009; Bessell et al., 2012). However, the levels of psychological functioning and stress vary among studies. For example, some studies have found that adolescents with CFCs do not differ in psychological adjustment or stress compared to their peers, whereas other studies have indicated discrepancies between reports of adjustment to CFCs (Rosenberg et al., 2011; Feragen and Stock, 2017; Johns et al., 2018). These findings highlight the complexity of psychological functioning in CFC populations.

Studies examining QoL, psychosocial adjustment, and social skills development among adolescents/young adults with CFCs have shown compromised functioning due to difficulties in social situations (Kapp-Simon et al., 2005; Topolski et al., 2005; Roberts and Shute, 2012; Feragen and Stock, 2017). Social stigma and derogatory social responses (ie, staring, laughing, mocking) associated with CFCs can lead to negative self-perceptions of attractiveness, increased appearance-related distress and anxiety, and feelings of incompetence and loneliness (Kapp-Simon et al., 2005; Roberts and Shute, 2011; Bessell et al., 2012; Crerand et al., 2017). Due to negative social responses, some adolescents with CFCs may exhibit social withdrawal in order to avoid painful social interactions. This lack of social exposure can lead to fewer opportunities to develop social skills and hinder social adjustment. This, in turn, can make it more difficult for individuals with CFCs to maintain and develop relationships, which negatively affects their self-concept, social competence, and self-satisfaction (Kapp-Simon et al., 2005; Edwards et al., 2011; Feragen and Stock, 2017). Moreover, Feragen et al. (2009) found that social stigma, teasing, and bullying not only negatively affect psychosocial functioning of those with CFCs but may also impact resilience and prevent them from effectively coping with negative social interactions. Thus, teaching social skills to adolescents with CFCs could increase the number of social interactions and the likelihood of developing more positive social relationships.

Studies have proposed resiliency and social skills training for young adults with CFCs as potential interventions to improve QoL, psychological well-being, and the ability to cope in difficult situations (Kapp-Simon et al., 2005; Topolski et al., 2005; Rumsey and Harcourt, 2007; Feragen et al., 2009; Bessell et al., 2012; Johns and Bava, 2019). Kapp-Simon et al. (2005) investigated the effect of social skills training on social interactions among adolescents with CFCs and found that those in the social skills training had a greater increase in the overall rate of social interactions and were 3 times more likely to engage in a conversation with their peers than they were before starting the group. Similarly, Johns and Bava (2019) compared baseline and postgroup psychosocial functioning to test norms among pediatric patients, aged 7 to 18, who completed support groups for those with CFCs. They found that postgroup participants reported significantly lower social stress than peer norms and participants displayed significant improvements in psychosocial functioning after completing the groups, including adaptive skills, self-esteem, and social stress (Johns and Bava, 2019). Prior research suggests increased social support may improve psychological outcomes, and some adolescents with CFCs are able to cope well in difficult situations (Meyerson, 2001; Strauss, 2001; Beaune et al., 2004; Roberts and Shute, 2012; Feragen and Stock, 2017). Johns et al. (2018) found that those with craniofacial microsomia used various coping strategies to improve resilience and enhance personal growth (eg, utilizing support systems, positive reframing techniques, acceptance, openness, self-confidence, etc). Similarly, Beaune et al. (2004) found that adolescents with Treacher Collins syndrome utilized resilient adaptive strategies (eg, optimism, motivation, and positive meaning making) to improve psychosocial adjustment and increase self-acceptance. Moreover, enhanced resiliency and optimism in this population could lead to improved health and well-being (Locker et al., 2005; Feragen et al., 2009). Although resiliency has been considered an innate characteristic that cannot be taught (Neihart, 2006), more recent research has shown how enhancing resiliency in various training modules among individuals with medical conditions can improve psychological and physical health (Tugade et al., 2004; Vranceanu et al., 2016). Improving resiliency in adolescents with CFCs would be particularly pertinent as they transition to adulthood. These findings suggest resiliency and social skills training may be beneficial for those with CFCs, and future interventions should consider adopting these strategies.

Adolescents with CFCs may experience significant distress and dysfunction, but the avenues to best address these concerns remain understudied. The current study sought to describe the psychosocial needs of adolescents diagnosed with a CFC through the use of focus groups. Qualitative studies provide further interpretation of health outcomes, enhance our understanding of contradictory findings, and contribute to the development of new interventions (Beaune et al., 2004; Roberts and Shute, 2011; Bogart et al., 2012). On quantitative measures, it was hypothesized that participants would fall below the clinical cutoff scores for QoL, self-esteem, social skills/supports, body image QoL, and mindfulness (ie, the ability to stay in the present moment and perceive thoughts and feelings without judgment), and above the cutoff scores for distress, resiliency, coping, and post-traumatic stress disorder (PTSD) symptoms. Participants were also asked for feedback on possible intervention approaches to gauge feasibility and acceptability.

Methods

The current study took place in an outpatient clinic at a major medical center in Manhattan, New York, in conjunction with myFace, a New York-based nonprofit craniofacial organization dedicated to providing funds and support for medical and psychosocial services and foster research and public awareness. Participants were recruited through myFace’s electronic database via e-mails and flyers. Those interested contacted the study team to discuss the study, review study criteria, and provide written informed consent. Inclusion criteria included: (1) age 14 to 18 years, (2) confirmed CFC diagnosis, and (3) fluency in spoken and written English. Exclusion criteria included: (1) inability to understand the content of the group due to cognitive, psychiatric, or other reasons based on the opinion of the group leader, a trained psychologist who would evaluate each participant’s level of understanding, and (2) inability or unwillingness to sign informed consent documents. Adolescents, aged 14 to 17 years, who agreed to participate, required a parent or guardian’s signature and signed assent on a separate assent form; those who were 18 years old signed their own consent form. After signing the informed consent document, participants completed baseline measures prior to starting the focus groups. A complete list of measures can be seen in Table S1, which includes the title, format, psychometric properties, and description of each measure. Comparative data used for each measure in the quantitative analyses are also described within Table S1, Carver (2006); Cash and Fleming (2002); Cohen and Janicki-Deverts (2012); Devilly and Borkovec (2000); Greco et al. (2011); Jerson et al. (2013) and Sherbourne and Stewart (1991).

Study participants (n = 14) joined 1 of 2 focus groups held in either June 2017 (n = 10) or April 2018 (n = 4) and were organized on a first come, first served basis, regardless of age or gender. Both focus groups ran approximately 3 hours with a lunch break midway through to prevent participant fatigue. Groups were semistructured and included topics relevant to growing up with a facial difference (ie, stressors, coping skills, social and community supports, etc). Group leaders also incorporated various activities to facilitate conversation and to trial potential interventions (ie, icebreakers, role-playing, mindfulness meditations). The role-playing activity lasted about 25 minutes and was conducted during the social skills/support portion of the focus groups, while the meditation lasted about 10 minutes and was done during the mindfulness section of the groups. Both of these activities were integrated into the focus groups to examine whether participants would be interested in including these in a future intervention. At the end of each focus group, the group leaders introduced the idea of a future weekly or monthly group intervention that would incorporate many of the topics discussed throughout the focus group (eg, stress, resiliency, social skills, etc).

To prevent participant bias and to facilitate open-ended discussion, the group leaders provided little information and detail regarding the future intervention. The following quote is taken from the script used to discuss the participants’ preferred modality for a future group: “We are hoping to develop a program to help adolescents with a facial difference manage their stress, and build skills to promote resiliency and social skills.” Subsequently, the group leaders sought participants’ feedback about the future intervention. Following the focus group, participants completed the adapted Credibility/Expectancy Questionnaire (CEQ). These procedures were approved by the institutional review board of NYU Langone Medical Center.

Analyses

Descriptive statistics were conducted for each of the measures to examine whether scores were within normative ranges. Both focus groups were audio-recorded, transcribed verbatim, and coded using an inductive approach to assess pertinent themes. Two independent coders, who were provided resources to achieve comparable levels of exposure to the craniofacial literature, analyzed the transcripts for themes that represented broader ideas from the focus groups. The researchers met to discuss and refine potential themes. There were no coding discrepancies between the 2 independent coders. Additionally, researchers coded transcripts for responses related to the proposed program.

Results

Descriptive Statistics

Table 1 provides a summary of demographic information. A total of 14 adolescents completed the study. The average age of the sample was 15.43 years (standard deviation [SD] = 1.22), and the majority of the sample was female (71.4%), white/Caucasian (71.4%), and had cleft lip and/or palate (CL/P; 50%). In the first focus group (n = 10), the average age of the sample was 15.3 years (SD = 1.06), and the majority of the sample was female (60%), white/Caucasian (70%), and had CL/P (40%). In the second focus group (n = 4), the average age of the sample was 15.75 years (SD = 1.71), and the majority of the sample was female (100%), white/Caucasian (75%), and had CL/P (75%). Table 2 displays mean scores on the measures with clinical cutoff scores. Due to the limited data on clinical interpretations for the Measure of Current Status Part A (MOCS-A) and MOS Social Support Survey, participants’ average scores on these measures were compared to normative sample data.

Table 1.

Demographic Characteristics of the Sample.

Demographics	Mean	SD	Range
Age (years)	15.43	1.22	14-18
Education level completed	9.71	1.33	8-13
		n	%
Gender
Female		10	71.4
Male		4	28.6
Craniofacial diagnosis
Cleft lip and/or palate		7	50.0
Craniosynostosis		1	7.1
Hemifacial microsomia		2	14.3
Other		4	28.6
Learning disability diagnosis
Yes		5	35.7
No		7	50.0
Unsure		2	14.3
Race
Asian		2	14.3
White/Caucasian		10	71.4
Other		2	14.3
Ethnicity
Not Hispanic or Latino		10	71.4
Hispanic or Latino		4	28.6
Parent income (yearly)
≤US$45 000		4	28.6
US$45 000-US$65 000		2	14.3
US$65 000-US$85 000		1	7.1
US$85 000-US$105 000		2	14.3
US$105 000-US$125 000		0	0.0
US$125 000-US$145 000		3	21.4
>US$200 000		1	7.1
Did not specify		1	7.1

Abbreviation: SD, standard deviation.

Table 2.

Average Scores for Measures Compared to Clinical Cutoff Scores or Comparative Samples.

Measure	Craniofacial Sample		Clinical Cutoff Scores
Measure	Mean	SD	Score Ranges	Clinical Interpretation
PedsQL total	73.9	13.1	>69.71^a	Not at risk
PedsQL physical	80.6	21.3	>72.98^a	Not at risk
PedsQL emotion	63.6	17.9	>59.57^a	Not at risk
PedsQL social	75.7	14.7	>66.61^a	Not at risk
PedsQL school	71.8	16.2	>62.99^a	Not at risk
PSS-10	14.8	6.4	14-26^b	Moderate stress
RS-14	81.1	9.8	64-81^c	High resilience
RSES	31.8	5.5	25-35^d	Normal range
BIQLI	1.8	1.0	>−0.95^e	No eating disorder indicated
CAMM	20.9	6.3	0-23^f	Low mindfulness
CPSS symptom total	15.6	9.6	≥15^g	Meets criteria for PTSD
			Comparative Sample
			Mean	SD
MOCS-A	35.1	9.1	41.0^h	8.5
MOS Social Support	81.8	13.3	77.1ⁱ	19.9

Abbreviations: BIQLI, The Body Image Quality of Life Inventory; CAMM, Child and Adolescent Mindfulness Measure; CPSS, Child PTSD Symptom Scale; MOCS-A, Measure of Current Status Part A; MOS Social Support, Medical Outcomes Study Social Support Survey; PedsQL, Pediatric Quality of Life; PSS-10, Perceived Stress Scale; PTSD, post-traumatic stress disorder; RS-14, The 14-Item Resiliency Scale; RSES, Rosenberg Self-Esteem Scale; SD, standard deviation.

^a Varni et al. (2003).

^b Cohen et al. (1983).

^c Wagnild (2009).

^d Rosenberg (1979).

^e Hrabosky et al. (2009).

^f Patterson and McDonald (2015).

^g Foa et al. (2001).

^h Park et al. (2017) (n = 26).

ⁱ Martyn-Nemeth et al. (2009) (n = 102).

Qualitative Analyses

Based on the qualitative analyses, 6 pertinent themes were found.

Stress

Stress was one of the most common themes throughout both focus groups. Participants were easily able to define stress and described stress as both a positive and negative construct. Some participants depicted stress as positive since it could be motivating (eg, “Maybe stress to do well on a test or do something that’s good for you…Because stress could also mean someone pushes you towards something.”). Others expressed stress as negative, since it could be detrimental to their emotional well-being (eg, “I was going to say maybe [stress is] something that’s hard for you because you’re anxious to do it, or you feel a little bit of anxiety doing it, or it makes you feel, I don’t know, bad in a way, or it’s just hard for you to get over.”). Participants endorsed external stressors including school, friends and family, meeting new people/dating, social media, and surgeries. Interestingly, although participants categorized school as a stressor, they also acknowledged a stress reduction over time. One participant described middle school as “brutal” and “awkward,” but later explained that it got better in high school:

You know, in middle school I was having trouble making friends because I was bouncing around trying to figure out who would be a true friend to me, and I didn’t find that in middle school. I found it in high school.

Participants also mentioned several internal stressors including lack of control or self-confidence, lack of understanding from others, and difficulties with expressing emotions. For example, one participant described the difficulties of expressing herself due to the lack of understanding from others:

If there’s one thing that I hate, it’s that nobody will understand. Nobody will truly understand…You will never understand my pain. You will never understand going to a doctor or going somewhere and continuing your life. People are like, “Oh! What’s next for you?” I’m like, “I have no idea!”…The reason I don’t share my feelings or my true true feelings is because no one will truly understand what it’s like.”

Bullying

Although bullying was considered a stressor to most, it warranted its own thematic category. Those who had (n = 11) experienced bullying described it as deviating from the norm and as one participant explained, “if you did not fit a certain kind of mold that they already came in and set for everyone, then [bullies] were going to make your life a living hell.” Participants described different types of bullying (eg, consistent bullying versus single event) and explained how bullying had become subtler over time:

The one thing about my school, which was disgusting, was that they bullied in a way that they couldn’t get in trouble for because it was cynical. And that’s how bullying has morphed itself…They’ll pretend to be your friend, and then they’ll make fun of you behind your back…They will make you look like a fool and they will pretend to be your friend and make you think you’re cool.

Additionally, participants discussed differences between bullying in public versus private schools and gender differences in bullying. Lastly, participants discussed the impact of social media on bullying experiences:

There were a lot of mean people [online]. I had someone…I don’t even know this person. They live in another country…told me that I look like I swallowed a curling iron because of my lips.

Coping

Many participants mentioned various coping skills that helped with stressors and bullying. Some participants learned these skills from their parents, family members, therapists, or other support systems, while others were self-taught. Participants mentioned skills such as deep breathing, restructuring negative thoughts, optimism, journaling, and mindfulness or staying in the present moment. Restructuring negative thoughts and utilizing optimism seemed to resonate among participants as noted by these 2 quotes:

I tried to focus my energy on things that I can control because we cannot control that we have surgeries coming up. That is not something that’s our fault…It’s also important to realize that what you’re going through is completely normal.

I’m happy. I know I still have a road ahead of me, but I’m happy about it. I’m happy that everyday I’m closer to becoming the person I want to be and looking more how I want to be and learning more about myself and my condition and helping other people.

They also discussed other coping strategies such as ignoring, humor, learning to be confident and open with others, and surrounding yourself with positivity. Several participants discussed using religion and therapy as additional coping methods.

Resiliency

Although resiliency was not explicitly discussed, most participants commented or provided insight that exemplified resiliency traits. Participants described instances in which they overcame difficult situations and explained the importance of focusing on “more important issues” and being proud of their differences. Many participants were aware that their CFCs were out of their control and explained that you need to be “happy with yourself” in order to be a “strong [person].” As noted by one participant:

I told myself that there are two options I can do. I said I could either prove them right, or I could prove them wrong. I always say to myself when I go to the gym, I say I’m on a mission to prove them wrong…You can only control how you handle a situation, not other people and things you are given.

Mindfulness

There were varying degrees of awareness of mindfulness among the participants in both focus groups. Some participants were more experienced and had practiced mindfulness techniques (n = 4), while others had limited exposure to such practices (n = 10). Those who had prior experience learned from their parents and/or other family members. One adolescent reported that learning meditation or other mindfulness skills could be beneficial for her:

I feel like my mind doesn’t really wander enough. I feel like it should because this was really relaxing. I feel like I’m always so tense, so like I have to do this and I have to do this. I have to do this this this. I’m never taking some time, besides sleeping, to just like chill out.

Most participants found the in-session meditation helpful, but some were more uncomfortable.

Social support

Participants mentioned many different forms of social support and the majority felt as though they received some sort of social support. Different sources of social support included teachers/school staff, friends/peers, organizations within their communities, and parents. Parents were one of the most common sources of social support. One participant stated, “I think the biggest support you can ever get is from your parents.” This sentiment seemed to resonate with many other focus group members as multiple participants mentioned that their parents treated them “normal” and “encouraged them” to make friends and be themselves. The following 2 quotes exemplified how thankful and supported participants felt by their parents and family:

My parents have always been supportive, whenever I’m bullied or anything…They are very supportive, and there would be a lot of advice over the years about how to handle those situations.

I cannot thank my parents enough…For just trying to understand how I feel. Thank you for helping me in the middle of the night. Thank you. I’m surely blessed by all of these people in my life.

Participants also noted several sources of negative social interaction, including ex-romantic partners and other individuals with CFCs who had different attitudes. For example, one participant mentioned how she dated an individual with a CFC who would emotionally “dump” his problems onto her, and his negative self-esteem made it challenging to have a relationship with him or feel supported. Overall, social support was a major factor in participants’ happiness and resiliency.

Evaluation of a Proposed Future Therapeutic Intervention

Participants were highly supportive of the idea of a future intervention for this population. Quantitative results from the adapted CEQ displayed moderate to high satisfaction with the proposed intervention (mean = 32.1, SD = 10.0). Additionally, participants completed short-answer questions to describe their ideal intervention and preferred topics for the proposed intervention. The most common topics from their responses included stress (eg, “how to reduce stress”), coping (eg, “how to cope with anxiety and having a facial difference”), resiliency (eg, “how people like me overcome this challenge”), social support (eg, “I would want to learn other people’s experiences knowing I am not alone”), and bullying (eg, “how to handle being talked down to”). In their responses to the short-answer questions, participants also mentioned specific aspects of the focus group that they found helpful which could also be incorporated into a future intervention. These included social support (eg, “it was nice to talk about problems with others and learning different experiences”), coping (eg, “how to cope with being bullied” and “learning new techniques”), bullying (eg, “this focus group really helped people who have bullying”), and stress (eg, “how to handle stress”).

In addition, several participants mentioned aspects of the focus group they would not recommend for the intervention in their questionnaires, including the in-session meditation (eg, “I found the meditation to not be helpful and stress me out more”), role-playing (eg, “not accurate of real life”), and the lack of specificity (eg, “It didn’t deal with anything specific to my life”).

Lastly, responses from the CEQ revealed most participants would attend a future intervention. Doctor’s appointments, homework, and distance were potential attendance barriers identified by several participants. Two participants mentioned that they would not attend currently, but they might attend in the future depending on the topic (eg, “the topic would make me want to attend”). All participants preferred the future intervention be held on a weekend, and most participants preferred the intervention be in person (n = 7, 50%) and in a group setting (n = 11, 78.6%).

Overall, findings suggested that, although some participants would not currently attend an intervention, they would have appreciated it when they were younger or they might attend if they became distressed in the future. Most participants agreed that the intervention would be helpful for those who lack skills necessary to improve well-being. During the focus group, one participant said, “If I had this 7 years ago, I think I would probably be a better person.” Another person discussed the importance of opening up and groups to talk about difficulties:

I think this would be really good for anyone who may be struggling with anything, just to talk, even if it’s not this, just that focus groups in general for people no matter what their situation is, it’s just kind of nice to talk about things I guess.

The following quote by one of the participants accurately summarizes most participants’ views on the potential future intervention:

Personally, I don’t need it. I rather help someone else. That’s why I decided to come today because I thought maybe if I said something, it could help someone else, especially younger than me because when I was younger, I probably could have needed this a lot, and I didn’t get that, but now that I’m older I want to be that kind of person for someone else.

Discussion

The purpose of this study was to understand the psychosocial needs of adolescents with CFCs through focus groups and to use feedback to inform intervention development. Findings from this study partially supported the first hypothesis such that adolescents with CFCs displayed mean scores outside of normative ranges for lower mindfulness, higher perceived stress, and higher PTSD symptoms, with an area of strength seen in higher resiliency. For multiple aspects of QoL, self-esteem, and body image QoL, participants’ average scores were within the normative ranges. When compared to normative samples, adolescents with CFCs displayed higher perceived social support, but lower coping.

Several findings were consistent with previous research. Similar to other studies (Crerand et al., 2017; Feragen and Stock, 2017; Johns et al., 2018), adolescents with facial differences in the current study did not display deficits across several domains of psychological functioning as indicated by their levels of QoL, self-esteem, and body image QoL that fell within the normative ranges. Previously, Crerand et al. (2017) found no significant difference in body image QoL between adolescents with CFCs and their peers, and the current findings were similar given that participants’ scores fell well above the clinical cutoff. However, qualitative data revealed participants experienced bullying, inappropriate social responses, social stigma, and multiple surgeries, and given that these factors have been found to lower self-esteem and lead to negative self-perceptions (Kapp-Simon et al., 2005; Roberts and Shute, 2011; Crerand et al., 2017), our findings indicate that self-esteem and QoL among adolescents with CFCs may vary. Additionally, the sample mean was just above the clinical cutoff for resiliency, with the SD reflecting a portion of the sample below the cutoff, indicating that adolescents with CFCs displayed high resiliency. Participants’ high resiliency scores and multiple quotes from the qualitative data that exemplified resiliency traits were consistent with several prior studies that suggested those with CFCs have resiliency above the range reported by the general population (Meyerson, 2001; Strauss, 2001; Beaune et al., 2004; Locker et al., 2005; Feragen et al., 2009).

Given that CFCs have been described as lifetime stressors and “traumatic” conditions (Rumsey and Harcourt, 2007; Edwards et al., 2011; Bemmels et al., 2013), it was hypothesized that participants would have PTSD symptoms above the clinical cutoff. The sample mean was just above the clinical cutoff (50% were above this score) for meeting PTSD criteria; however, the SD was wide. PTSD symptoms may not be relevant to all adolescents with CFCs, and thus, referrals for individual support may be more appropriate than trauma-specific interventions. Nonetheless, these findings highlight the need for interventions that address aspects of enhancing QoL, while making sure to assess for self-esteem and PTSD symptoms within this population.

In contrast to prior research, the qualitative data revealed that adolescents with CFCs had many positive forms of social support. Furthermore, their social support scores were above the scores reported by adolescents without CFCs. Multiple studies have examined social functioning of adolescents with CFCs and found poorer psychosocial adjustment and social skills than their peers (Kapp-Simon et al., 2005; Topolski et al., 2005; Patrick et al., 2007; Feragen et al., 2009; Roberts and Shute, 2012). Although social dysfunction is not synonymous with low social support, studies have suggested poor psychosocial adjustment and social skills lead to fewer social supports and less social interactions (Kapp-Simon et al., 2005; Rumsey and Harcourt, 2007; Edwards et al., 2011). Despite some indicators of social struggles (eg, discussion of social stigma and bullying), participants reported strong social support. This may be explained by differing methodologies of prior studies (Kapp-Simon et al., 2005; Edwards et al., 2011), as the current study did not measure these constructs directly. Moreover, participants’ connections to other resources (eg, myFace, previous therapy, etc) may have impacted the results given that these resources could be providing additional social support. Future interventions should address how adolescents can utilize their support networks to cope with difficult social situations.

Participants’ mean scores were also just above the clinical cutoff for perceived stress, with the SD reflecting a portion of the sample below the cutoff, indicating that participants demonstrated moderate stress. Participants also displayed coping scores below the range of the normative data, suggesting that they felt they had moderate stress and also had limited abilities to effectively cope with that stress. However, participants noted various coping strategies to help with those stressors in their open-ended responses. Prior research has found that adolescents with CFCs have higher levels of coping compared to the general public (Locker et al., 2005; Rumsey and Harcourt, 2007; Feragen et al., 2009; Bemmels et al., 2013). The qualitative data revealed many different coping strategies and the specific need for these coping strategies to decrease stress, but it is possible these strategies are not sufficient, as shown by lower scores on the MOCS-A. Findings from Roberts and Shute (2012) suggest that more effective coping can increase the likelihood of positive adjustment. Therefore, these individuals would benefit from learning additional coping skills to enhance their ability to cope in difficult situations given their low scores. Future interventions should promote effective coping skills and stress reduction techniques.

Finally, findings suggested strong support for a proposed intervention. Both the qualitative and quantitative data suggested high feasibility and acceptability of a future program and provided insight into relevant topics to be included (eg, stress, coping, resiliency, social support, and bullying). Although the meditations and role-playing were not found to be as helpful, this was not consistent throughout all participants. Future interventions may want to consider adapting these activities to be more specific to the craniofacial population and offer choices of coping strategies. For example, future interventions could employ more relevant vignettes or videos involving social situations, open discussions on related real-life experiences, peer-to-peer interviews, or other alternative methods examining similar constructs. Similarly, the varied PTSD scores and conflicting views on mindfulness/meditations indicate the need for flexible interventions that address the needs of particular adolescents with CFCs.

Limitations and Future Directions

There are several limitations to this study. First, the sample was small and had similar demographics and characteristics. The majority of this sample was female, white/Caucasian, not Hispanic or Latino, and diagnosed with CL/P. In previous studies (Rosenberg et al., 2011; Bogart et al., 2012), most participants had higher parental incomes and were relatively well adjusted and socially adapted. Rosenberg et al. (2011) suggest that those who are more willing to attend these types of programs tend to come from more privileged households with more economic resources and opportunities. Therefore, the results from this study may not be representative. Future studies should recruit from larger, more heterogeneous craniofacial populations from multiple health centers. Moreover, interpreting mean scores of measures across studies may also be limited given the lack of statistical testing, different sample sizes and demographics, and inconsistent procedures across studies. However, it is important to identify ranges of scores in order to better understand the varying levels of constructs and symptomatology present at baseline in future interventions. Randomized controlled trials and longitudinal studies could help address these limitations. Second, as with most self-report measures, there is risk of subject bias when collecting data. Incorporating more diverse measures, as well as caregiver reports, could help address this limitation. Third, a limitation of qualitative research is that the data can be challenging to characterize and interpret; thus, it is difficult to determine whether the qualitative themes were specific to adolescents with CFCs or common to all adolescents. Fourth, several environmental factors (ie, parenting styles, type of schooling, comorbid diagnoses, etc) were not rigorously evaluated in this study, which could impact participants’ experiences and the results of the study. Although we did not specifically ask about environmental issues, several environmental factors came up during our discussions in the focus groups (ie, parents, schooling, social media, friends and social relations, etc). Based on the opinions of the independent coders, these environmental influences did not warrant their own thematic category, but rather seemed more appropriately placed within the other identified themes. Future studies should attempt to account for these differences. Fifth, the MOCS-A had not previously been tested with adolescents, and both the MOCS-A and MOS Social Support Survey lacked clinical interpretations, so it is unclear whether these were appropriate measures for this population. Finally, given that several participants mentioned that they would have preferred an intervention when they were younger, future focus groups may want to recruit younger patients to gauge their interests in a potential intervention.

Conclusions

The current study sought to address deficits in craniofacial research by explicating the psychosocial needs of adolescents with CFCs. Findings from this study will guide researchers in developing and refining a useful psychotherapeutic intervention for this population and have increased understanding of the varied psychological needs of adolescents with CFCs. Present findings could be indicative of the heterogeneity of symptoms among this population, which necessitates holistic, flexible intervention approaches. Moreover, adolescents in this study showed high levels of support for and interest in future interventions. The current study provides findings regarding interventions from the perspectives of adolescents with CFCs themselves, while utilizing mixed-methods to inform their development, and demonstrates the importance of creating interventions to target the specific psychosocial needs of adolescents with CFCs.

Supplemental Material

Supplemental Material, Riklin_CFC_FGs_CPCJ_Table_S1 - Examining the Psychosocial Needs of Adolescents With Craniofacial Conditions: A Mixed-Methods Approach

Supplemental Material, Riklin_CFC_FGs_CPCJ_Table_S1 for Examining the Psychosocial Needs of Adolescents With Craniofacial Conditions: A Mixed-Methods Approach by Eric Riklin, Dominique Calandrillo, Aileen Blitz, Dina Zuckerberg and Rachel A. Annunziato in The Cleft Palate-Craniofacial Journal

Footnotes

Declaration of Conflicting Interests

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

Funding

The author(s) received no financial support for the research, authorship, and/or publication of this article.

ORCID iD

Eric Riklin, MA

Supplemental Material

Supplemental material for this article is available online.

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