Psychological Distress in Patients with Pectus Excavatum as an Indication for Therapy

doi:10.4236/ijcm.2011.23050

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International Journal of Clinical Medicine, 2011, 2, 295-300

doi:10.4236/ijcm.2011.23050 Published Online July 2011 (http://www.SciRP.org/journal/ijcm)

295

Psychological Distress in Patients with Pectus

Excavatum as an Indication for Therapy

Susanne Habelt1, Stephanie Korn2*, Angelika Berger3, Jozef Bielek1

1Department of Pediatric Surgery, University Childrens’ Hospital Basle, Basle, Switzerland; 2Pulmonary Department, Mainz Univer-

sity Hospital, Mainz, Germany; 3Child and Adolescent Psychitric Department Baselland, Baselland, Switzerland.

Email: *[email protected]

Received February 6th, 2011; revised April 11th, 2011; accepted April 22nd, 2011.

ABSTRACT

Background: Adolescents with pectus excavatum (PE) are often affected by their body shape. The aim of our approach

was to quantify the patients’ individual psychological distress and to create a psychological indication for treatment.

Methods: 10 adolescents (8 male, median age 16 years, interquartile rang e 15 - 17 yrs.) with PE were examined at our

psychological department. Using standardized psychological tests, projective tests and interviews psychologists vali-

dated the patients’ individual psychological status. All patients were offered psychological therapy and correction of

the deformity. In addition, the children were follo wed-up by a telephone questionnaire (median fo llow-up after starting

therapy 12.8 months (5.9 - 18.0). Results: No patient had a relevant physiological limitation. The median follow-up

since presentation to our psychologists was 15.0 months (9.1 - 20.6). 8 patients (5 were operated, 2 used the vacuum

bell, 1 will undergo surgery) had distinct psychological limitations especially concerning the dimensions attractiveness,

self-esteem and somatisation. They demonstrated increased insecurity, anxiety and denegation of their body. Since all

patients were within puberty the psychological distress due to the PE has to be interpreted as disadvantageous for their

further development. 7 patients completed the follow-up questionnaire and reached a median score of 80.8% (76.4% -

86.8%), which indicates a good improvement in all patients. Conclusions: We conclude that the psychological indica-

tion for treatment is justified, since our results support this indication.

Keywords: Chest Wall Deformity, Pectus Excavatum, Psychological Distress, Surgery, Vacuum Bell

1. Introduction

Pectus excavatum (PE), or funnel chest, is the most

common congenital chest wall deformity occurring in

approximately one per thousand children, with a 4:1

male predominance [1]. Various surgical [2,3] and non-

surgical [4,5] techniques are described for repair of PE.

Although many objective criteria for evaluating chest

wall deformities and determining indications have been

reported such as the Haller Index [6,7], few studies have

examined the psychological problems associated with

PE [8].

In many cases, the degree of pectus deformity does

not warrant repair due to physiological limitations.

Therefore, treatment is often performed on a cosmetic

basis. This creates problems since therapy for cosmetic

reasons is usually not covered by insurance.

It is well known that children with PE are affected by

their body image, that they often experience embarrass-

ment, have low self-esteem and feelings of stigma [9].

These psychological criteria influence the patient’s life

deeply. In the past years, several studies examined the

quality of life of these children, which demonstrated that

the adolescents improve after therapy [10,11]. Neverthe-

less, until now repair of PE a psychological basis with-

out any physiological restrictions has been controversial

[8,11]. It is now recommended to extend the indication

for therapy to psychological factors [12].

With this as background the aim of our evaluation

was to perform an extended psychological status in order

to establish a psychological indication for treatment.

Therefore, psychologists quantified psychological char-

acteris- tics of adolescents suffering from PE, who had

no phy- siological limitations.

2. Materials and Methods

10 consecutive teenagers (8 male, median age at time of

psychological assessment 16 years (interquartile range 15

- 17 yrs.)) with PE were examined at our psychological

Psychological Distress in Patients with Pectus Excavatum as an Indication for Therapy

296

department in Basle, Switzerland as part of our routine

assessment prior to treatment. Our assessment includes

lung function tests, echocardiographic examinations and

computed tomographies of the chest. Sternum-vertebral

ratio indices are not obtained on a regular basis. In ac-

cordance with Krasopoulos et al. [10] we remain un-

aware of the significance of such indices as a parameter

for the individual distress.

Using special standardized psychological tests and

projective tests in combination with interviews with the

adolescents and their parents’ psychologists assessed the

patients’ individual psychological strain.

All children were offered surgical or non-surgical

treatment. The choice of therapy depended on the pa-

tient’s personal preference. Surgical correction was done

using the Nuss procedur [3]. In the case of non-surgical

therapy the patients used the vacuum chest wall lifter

first described by Schier et al. [5].

All children were followed-up at our outpatient clinic.

In addition, using a telephone questionnaire they were

asked about their social and psychological status and

their individual satisfaction after therapy.

2.1. Tests

For psychological examination several specific psycho-

logical tests were used. All tests were assessed by

Hogrefe, a German test agency (Göttingen, Germany).

Body appraisal inv en tory (FBeK):

The subjective body-experience is examined with a

combination of body and complaint centered question-

naires. It consists of 4 scales: insecurity/worrying, con-

cern of physical appearance, attractiveness/self-confi-

dence, and negative feelings about physical-sexual func-

tioning.

Body image inventory (FBK-20):

This test consists of 2 scales: negative body appraisal

as well as vitality and body-dynamic.

Manual for the self-esteem for children and youngsters

(ALS):

The manual for the self-esteem for children and ado-

lescents is a self-reporting inventory for classifying chil-

dren’s perception of themselves in various specific do-

mains of life: school, leisure time, family or children’s

home. These scales were constructed on the assumption

that an instrument with separate information of one’s

self-esteem in different domains would provide a more

differentiated picture than those instruments providing

only a single self-concept score.

Youth Self Report (YSR) and Child Behavior Checklist

(CBCL):

In the YSR the adolescents classify each position

themselves and rate for how true each item is. The CBCL

is rated by the parents and allows to cross check values

given by the children. Both tests use a three-point re-

sponse scale. The YSR and CBCL scoring profiles pro-

vide scores for two competence scales (activity scale:

measuring the child’s participation in hobbies, games,

sports, jobs, chores, friendship; social scale: measuring

withdrawn, somatic complaints, anxiety and depression,

social problems, thought problems, attention problems,

aggressive behaviour, and delinquent behaviours), for a

total competence scale and for the eight syndrome scales

that form three sub-dimensions. The subdimensions are

referred to as ‘internalizing,’ ‘externalizing’ and ‘neither

internalizing nor externalizing’.

Overall behavioural and emotional functioning are

measured by the total problem scale. In addition, the

CBCL consists of a third scale regarding school achieve-

ment.

Symptom Checklist-90-R (SCL-90-R):

The Symptom Checklist-90-R is used as an instrument

to evaluate a broad range of psychological problems and

symptoms of psychopathology. The test describes 9 pri-

mary symptom dimensions (somatisation, obsessive-

compulsiveness, interpersonal sensitivity, depression,

anxiety, hostility, phobic anxiety, paranoid ideation,

psychoticism) and is designed to provide an overview of

a patient’s symptoms and their intensity at a specific

point of time. In addition, the test provides three global

indices: Global Severity Index (GSI): designed to meas-

ure the overall psychological distress, it can be used as a

summary of the test; Positive Symptom Distress Index

(PSDI): designed to measure the intensity of symptoms

and Positive Symptom Total (PST): reports the number

of self-reported symptoms.

Follow-up questionnaire:

This questionnaire consisted of questions of the single

step questionnaire by Krasopoulos et al. [10], of the

Nuss-Questionnaire as published by Lawson et al. [13]

and of questions chosen by a psychologist of our psy-

chological department. The questionnaire consisted of 9

questions (Table 1) and the scoring ranges from 1 to 5

points per question (1 = much worse (--), 2 = worse (-), 3

= about the same (0), 4 = better (+), 5 = much better (++))

resulting in a minimum score of 9 and a maximum score

of 45. A total score of >27, accordingly >60% means

improvement, whereas ≤60% means no change or wors-

ening.

2.2. Statistical Analysis

Scoring profiles of the psychological tests provide raw

scores and percentiles. The interpretive reports are based

on age-appropriate non-patient norm groups. Using the

percentiles data description is primarily based on medi-

ans and quartiles (interquartile range) for continuous

Psychological Distress in Patients with Pectus Excavatum as an Indication for Therapy

297

Table 1. Follow-up questionnaire.

Much worse

(--)

Somewhat worse

(-)

About the same

(0)

Somewhat better

(+)

Much better

(++)

1 Overall feeling/satisfaction after therapy 1 2 3 4 5

Difficulties (sports without shirt, e.g. swimming,

subjective feeling of shortness of breath) after

therapy

1 2 3 4 5

3 Health in general after therapy 1 2 3 4 5

4 Exercise capacity after therapy 1 2 3 4 5

5 Satisfaction with overall appearance of the chest

after therapy 1 2 3 4 5

6 Impact to social life 1 2 3 4 5

7 Self-consciousness because of chest/self-esteem 1 2 3 4 5

Never

(--)

(-)

Neutral

(0)

Yes

(+)

Absolutely

(++)

8 Therapy was considered to be good for me 1 2 3 4 5

9 Going back, would you have the elected therapy

again? 1 2 3 4 5

endpoints and on frequencies for binary endpoints. Every

psychological test was analyzed on the basis of its

sub-dimensions, its global results and on its special crite-

ria. In addition, all tests combined with the projective

tests and the interviews were used to assess the individu-

als’ psychological distress as a whole.

3. Results

Eight male and two female patients with a median age of

16 years (interquartile range 15 - 17 years) were seen at

our Department of Pediatric Surgery for evaluation of

their pectus excavatum (PE). Prior to treatment they were

examined according to our routine assessment program.

None of the patients had a physiological limitation,

echocardiographic examination was defined as normal in

all patients, lung function tests were normal in 8 patients.

One patient demonstrated a limited restrictive defect and

one patient was known to have asthma (Table 2). Addi-

tionally, the patients were assessed to our psychological

department and a psychological status was obtained. The

median follow-up since presenting at the psychologist is

15.0 months (interquartile range 9.1 - 21.4 months); the

psychological status was taken at the same time or within

a few days after medical examination and at a median of

17.8 weeks (interquartile range 2.4 - 27.0 weeks) before

Table 2. Lung function tests preoperatively.

n Median

(interquartile range)

Minimum;

Maximum

Vital capacity

(% predicted) 10 84.1 (77.7- 91.6) 69.0; 101.3

FEV1 (% predicted) 10 90.2 (80.9 - 92.8) 70.0; 99.4

Residual volume (%) 10 109.9 (88.3 - 143.4) 54.9; 192.0

Total lung capacity (%) 10 92.9 (81.0 - 100.6) 74.6; 108.6

Displayed are median scores, interquartile ranges, minimum and maximum

of the lung function parameters.

starting therapy.

The results of the single tests and their sub-dimensions

are demonstrated in Table 3. In addition, we specified

how many patients were at risk in a sub-dimension and in

the specific test. Overall, 8 patients had psychological

limitations especially concerning the dimensions attrac-

tiveness, self-esteem and somatisation. Eight children

shied away from presenting their body for instance when

going swimming or doing sports. This resulted in in-

creased insecurity, anxiety and denegation of the own

body. Four patients complained about breathlessness

although they did not have any pulmonary limitation. All

patients were offered surgical treatment, since 2005 sur-

gical or non-surgical treatment. Of the 8 patients with

psychological distress 6 elected surgery (one will un-

dergo surgery in two months), the remaining 2 decided to

use the vacuum chest wall lifter. Of the 2 patients with-

out psychological limitation one did not desire any ther-

apy and the other one elected therapy with the vacuum

bell for a better cosmetic result.

All patients were followed up at our outpatient clinic

and were asked to complete the telephone questionnaire

despite the one who did not desire therapy and the one

who has not undergone surgery by now. 7 patients (of 8

interview patients) completed the questionnaire with a

median follow-up of 13 months (interquartile range 6 -

18 months) after starting therapy; one patient could not

be asked due to being in Africa for one year. The teenag-

ers reached a median score of 80.8% (interquartile range

76.4% - 86.8%). Since a total score of >60% was defined

as improvement, all patients demonstrated good im-

provement after therapy and stated satisfaction with the

overall result of the treatment. All children improved

concerning earlier difficulties, attractiveness, self-esteem

and social life and recommended their type of therapy.

Psychological Distress in Patients with Pectus Excavatum as an Indication for Therapy

298

Table 3. Results of the different psyc hol ogical tests including their subdimensions.

n Median

(interquartile range) Min.; Max. at risk (n)

Body appraisal inventory (FBeK)

Insecurity/worrying

(scores above 84% are classified as at risk) 9 49.0 (18.0 - 86.5) 6.0; 97.0 2

Concern with physical appearance

(scores below 16% are classified as at risk) 9 57.0 (25.0 - 62.5) 15.0; 98.0 1

Attractiveness/self-confidence

(scores below 16% are classified as at risk) 9 14.0 (1.5 - 60.0) 1.0; 100.0 5

Physical-sexual functioning

(scores above 84 are classified as at risk) 7 65.0 (22.0 - 69.0) 22.0; 93.0 1

Total 5

Body image inventory (FBK-20)

Negative body appraisal

(scores above 84% are classified as at risk) 9 60.0 (8.0 - 87.5) 1.0; 99.0 4

Vitality and body dynamic

(scores below 16% are classified as at risk) 9 40.0 (15.0 - 47.5) 4.0; 90.0 2

Total 6

Manual for the self-esteem for children and youngster (ALS)

(scores of 11% - 25% are classified as at risk)

School 8 0 1

Leisure time 8 1 1

Family/children’s home 8 1 1

Total 3

Youth Self Report (YSR)

(scores above 98% are classified as at risk; activity and social scale: scores below 2% are classified as at risk)

Withdrawn 5 84.0 (50.0 - 88.0) 50.0; 92.0 0

Somatic complaints 5 92.0 (58.5 - 98.3) 50.0; 98.5 2

Anxiety/depression 5 50.0 (50.0 - 84.2) 50.0; 98.3 1

Social problems 5 50.0 (50.0 - 65.0) 50.0; 80.0 0

Thought problems 5 50.0 (50.0 - 83.0) 50.0; 93.0 0

Attention problems 5 50.0 (50.0 - 75.0) 50.0; 100.0 1

Delinquent behaviours 5 50.0 (50.0 - 83.5) 50.0; 90.0 0

Aggressive behaviour 5 50.0 (50.0 - 85.0) 50.0; 86.0 0

Activity scale 5 69.0 (63.5 - 69.0) 58.0; 69.0 0

Social scale 5 69.0 (57.0 - 69.0) 46.0; 69.0 0

Total 2

Child Behaviour Checklist (CBCL)

(scores above 98% are classified as at risk; activity and social scale: scores below 2% are classified as at risk)

Withdrawn 4 59.5 (50.0 - 81.8) 50.0; 86.0 0

Somatic complaints 4 68.0 (54.5 - 68.8) 50.0; 69.0 0

Anxiety/depression 4 61.0 (50.0 - 86.3) 50.0; 91.0 0

Social problems 4 68.0 (54.5 - 68.8) 50.0; 69.0 0

Thought problems 4 55.0 (50.0 - 86.3) 50.0; 95.0 0

Attention problems 4 60.0 (52.5 - 81.0) 50.0; 88.0 0

Delinquent behaviours 4 50.0 (50.0 - 57.5) 50.0; 60.0 0

Aggressive behaviour 4 59.0 (52.0 - 66.8) 50.0; 69.0 0

Activity scale 4 69.0 (45.0 - 69.0) 37.0; 69.0 0

Social scale 4 69.0 (36.0 - 69.0) 25.0; 69.0 0

School scale 4 60.5 (49.8 - 69.0) 49.0; 69.0 0

Total 0

Symptom Checklist 90-R (SCL-90-R)

(scores above 84% are classified as at risk indicating psychological problems)

Somatisation 1096.0 (6.0 - 96.9) 3.0; 99.0 5

Obsessive-compulsive 1060.5 (12.8 - 98.1) 5.0; 99.0 5

Interpersonal sensitivity 1052.5 (5.0 - 94.3) 5.0; 100.0 4

Depression 1044.0 (7.0 - 98.1) 7.0; 100.0 4

Anxiety 1061.0 (7.0 - 98.6) 7.0; 100.0 4

Hostility 1025.5 (20.8 - 93.3) 7.0; 94.0 4

Psychological Distress in Patients with Pectus Excavatum as an Indication for Therapy299

Phobic anxiety 1073.5 (55.8 - 94.8) 22.0; 100.0 4

Paranoid ideation 1065.0 (11.0 - 98.1) 11.0; 100.0 4

Psychoticism 1043.5 (11.0 - 94.4) 11.0; 99.0 3

GSI 1057.5 (10.5 - 99.0) 2.0; 100.0 4

PSDI 1056.0 (9.0 - 98.0) 9.0; 99.0 4

PST 1054.0 (2.8 - 96.5) 2.0; 99.0 4

Total 6

Total 10 8

Displayed are median scores, interquartile ranges, minimum and maximum of the several psychological tests with sub-dimensions reached by the patients. In

addition, number of patients with at risk findings, indicating psychological problems are shown. Total: number of patients in whom the test was classified as at

risk (in one or more sub-dimensions).

4. Discussion

Today, the indications for treatment of PE are mainly

based on physiological symptoms or cosmetic factors

[12]. Indications based on psychological or psycho-social

stress are frequently discussed [8,12] and are part of

questionnaires that assess the patients’ preoperative con-

dition [8,10].

Therefore, we picked-up the suggestion made by Suita

et al. [12] and Ohno et al. [8] to extend the indications

for treatment, especially for surgery, in regard to the

patients’ individual psychological stress.

We described the way of establishing a psychological

indication for therapy of PE. To date, there has been in-

creasing research on the psychological aspects of pectus

deformities or other disfiguring conditions in general and

medical procedures used to correct them [14,15]. The

psychological importance of the body image including

the notion that medical treatment can improve quality of

life has been described in some detail and there are ques-

tionnaires to assess the preoperative status of children

with PE [10,11]. Nevertheless, to perform an accurate

and extensive psychological assessment it is important to

identify the children’s individual psychological strain

including various dimensions.

It is well known that patients with PE often experience

embarrassment and shame over the perceived differences

in their physical appearance and that this can lead to

lowered self-concept, feelings of inferiority, depression,

shyness and social anxiety [16]. This was found in the

majority (8/10) of our children. The results of our ex-

amination demonstrated that the handicaps of PE influ-

ence all areas of life. Defensive camouflaging with poor

posture and folded arms, restrictions in lifestyle includ-

ing avoidance of swimming, sports, hugging and intimate

relationships were described by our patients. Particularly,

since all patients hit puberty the psychological strain due

to the PE has to be interpreted as disadvantageous for

their further development.

After psychological assessment our patients were of-

fered surgical and non-surgical treatment. All adolescents

with psychological problems decided to be treated. One

patient wanted to be treated with the vacuum bell some

time after presentation at the psychological department

due to a better cosmetic result although he did not feel

limited. According to the study by Ohno et al. [8] not all

distressed patients wanted surgery, 2 patients wanted to

try therapy with the vacuum bell, which is understand-

able since it is a less invasive therapeutic option produc-

ing increased interest with the patients during the last

year [5].

It is well known that medical therapy improves quality

of life as described by Roberts et al. using the QOL out-

lined by Keith and Shacklock [11]. Their results indicate

an improvement in each of the four areas satisfaction,

social belonging, well-being and empowerment/control.

In accordance, Lawson et al. created a Pectus Excavatum

Evaluation Questionnaire and demonstrated a positive

impact on the well-being of children after surgical repair

[13]. Since there are several studies which confirm im-

provement after correction of the chest deformity we

only used a short questionnaire to ask the patients about

their individual improvement after therapy. Therefore,

we modified the questionnaire of Krasopoulos et al. [10]

and Lawson et al. [13] to especially evaluate the factors

attractiveness, body image, social improvement and sat-

isfaction in general. Nearly all children reported im-

proved endurance and participation in physical education

activities after therapy. We reason this with the better

quality of life and better satisfaction with their physical

appearance, not due to any physiological improvement

since the children did not have any physical limitation

prior to treatment.

A limitation of our study is the small number of pa-

tients. All consecutive patients with pectus excavatum

were included to avoid selection bias. However, it cannot

be excluded that the results of the present study may

slightly change if more patients over a longer study pe-

riod would be included. However, from previous experi-

ence we strongly feel that the results of the present study

are representative for a large referral center in a devel-

oped county such as Switzerland.

Psychological Distress in Patients with Pectus Excavatum as an Indication for Therapy

300

Suita et al. comment that the precise indications for

surgical repair of PE is still open [12]. In our opinion, the

definition of the indication should depend on physical

and psychological characteristics in each case. In the

course of this, not only somatic data, but also psycho-

logical factors should be considered. We think psycho-

logical aspects should not only be taken into considera-

tion but the children with PE should be presented to a

psychologist. We recommend including psychological

distress to the list of indications for therapy as an inde-

pendent factor. Nevertheless, further studies with more

patients will be needed to underline our findings.

5. Conclusions

In summary, most children with PE suffered from psy-

chological strain and all patients improved after therapy.

We therefore recommend working together with psy-

chologists to extend the indications for therapy of PE to

psychological problems. The individual psychological

limitation can be assessed by psychologists using special

medical tests, projective tests and interviews with the

patients and their parents. Thus, it is possible to gain in-

formation in several dimensions especially attractiveness,

self-esteem and social life which affect the patients in all

areas of life. We conclude that the psychological indica-

tion for treatment is justified, since our results support

this indication.

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