Prevalence Of Chest Wall Deformity In Turkish Population

Prevalence of Chest Wall Deformity

Abstract

Background

Chest wall deformities are seen in the ratio of 1/400-1/1000 in general population. It is most common among males.Prevalence Of Chest Wall Deformity In Turkish Population.  It develops from anterior protrusion or posterior depression of sternum due to abnormal development of costal cartilage.

Aim

In this study, it was aimed to detect the children who have chest wall deformity, the prevalence of deformity, the factors affecting the psychological and physical disorders caused by the deformity, in Mamak district of Ankara province.

Study Design

Cross-sectional Study

Methods

The study was conducted with 14108 girls and boys aged between 11-14 years, who were attending in class 5-8 in Mamak district of Ankara province between October 2014 and March 2015. A questionnaire form composed of 2 parts of questions and physical examination findings was filled out for the children who were detected to have chest wall deformity.

Results

Of 14108 students in our study, the mean age of the children was 12.53±1.11 (median 12.54, 11-14 years) and chest wall deformity was detected in 199 (1.41%) students. Male/female ratio was 2.16 and pectus carinatum/pectus excavatum ratio was 1.59. According to multivariate logistic regression analysis table, physical disturbance ratio was found statistically significantly higher in children in age group 11 ( p<0.011), in children who were aware of the deformity (p<0.006) and psychological disturbance ratio was found statistically significantly higher in girls (p<0.015), in presence of family history ( p<0.012).

Conclusions

In this study conducted in a large population, CWD was found more prevalent in boys (1.41%) and PC was found as the most common deformity type in our country, on the contrary to literature. While age group 11 and deformity awareness were effective on physical disturbance in CWD, family history, and female gender were effective on a psychological disturbance. Prevalence Of Chest Wall Deformity In Turkish Population.

Introduction

Chest wall deformities are seen in the ratio of 1/400-1/1000 in general population. It is most common among males. It develops from anterior protrusion or posterior depression of sternum due to abnormal development of costal cartilage (1). Pectus excavatum (PE) is the most common chest deformity and most common in males (2). Pectus carinatum (PC) is seen less than pectus excavatum in general population (3). Poland syndrome is another chest wall deformity seen in the ratio of 1/30 000 (4,5).

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In this study, it was aimed to detect the children who have chest wall deformity, the prevalence of deformity, the factors affecting the psychological and physical disorders caused by the deformity, early diagnosis of these psychological and physical disorders and take the required measures in Mamak district of Ankara province.

Material and Methods

Approval was obtained from National Education Directorate of Ankara province (date/number: 14.11.2014/5317227) and Clinical Research Ethics Committee of Ankara Numune Research and Training Hospital (date/number: 01.10.2014/E-14-306). The study was conducted with 14108 girls and boys aged between 11-14 years, who were attending in class 5-8 in Mamak district of Ankara province between October 2014 and March 2015. Seventeen schools were randomly selected among the intermediate schools in Mamak. Written informed consent was obtained from the families one day before visiting the school. The students whose families did not agree to participation were excluded from the study. Male and female students were examined separately in single cabins top clothes so as to be completely removed, their anterior and posterior chest walls were inspected. A questionnaire form composed of 2 parts of questions and physical examination findings was filled out for the children who were detected to have chest wall deformity. While question part of the questionnaire was filled out by the children, physical examination part was filled out by the researchers. The first part of the questionnaire form included names of the students, whether they knew about chest wall deformity (CWD), whether they were aware of CWD, family history of CWD, in whom if present, the most common physical disturbance they have ever experienced (chest pain, dyspnea, exertional dyspnea, palpitation or no disturbance) and whether they felt any psychological disturbance due to this deformity. Physical examination was done in the second part of the questionnaire. Weight and height were measured, presence of murmur was examined on cardiac auscultation, scoliosis was examined on inspection; in the students who were detected to have pectus excavatum deformity, deformity severity was detected with anthropometric index calculation by the proportion of sternal deformity depth to the largest diameter of chest wall; deformity type and deformity degree (mild, moderate, severe), subtype of pectus carinatum (chondromanubrial, chondrogladial symmetrical, chondrogladial asymmetrical and mixed type) were detected on inspection (6).Prevalence Of Chest Wall Deformity In Turkish Population.  Subjective deformity degree detection on inspection was done by the same physician for all students.

Body mass index (BMI) (kg/m2) was calculated using weight and height. BMI percentiles according to age and gender were calculated using childhood and adolescence (2-20 years) percentile curves of Centers for Disease Control (CDC 2000) (7, 8). The student was accepted as thin if BMI was below 5, normal if BMI was between 5-84, overweight if BMI was between 85-94, obese if BMI was ≥ 95.

The students who were detected to have CWD were referred to the hospital for further investigation.

Statistical analysis

The sample was calculated before starting the research. Chest deformity prevalence was found as 1.03% in the study of Rajabi-Mashhadi et al. (9). Based on this prevalence value, the power of the test was found as 80% with a ±20% deviation and the minimum was found as 14000 children when type 1 error was taken as 0.05. These 14000 children were distributed proportionally according to age and gender (R3.0.1 open source program). Mean unit size was calculated as 800 students considering the number of children at schools and 17 schools was seen to be enough and 3 schools were determined as a replacement. All of the students aged between 11-14 years were analyzed.

Statistical analysis was done using SPSS for Windows 18.0 (SPSS Inc, Chicago, IL, USA) package program. Categorical data were calculated with Fisher’s exact test, Pearson chi-square test, and Yates correctional chi-square test. Logistic regression analysis was used for analysis of the variables which gave significant results in univariate analysis and consistent with the literature. A p <0.05 was accepted as statistically significant.

Results

Of 14108 students in our study, 6828 (48.4%) were girls and 7280 (51.6%) were boys. Mean age of the children was 12.53±1.11 (median 12.54, 11-14 years). Mean age of girls was 12.53±1.11(median 12.54, 11-14 years). Mean age of boys was 12.53±1.11(median 12.54, 11-14 years). There were 1621(23.7%) girls and 1726(23.7%) boys in age group 11; there were 1669 (24.4%) girls and 1808 (24.8%) boys in age group 12; there were 1869 (27.4%) girls and 1912 (25.2%) boys in age group 13; there were 1669 (24.4%) girls and 1834 (25.2%) boys in age group 14. There was not a difference between age groups with regard to gender (p=0.452). CWD was detected in 199 (1.41%) students. Of them, 63 (31.7%) were girls and 136 (68%) were boys. While prevalence of deformity was 0.45% (n=63) in girls, this ratio was 1.96% (n=136) for boys (p<0.0001) (Table 1). Male/female ratio was 2.16 in CWD. Mean age of the students in whom deformity was detected was
12.78±1.98 (median 13 years, 11-14 years). The deformity was most frequent in 14 age group with 68(34.2%) students and demographic characteristics of the patients are given in Table 1 (Table 1). Among all students, there were 121 (0.86%) PC, 76 (0.54%) PE, 1 (0.007) Poland syndrome and 1 (0.007%) rib anomaly. PC/PE ratio was 1.59. Chondrogladial symmetrical type was the most common PC subtype with the ratio of 49 (40.5%) (Table 2). On inspection, deformity was mild in 123 (61.8%), moderate in 69 (34.7) and severe in 7 (3.5%). Prevalence Of Chest Wall Deformity In Turkish Population. In 76 students with PE, deformity was mild in 42 (55.3%), moderate in 30 (39.5%) and severe in 4 (5.3%). Among 76 students with PE, anthropometric index was 0.12 and above in 22 (28.9%) and below 0.12 in 54 (71.1%). While scoliosis was detected on inspection in 10 (5.03%) of children who were detected to have CWD, it was not detected in 189 (94.97%). CWD degree of scoliosis was mild in 1 (10%), moderate in 8 (80%) and severe in 1 (10%). Of the children with CWD, 48 (24.1%) stated that they knew about the deformity , 67 (33.7%) stated that they were aware of the deformity. While there was the family history of CWD in 15 (7.6%), 136 (68.3%) stated that they did not know about the family history of CWD. CWD was reported in the father most commonly (n:7, 46.7%) followed by mother in 3 (20%), sibling in 2 (13.3%), grandfather in 2 (13.3%) and cousin in 1 (6.7%). The most commonly reported CWD-related physical disturbance was dyspnea in 20 (10.1%), chest pain in 10 (5%), exertional dyspnea in 6 (3%), palpitation in 6 (3%). Nine (4.52%) children with CWD reported psychological disturbance due to CWD. This ratio was found as 11.1% in girls and 1.5% in boys and the difference was found statistically significant (p=0.005).

Prevalence of awareness of deformity was found statistically significantly higher in children with family history of CWD (p=0.001).

According to uni-variate logistic regression analysis table between physical disturbance and gender, age groups, deformity degree, deformity awareness, family history, having information about CWD and BMI percentile curves, physical disturbance was found statistically significantly higher in age group 11 ( crude OR, 95% CI, p<0.015), mild deformity (crude OR, 95% CI, p<0.004) and moderate deformity (crude OR, 95% CI, p<0.017) group and the ones who are aware of the deformity (crude OR, %95 CI, p<0.001). According to multivariate logistic regression analysis table, physical disturbance ratio was found statistically significantly higher in children in age group 11 [16.011 (1.890-135.614) adjusted OR, 95% CI, p<0.011], in children who were aware of the deformity [0.307(0.132-0.713) adjusted OR, %95 CI, p<0.006] (Table 3).

According to uni-variate logistic regression analysis between psychological disturbance and gender, age groups, deformity degree, deformity awareness, family history, having an information about CWD and deformity type, psychological disturbance was found statistically significantly higher in girls (crude OR, 95% CI, p<0.002), moderate deformity group (crude OR, %95 CI, p<0.035), in children who were aware of the deformity (crude OR, %95 CI, p<0.0003), in children with family history (crude OR, %95 CI, p<0.0001), who knew about CWD (crude OR, %95 CI, p<0.0001). According to multivariate logistic regression analysis table, psychological disturbance ratio was found statistically significantly higher in girls [15.440(1.684-141.586), adjusted OR, %95 CI, p<0.015], in presence of family history [18.656 (1.917-181.605), adjusted OR, %95 CI, p<0.012] (Table 4). Prevalence Of Chest Wall Deformity In Turkish Population.

Of children with PE, 36.8% (n=28) and of children with PC, 31.4% (n=38) were found to be aware of the deformity. A statistically significant relationship could not be detected between deformity type and awareness of deformity (p=0.431).

A statistically significant relationship was detected between deformity degree and awareness of deformity (p=0.0001). Awareness increases as deformity degree increases.

A statistically significant relationship was not detected between deformity degree and BMI according to age percentile curves (p=0.321).

A statistically significant relationship was not detected between deformity type and BMI according to age percentile curves (p=0.550).

Discussion

Congenital chest deformity ratios vary in general population and among communities. CWD prevalence was reported as 1% in general population (10). CWD prevalence varies between 1% and 1.95% in the literature (9,11). CWD prevalence was reported higher in the white race in the study of Westphal et al. (11). This ratio was found as 1.41% in the Turkish population, consistently with literature.

CWD ratio was higher in boys than in girls, consistent with the literature (9,12).

PE/PC ratio was reported as 2.2-5/1 in literature (9,11-15). Although PE prevalence is higher than some PCs, PC ratio was reported higher than PE in Argentina and Africa populations (10). In our study, PC prevalence was found higher than PE (PC/PE=1.59) similarly to Argentina and Africa populations, on the contrary to classical data.

Co-existence of CWD and scoliosis is a generally accepted knowledge. While this co-existence was reported as 15% in literature, this ratio was 5.03% in our study (11). This is caused by due to lack of concurrent use of inspection and radiological examination. Scoliosis and deformity degree was not found to be associated with the study of Frick et al. (16). An association could not be found between CWD degree and scoliosis in our study, similarly with that study.

Deformity awareness is higher in children with a positive family history as children think that this is a normal body appearance, particularly in closed societies. Awareness increases in children with a positive family history of CWD as this disease are known within the family. Prevalence Of Chest Wall Deformity In Turkish Population.

An ample amount of studies are available in the literature reporting that CWD affects cardio-pulmonary functions. Cardio-pulmonary functions were reported to improve post-operatively, particularly FEV1 was reported to improve 3 years after pectus bar had been removed in PE and cardiac symptoms were reported to improve as the heart reached its normal position (17-20). Loff S et al. (21) reported that PC did not cause physical disturbances however caused psychological disturbances. Koumbourlis et al. (22) reported that there was no physical symptom during rest in PE and obstructive pulmonary disease prevalence was high, on the other hand, Coskun ZK et al. (12) reported that restrictive pulmonary disease prevalence is higher in PE, more than half of CWD patients did not have physical complaints and there was not a significant impairment in respiratory function tests. Westphal et al. (11) reported chest pain as the most common physical disturbance however we found dyspnea as the most common physical disturbance. In our study, physical disturbance ratio was found higher in the age group 11 and in children who were aware of the disease in multivariate logistic regression analysis. Physical disturbance may have been found higher in the other age groups as deformity has become more evident in age group 11. Higher physical disturbance in children who were aware of the disease suggests that deformity affects the child psychologically rather than affecting physically.

In literature, the main reason for correcting CWD was correcting body image rather than correcting physical disturbance and related psycho-social causes (12,23-25). Hadolt B et al. (26) reported positive outcomes after Nuss operation in PE in the young who did not have self-esteem and who felt bad psychologically. Ji et al. (27) reported that children with PE had more psycho-social problems compared to general population and these problems were negatively affected by being between 12-16 years old, deformity severity and being mocked. In our study, psychological disturbances were found higher in the female gender, in presence
of family history in multivariate logistic regression analysis. Increased awareness and increased physical disturbance in children with family history arise from the reflection of psychological disturbance as a physical disturbance, but not from increased physical disturbance. Girls caring body image more than boys and being more sensitive than boys have resulted in female gender’s being an important factor which affects psychological disturbance in CWD. We observed during our examination that children with CWD came to the examination hesitatingly as their appearance caused psychological disturbance.

In the present study, awareness naturally increased as deformity degree increased and deformity became more evident as its degree increased.

In this study conducted in a large population, CWD was found more prevalent in boys (1.41%) and PC was found as the most common deformity type in our country, on the contrary to literature. While age group 11 and deformity awareness were effective on a physical disturbance in CWD, family history, and female gender were effective on psychological disturbance.

Financial Disclosure: No financial disclosure was declared by the authors.

Conflict of Interest: No conflict of interest was declared by the authors. Prevalence Of Chest Wall Deformity In Turkish Population.

References

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2. Morshuis W, Folgering H, Barentsz J, van Lier H, Lacquet L. Pulmonary function before surgery for pectus excavatum and at long-term follow-up. Chest 1994;105(6):1646-52.

3. Williams AM, Crabbe DC. Pectus deformities of the anterior chest wall. Pediatr Respir Rev. 2003; 4(3):237-42.

4. Gocmen H, Akkas Y, Doganay S. Poland syndrome: rare presentation in two cases.The New Zeland Med Journal.2010; 123:71-77

5. Stevens DB, Fink BA, Prevel C. Poland’s syndrome in one identical twin. J Pediatr Orthop 2000;20:392-5.

6. Rebeis EB, Campos JRM, Fernandez A, Moreira LFP, Jatene FB. Anthropometric index for pectus excavatum. Clinics 2007;62(5):599-606.

7. Bradford NF. Overweightand obesity in children and adolescents. Prim Care 2009; 36(2):319-29.

8. Akcam M, Boyaci A, Pirgon O, Dundar B. Evaluation of the change in the prevalence of childhood obesity in ten schools of Isparta city. Turk Arch Ped 2013; 152-5.

9. Rajabi-Mashhadi MT, Ebrahimi M, Mobarhan MG, Moohebati M, Boskabady MH, Kazemi-bajestani SMR. Prevalence of chest wall deformities in a large sample of Iranian children aged 7-14 years. Iran J Pediatr 2010;20(2):221-224. Prevalence Of Chest Wall Deformity In Turkish Population.

10. Goretsky MJ, kelly Jr. RE, Croitoru D, Nuss D. Chest wall anomalies:Pectus excavatum and pectus carinatum. Adolesc Med Clin 2004;15(3):455-71.

11. Westphal FL, de Lima LC, Neto JCL, Chaves AR, Junior VLS, Ferreira BLC. Prevalence of pectus carinatum and pectus excavatum in students in the city of Manaus, Brazil. J Bras Pneumol 2009;35(3):221-226.

12. Coskun ZK, Turgut HB, Demirsoy S, Cansu A. The prevalnece and effects of pectus excavatum and pectus carinatum on the respiratory function in children between 7-14 years old. Indian J Pediatr 2010;77:1017-19.

13. Haje DP, Haje SA, Simioni MA. Prevalencia das deformidades pectus carinatum e excavatum em escolares do Distrito Federal. Brasilia Med. 2002;39(1/4):10-5.

14. Fonkalsrud EW, Dunn JC, Atkinson JB. Repair of pectus excavatum deformities:30 years of experience with 375 patients. Ann Surg. 2000; 231(1):443-8.

15. de Matos AC, Bernardo JE, Fernandes LE,, Antunes MJ. Surgery of chest wall deformities. Eur J Cardiothorac Surg 1997; 12(3):345-50.

16. Frick SL. Scoliosis in children with anterior chest wall deformities. Chest Surg Clin N Am 2000;10(2):427-36.

17. Akcali Y, Ceyran H, Hasdiraz L. Chest wall deformities. Acta Chir Hung 1999; 38(1):1-3.

18. Lida H. Surgical repair of pectus excavatum. Gen Thorac Cardiovasc Surg 2010; 58(2):55-61.

19. Maagaard M, Tang M, Ringgaard S et al. Normalized cardiopulmonary exercise function in patients with pectus excavatum three years after operation. Ann Thorac Surg 2013; 96(1):272-8.Prevalence Of Chest Wall Deformity In Turkish Population.

20. Jeong Y, Park HJ, Lee J, Park JK, Jo KH. Cardiac morphologic changes after nuss operation for correction of pectus excavatum. Ann Thorac Surg 97(2):474-8.

21. Loff S, Sauter H, Wirth T, Otte R. Highly efficient conservative treatment of pectus carinatum in compliant patients. Eur J Pediatr surg 2015; 25(5):421-4.

22. Koumbourlis AC, Stolar CJ. Lung growth and function in children and adolescents with idiopathic pectus excavatum. Pediatr Pulmonol 2004; 38(4):339-43.

23. Steinmann C, Krille S, Mueller A, Weber P, Reingruber B, Martin A. Pectus excavatum and pectus carinatum patients suffer from lower quality of life and impaired body image: a control group comparison of psychological characteristics prior to surgical correction. European journal of Cardio-thoracic Surgery 2011; 40:1138-45.

24. Davis JT, Weinstein S. Repair of the pectus deformity:results of the ravitch approach in the current era. Ann Thorac Surg 2004; 78(2):421-6.

25. Matos AC, Bernardo JE, Fernandes LE, Antunes MJ. Surgery of chest wall deformities. Eur J Cardiothorac Surg 1997; 12(3):345-50.

26. Hadolt B, Wallisch A, Egger JW, Höllwarth ME. Body-image, self-concept and mental exposure in patients with pectus excavatum. Pediatr Surg Int 2011; 27(6):665-70.

27. Ji Y, Liu W, Chen S et al. Assesment of psychosocial functioning and its risk factors in children with pectus excavatum. Health and Quality of Life Outcomes 2011; 9:28.

Table 1: Deformity prevalence according to socio-demographic characteristics

CWD(+) CWD(-) Total p

N % n % n %

Gender

Female

Male

Total

63

136

199

0.45

0.96

1.41

6765

7144

13909

47.95

50.64

98.59

6828

7280

14108

48.4

51.6

100

0.0001

Age groups

11

12

13

14

Total

30

51

50

68

199

0.22

0.36

0.35

0.48

1.41

3317

3426

3731

3435

13909

23.51

24.28

26.45

24.35

98.59

3347

3477

3781

3503

14108

23.73

24.64

26.80

24.83

100

0.003

Table 2: Distribution of patients with PC according to gender 

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Female Male Total

n % n % n %

Chondrogladial symmetrical type 3 23,1 46 42,6 49 40,5

Chondrogladial asymmetrical type 4 30,8 41 38 45 37,2

Mixed type 2 15,4 20 18,5 22 18,2

Chondromanubrial type 4 30,8 1 0,9 5 4,1

Total 13 100 108 100 121 100

Table 3: Uni-variate and multivariate logistic regression analysis table between physical disturbance and gender, age groups, deformity degree, deformity awareness, family history, having knowledge about CWD, BMI percentile curves in patients with CWD. Prevalence Of Chest Wall Deformity In Turkish Population.

Risk Factor Physical disturbance (+) Physical disturbance (-) Crude OR

(95% CI) p Adjusted OR (95% CI) p

n % n %

Gender

Female < br />

*Male

12

30

19

22.1

51

106

81

77.9

0.8314(0.3935-1.757)

0.6282

1.265(0.535-2.991)

0.593

–

Age groups

11

12

13

*14

1

12

8

21

3.3

23.5

16

30.9

29

39

42

47

96.7

76.5

84

69.1

12.957(1.653-101.540)

1.452(0.635-3.319)

2.346(0.940-5.854)

–

0.015

0.376

0.068

16.011(1.890-135.614)

1.196(0.477-2.998)

2.574(0.932-7.107)

0.011

0.702

0.680

–

Deformity degree

Mild

Moderate

*Severe

21

16

5

17.1

23.2

71.4

102

53

2

82.9

76.8

28.6

12.143(2.206-66.853)

8.281(1.464-46.829)

–

0.004

0.017

–

5.350(0.688-41.616)

4.397(0.594-32.527)

–

0.109

0.147

–

Deformity awareness

Yes

*No

23

19

34.3

14.4

44

113

65.7

85.6

3.109(1.543-6.262)

–

0.001

0.307(0.132-0.713)

–

0.006

–

Family history

Yes

*No

5

37

33.7

20.1

10

147

66.7

79.9

1.986(0.640-6.164)

–

0.2280

–

–

–

–

Having knowledge about CWD

Yes

No

14

28

29.2

18.5

34

123

70.8

81.5

1.809 (0.858-3.812)

–

0.1162

–

–

BMI percentiles

*<5%

5-84%

85-94%

95%<

6

32

1

3

20

19.9

50

50

24

129

1

3

80

80.1

50

50

–

1.008 (0.380-2.671)

0.250(0.014-4.601)

0.250(0.040-1.564)

0.988

0.351

0.138

–

–

*Reference group

Table 4: Uni-variate and multivariate logistic regression analysis table between psychological disturbance and gender, age groups, deformity degree, deformity awareness, family history, having knowledge about CWD deformity and deformity type in children with CWD. Prevalence Of Chest Wall Deformity In Turkish Population.

Risk Factor Psychological disturbance(+) Psychological disturbance (-) Crude OR

(95% CI)

p Adjusted OR

(95% CI)

p

n % n %

Gender

Female

*Male

7

2

11.1

1.5

56

134

88.9

98.5

8.375(1.687-41.57)

–

0.002

–

15.440(1.684-141.586)

–

0.015

–

Deformity degree

*Mild

Moderate

Severe

2

6

1

1.6

8.7

14.3

121

63

6

98.4

91.3

85.7

–

0.174(0.034-0.885)

0.099(0.008-1.253)

0.035

0.074

–

0.656(0.075-5.766)

0.308(0.006-14.879)

–

0.551

0.704

Deformity awareness

Yes

*No

8

1

11.9

0.8

59

131

88.1

99.2

17.76(2.172-145.2)

–

0.0003

–

0.467(0.012-17.546)

0.681

Family history

Yes

*No

4

5

26.7

2.7

11

179

73.3

97.3

13.02(3.057-55.43)

–

0.0001

–

18.656(1.917-181.605)

0.012

–

Having knowledge about CWD

Yes

*No

7

2

14.6

1.3

41

149

85.4

98.7

12.72(2.545-63.57)

0.0001

–

0.142(0.004-5.294)

0.290

–

**Deformity Type

PE

*PC

15

26

19.7

21.5

61

95

80.3

78.5

0.8985(0.4408-1.831)

–

0.7683

–

0.850(0.129-5.615)

–

0.866  . Prevalence Of Chest Wall Deformity In Turkish Population.