Assessment of psychosocial functioning and its risk factors in children with pectus excavatum

Pectus excavatum (PE) is the most common chest wall malformation and one of the most frequent major congenital anomalies. It approximately occurs in up to 1 in 300 to 1 in 1000 births, with a 4:1 male predominance [1, 2]. In patients with PE, the sternum and adjacent chest wall are displaced posteriorly toward the spine, creating a depression of the anterior chest wall (Figure 1). Clinically, in many patients with PE, the deformity does not cause significant physiological dysfunction. However, the cosmetic disfigurement can cause a serious loss of self-esteem and affect social behaviour. Precisely when the patient is establishing an independent identity, choosing a trade, or beginning involvement with the opposite sex, he or she is afflicted with a deformity that reduces his or her capacity to do those things.

Yet, despite well documented [3‐9] importance of surgery to improve psychological distress and self-esteem in patients with PE, the psychosocial status has not been systematically studied in patients who are not currently undergoing surgical treatment. Additionally, questions exist as to whether clinical features are associated with psychosocial problems in PE patients. To address these issues, the current study examined the emotional and behavioural problems of PE patients by using Chinese Mandarin version of the CBCL, and investigated the risk factors of psychosocial problems by using univariate and multivariate analysis. It was anticipated that children in the control group would report lower frequency of psychosocial difficulties than children with PE. We believe that a better understanding of the features associated with psychosocial problems might improve assessment of emotional risk and assist in the development of intervention and prevention strategies.

We studied the psychosocial functioning of 337 children aged 6 to 16 years old who were born in China. The subjects included not only the children who would choose the surgery but also the children who would not. Our findings demonstrated that 74.8% of children with PE discovered they were different around the age of 4 to 6. This is when name-calling and teasing begins, and the children learn what it means to be different. Their first problem tended to be not an organic but a psychosocial one, caused by self-awareness of the deformity and/or by being teased by others. Many children, especially some older children want the deformity corrected as soon as possible.

As identified by Lavigne et al [18], there is wide variation in the psychosocial functioning of children with a chronic illness. In this study, assessment of psychosocial functioning was achieved by CBCL. We found that children with PE had significantly more emotional and social problems as reported by their parents on standardized question, in comparison with children in the general population. This result is comparable with previous reports of PE-induced deterioration of psychosocial functioning across a wide age range [6, 19]. Meanwhile, we found that children with PE did not have more behavioural problems than control group. This finding is also in line with studies of psychosocial functioning in children with PE [8, 19], but contradictory to studies on children with congenital microtia or hypospadias [20, 21]. We presume that this may be attributed to the different diseases have different risk factors, which have different relative impacts on psychosocial functioning [18].

However, one limitation of the study was that the study sample was entirely composed of patients seeking medical evaluation or treatment for PE and cannot be considered representative of the general population of PE subjects. In this respect, poor psychosocial functioning could be a motivation for referral and higher scores are usually observed in clinical-based samples when compared with population-based surveys. Another limitation of the study is that there had no reference values for externalizing problems and internalizing problems in our community [12, 13]. For this reason, we can not present the prevalence of psychosocial problems which are based on these two scales. Although the CBCL can be complemented by the Youth Self-Report form (YSR), the YSR is only constructed for children aged 11 years and older, and thus would not be suitable for use in the current study focused on children from age 6. Furthermore, the information on child emotional and behaviroal problems was obtained by questionnaires, and not by semi-structured clinical interviews conducted by clinical professionals. Although the patients who had higher scores on CBCL scales were recommended for further assessment in psychiatric outpatient department, identification of children in control group is typically impossible due to ethical reasons for protecting anonymity.

On the other hand, this study provided one of very few representative samples of PE patients. Information from a standardized behavioural checklist provided a wealth of information about the psychosocial status of children with PE. Moreover, we compared the patients with normal children matched for sex and age, and all subjects resided in the same health care catchment areas and assessment were carried out in closely related time periods in both groups. Therefore, the study of these children could provide a more accurate picture of PE individuals referring to clinical practices, and provide relevant clues for treatment programs. We, like others, believe that poor body image and impaired psychosocial functioning should be an enormously important concern for surgical repaired and/or psychosocial intervention [8, 22, 23].

This study showed a tendency for the prevalence of psychosocial difficulties to increase with age in patients who have not had surgery. It would seem to reflect the increasing burden of psychosocial trials which individuals face as they grow older. In other words, if PE patients do not have psychotherapeutic help or surgical repair, if they are left alone with their difficulties, psychosocial problems may increase with age. From the psychological point of view, the early surgery is ideal. Additionally, mounting evidence has emerged to suggest that many patients who do not undergo repair of severe PE deformities in childhood will experience worsening symptom in their adult lives [24, 25]. However, there is debate in the literature regarding the best age for repair. Some authors argued that repair at too early of an age may result in improper growth of the chest wall and other complications, so the surgery should be performed during the stage of teenage (close to the age of skeletal maturity) when the operation for correction of PE can be readily accomplished [26]. In contrast, a vast amount of research has shown that continuous technical refinements have significantly decreased the complication rates and postoperative morbidity, and Nuss procedure has been demonstrated to be effective for quality repair of PE patients aged from 3 to 50 years [7, 27‐31].

Within the study of visible or invisible (i.e.: normally hidden by clothing) difference or deformity, the presumption that more significant pathology is associated with greater psychological distress is strongly held. It is reasonable to expect that a person with extensive scarring, for example, may experience greater difficulty than a similar person with fewer scars [32, 33]. In our study, univariate analysis showed that the severity of PE was significant associated with psychosocial adjustment. Multivariate logistic regression analysis also identified it as a risk factor for psychosocial adjustment. This is a fascinating finding, providing very strong support for the inclusion of objective measures of appearance in studies exploring the psychosocial impact of PE, particularly where this includes an evaluation of treatment. The result is consistent with finding from Ohno et al.'s study [16] which included a group of PE patients who have not undergone the surgery. However, this finding stands into contrast with another finding that there was no significant relationship between the severity of PE and mental health [8]. We presumed that contradictory results may be attributed to the following reasons: first, in previous study, patients who did not undergo surgery were excluded from the psychosocial study. In fact, patients with mild/moderate deformations are associated with small loss of function and little focus on the appearance related aspects of the PE, their parents may be reluctant to take the risk of surgery unless the symptoms seem to be getting worse. This may lead to the reduction in psychosocial morbidity of the patients with mild/moderate PE in clinical research. Second, the psychometric testing tools in previous study were distinctly different from ours: in previous study, parents were interviewed by the Pectus Excavatum Evaluation Questionnaire (PEEQ). However, the PEEQ was developed not for the detection of psychosocial problems but mainly for the assessment of impact of surgical repair [4, 5].

Teasing is seen as a negative and repetitive action committed by one or more person and is associated with a host of negative consequences for children. The results of current study indicate that approximately 22.8% of patients had been teased about their PE by others, mainly their peers. We find that children with PE report having been teased less frequently than children with facial anomalies [34]. This may due to the fact that the upper part of the body is expected to be covered with clothes. Furthermore, many children with PE tried to avoid exposing their chest in public places because of the fear of being teased, and 'an unwillingness to be seen without a shirt while swimming and participating in sport or social activities' is the most frequently quoted complaint [22]. Unfortunately, researchers have found that the experience of being teased and the fear of being teased can result in anxiety, emotional distress, and difficulties with social integration [14, 35, 36]. In this study, teasing was associated with significantly higher prevalence of psychosocial difficulties in patients. This finding is not surprising given that our results indicate teasing history is greater for PE children with psychosocial problems. Surgeons and pediatricians need to be aware that psychosocial functioning of children with PE might be impaired by teasing remarks from peers.

In summary, this study examines a relatively large sample of patients with mild to very severe PE. The result shows that children with PE experience significantly decreased psychosocial functioning in emotional and social domains of the CBCL. Moreover, we find that age, severity of malformation, and being teased about PE has significant effects on psychosocial problems. Thus, a better understanding of the influence of PE on psychosocial functioning, including the identification of patients most vulnerable to psychosocial impairment, is acquired. This improved understanding could enable the application of strategies in both clinical practice and public health that seek to prevent further psychosocial problems associated with PE patients. Clinicians involved in care for patients with PE should be aware of the potential factors associated with psychosocial functioning. Studies looking at psychological intervention to ameliorate psychosocial problems in these patients are also warranted.