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Master thesis

Department of Pediatric Surgery

“Establishment of right heart function changes of cardiac

echocardiography in patient with pectus excavatum before and

after surgery with Nuss procedure.”

A thesis submitted in part fulfillment for the degree of Master of Medicine

Marta Maria Ejsmont

Medical Faculty

Supervisor: Doc. Artūras Kilda

Lithuanian University of Health Sciences

Kaunas 2015/2016

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TABLE OF CONTENT

1. Summary ... 3 2. Acknowledgments ... 4 3. Conflicts of interest ... 5

4. Clearance issued by the Ethics Committee ... 6

5. Abbreviations ... 7

6. Introduction ... 8

7. Aim and objectives ... 9

8. Literature review ... 10

9. Research methodology and methods ... 15

10. Results ... 16

11. Discussion of the results ... 21

12. Conclusions ... 24

13. Practical recommendations ... 25

14. Literature list ... 26

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SUMMARY

Author's name and surname: Marta Maria Ejsmot

Research title: “Establishment of right heart function changes of cardiac echocardiography in patient

with pectus excavatum before and after surgery with Nuss technique.”

Aim: Establishment of function of right heart in patient with pectus excavatum (PE) before and 6

months after surgery with Nuss procedure using echocardiography and compare results with healthy control subjects.

Objectives:

1. Influence of Haller index on right ventricle (RV) volume.

2. Changes of right heart echocardiographic parameters after 6 months of PE repair with Nuss method. 3. Possible correlation and comparison of echocardiographic heart parameters in children with PE and healthy controls.

Methodology: The group of 6 asymptomatic patients, age 11-17, with no heart pathologies, was

analysed with echocardiography, before and 6 months after Nuss procedure. Parameters such as: height, weight, body surface area (BSA), heart rate (HR), ejection fraction (EF), Haller index (HI), RVEDD(right ventricle end diastolic diameter), RVEDV (RV end diastolic volume), RVESD (RV end systolic diameter), RVESV (RV end systolic volume) and TAPSE (tricuspid annular plane systolic excursion) were assessed. Results were compared with age- matched healthy control subjects.

Results: Results showed significant increase of RV diameters and volumes. Highest significance and

increase was in case of RVESD (p<0,001; increase by 30,19%). The lowest increase was in RVEDV, by 9,36% (p<0,005). In statistical correlation analysis we found strong downhill relation between TAPSE and RVEDD before and after Nuss procedure, as well as significant strong uphill correlation between TAPSE and Haller index before operation.

Conclusions: PE has direct impact on heart function and because of compression it is diminished. Recommendations: It is suggested that additional evaluation should be made 12 months after repair

and after bar removal to have total overview of heart changes during healing process.

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ACKNOWLEDGEMENTS

I would like to thank for help in performing echocardiographic examination to doctor Aristida Ziutelienė, MD. My gratitude also for doc. Renata Šimoliūnienė for help in statistical part of this study. Without their participation, the study could not have been successfully performed.

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CONFLICT OF INTEREST

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ETHICS COMMITTEE CLEARANCE

The ethics committee in the bioethics centre at Lithuanian University of Health Sciences allows the medical student Marta Maria Ejsmont, from VI course, on 2016-03-14 by her request Nr. BEC-MF-347, to proceed with her scientific research “Establishment of right heart function changes of cardiac echocardiography in patient with pectus excavatum before and after surgery with Nuss technique” which consists of echocardiographic measurements of heart parameters in patients with pectus excavatum and healthy controls and comparison between groups. All the research will be under the supervision of Dr. Artūras Kilda, from Pediatric Surgery Clinic.

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ABBREVIATION LIST

2D ECHO- two dimensional echocardiography BSA- body surface area

CI- cardiac index

CMR- cardiac magnetic resonance CO- cardiac output

CT- computer tomography ED- end diastole

EF- ejection fraction ES- end systole HI- Haller index HR- heart rate LV- left ventricle

LVSVI- left ventricle stroke volume index PE- Pectus excavatum

PI- pectus index RH- right heart RV- right ventricle

RVEDD- right ventricle end diastolic volume RVEDV- right ventricular end diastolic volume RVEDVI- right ventricle end diastolic volume index RVEF- right ventricle ejection fraction

RVESD- right ventricle end systolic diameter RVESV- right ventricle end systolic volume RVESVI- right ventricle end systolic volume index RVFAC- right ventricular fractional area change RVIVA- right ventricle isovolumic acceleration RVLD- right ventricle long dimension

RVSD- right ventricle short dimension SV- stroke volume

SVI- stroke volume index

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INTRODUCTION

Pectus excavatum (PE), also called funnel chest, is a congenital deformity of anterior chest wall characterized by posterior depression of sternum and adjacent ribs with simultaneous left side displacement and rotation of heart (1-3). Etiology of this anomaly is unclear although suspected is the costal cartilage overgrowth (2) or rib overgrowth (4), what prevents sternum elevation during development. It occurs approximately 1 to 300-400 of born and is 4-5 times more prevalent in males (3, 5, 6). It is measured by Haller index which is the ratio between transverse diameter and the anteroposterior diameter of the inner ribcage. Normal value is 2,5. Haller index (HI) > 3.6 is considered as severe PE. Symptoms are rare in children and adolescence. (3, 6). PE may be associated with mitral valve prolapse. Most commonly cosmetic discomfort occurs.

Retraction of the sternal bone decreases space in the chest cavity, therefore it may influence cardiopulmonary function. Studies suggest that underlying heart, especially right side may be compressed by reversed sternum and vertebral column. This may diminish filling of right heart chambers, consequently decreasing cardiac output (CO). As a result less blood with oxygen is delivered to muscles, what reflects in exercise intolerance (7). Many previous studies present significant improvement of cardiopulmonary function after Nuss repair of PE. (8-11). Some studies show significant increase of RVEDVI (right ventricle end diastolic volume index), RVESVI (right ventricle end systolic volume index), (8, 9) and SVI (stroke volume index) (9). Some researches after analysis have found minimal or none changes (12-13).

Most of recent studies follow trends of the earlier investigations. Nowadays evaluation of right heart in PE patients shows, significant increased of its diameter and volume (14). There are also analysis which, show no change between pre and postoperative values (3, 15- 16). Lately newer imaging modes are use for heart function assessment. CMR (cardiac magnetic resonance) is following. The purpose of study was to assess right heart function in patients with PE before and after surgery with Nuss method, using echocardiography and compare the results with healthy age- matched control subjects.

The group of 6 asymptomatic patients, age 11-17, with no heart pathologies, was analysed with echocardiography, before and 6 months after Nuss procedure. Results were compared with age- matched healthy control subjects.

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AIM AND OBJECTIVES OF THE THESIS.

Aim of this study was to establish function of right heart in patient with pectus excavatum (PE) before and 6 months after surgery with Nuss procedure using echocardiography and compare results with healthy control subjects.

Objectives:

1. Influence of Haller index on right ventricle (RV) volume.

2. Changes of right heart echocardiographic parameters after 6 months of PE repair with Nuss method. 3. Possible correlation and comparison of echocardiographic heart parameters in children with PE and controls.

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LITERATURE REVIEW

Pectus excavatum (PE, funnel chest) is congenitally developing skeletal deformity of anterior chest wall. It is depression of sternal bone and neighboring ribs toward vertebral column with simultaneous displacement of the heart to the left and its rotation (1). Most often sternum is depressed to the right of the midline (~80%), but it also may be moved to the left of the midline (~10%) or remain in the midline position (~10%) (17). Etiology is not clear. There are two hypotheses. First and earlier one talks about ribs overgrowth (4), while the new suspicion point on overgrowth of costal cartilage (2). Both result in retraction of sternum into the chest. Symptoms, such as discomfort, fatigue, dyspnea, especially on exertion, increased breath rate, exercise intolerance, palpitations dizziness and anterior chest pain, occur rare, usually in severe cases (Haller index >3.6) (6, 18). Usually, the problem is the cosmetic discomfort.

There is analysis which classifies PE into a few different groups. According to phenotype distribution we can distinguish cup- shaped (localized) PE, which is most common among analyzed patients, saucer- shaped (diffused), trench- like and Currarino- Silverman, which was found only in about 1% of analyzed population. Classification of PE based on deepest point position in the chest can be: to the right of midline- most common, to the left of the midline and on the midline. Findings are base of medical records, preoperative photographs and CT imaging (17).

There are two methods of repair: Ravitch method (open surgery) and Nuss procedure (minimally invasive procedure). Nuss procedure, also “minimal invasive” or “closed” procedure was invented in 1987 by Dr. Donald Nuss and published in 1998. It requires two incisions on both lateral sides of chest and formation of subcutaneous tunnel through which introducer and then stainless bar is places posteriorly to the sternum and anteriorly to the heart and lungs (in pleural space). Third incision is made for thoracoscope for monitoring the procedure. (19)

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Table 1. Criteria for Surgical Referral (5)

1. Presence of symptoms 2. Progressing deformity

3. Paradoxical movement of the chest wall with deep inspiration 4. Haller index on CT > 3,0

5. Compression or displacement of heart 6. Lungs' compression

7. Pulmonary function test shows significant restriction 8. Mitral valve prolapse

9. Other cardiac pathologies, secondary to deformity. 10. Significant body image abnormalities

In addition:

Fail previous repair

Pathological cardiopulmonary studies

First report of successful repair of PE with Nuss procedure was published in 1998. Inventor, American surgeon Donald Nuss presented data of over 10 years studies, describing work on 45 cases of PE with positive results in repair with minimally invasive method (18). Analysis of heart function were started prior to Nuss’ publication. The earliest one was reported in 1948 (20). By 2012 were established approximately 168 articles. The results are very different, but trends are toward beneficial influence of PE repair on cardiac function. There is no definitive, proved statement, which stands by either opinion.

Below are presented opinions about influence of Nuss procedure on heart function changes from 1996 to 2015. It was taken under consideration method of examination, age, sex distribution, time of examination, analyzed data.

In the very first observations, specialists suggested improvement of cardiac function. It was based on increase in exercise tolerance and O2 pulse (11). Later, more specific data showed function improvement, especially in right heart. There were analyzed two groups divided into patients with mild or moderate and severe degree of PE. By using echocardiography RVEDVI and RVESVI were calculated. Both parameters increased after surgery in analyzed groups, but only the first measurement was significant for two analyzed groups (mild or moderate and severe). The second index was significantly changed only in second- severe group (8). The same group some time later took under consideration one more parameter- left ventricle stroke volume index (LVSVI). They confirmed previous findings about increase of RVEDVI and RVESVI, as well as found increase of LVSVI.

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However, they state that increase of LVSVI occurred only in patients with severe deformities and in parallel studies conclude that Nuss repair did not change LV function in patients with mild or moderate deformity degree. Conclusion is that RV volumes may increase with the enlargement of thoracic cavity after repair until total decompression. There is no quantitative relationship of RV structural and functional improvement with degree of sternal elevation (9).

In 2005 Bawazir et. al. presented studies analyzing cardiopulmonary effect after Nuss repair of PE. By echocardiography stroke volume (SV), cardiac output (CO) and cardiac index (CI) were measured 3 and 21 months after operation and after bar removal. They concluded significantly positive changes over long term monitoring and suggested changes in teaching about lack of influence of PE on physiological data (10). Similar conclusion made Sigaet et. al. in 2007. He analyzed SV, CO and CI before Nuss procedure and after bar removal. It was suggested that obtained findings are result of better chest wall efficiency and CO during exercise (21). There is specified more physiological suggestion, how compressed chest impacts on right atrium and ventricle. It can decrease diastolic filling, thereby decreasing total CO, oxygen transport to muscles and finally exercise tolerance. (7)

In later studies ejection fraction was analyzed. Saleh et. at compared cardiothoracic structure and function in PE patients and healthy ones using cardiovascular magnetic resonance. Although this method differs from used in our study, it was used as a reference to compare results of assessment of the same data by different method. It was achieved statistical significance of about 6% in RVEF difference between healthy individuals and those with PE, however no significant correlation with pectus index (PI). They also analyzed RV short (RVSD) and long (RVLD) dimensions at end-systole (ES) and at end- diastole (ED). RV short dimension was significantly less in PE patients during both cardiac phases. RV long dimension was significantly greater during ED in PE patients. RVSD shortening is smaller in PE group, but RVLD shortening in the same group was higher. Conclusion is that pectus patients have decreased RV short axis diameter and increased long axis, as well as decreased RVEF. It shows structural impact on heart performance, rather than physiological dysfunction (2).

Tang et. al. hypothesized that Nuss procedure results in improvement of cardiopulmonary function during exercise. A group of 75 teenagers (49 patients and 26 controls) was analyzed at rest and during exercise before surgery and 12 months after. Analysis of LV showed that fractional shortening and EF did not differ between controls and patients, neither before operation nor 12 months after. Diastolic dimensions, despite some changes over the year, were not significant between analyzed groups. There were meaningful changes in maximum CI. It remained lower than the control but there was significant improvement 12 months after surgery in comparison to preoperative value. It is thought to be caused by facilitating heart filling during exercise after increase of space in thoracic cavity following the Nuss procedure. The other reason may be that children are more prone to exercise

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because of new appearance. SVI (stroke volume index), which was lower in pectus patients comparing to healthy ones, normalized after operation. It suggests that improvement in CO was a consequence of increase in maximum HR and SV among patients. (22)

Most studies analyze patients with pectus excavatum before and after surgery and compare them with healthy control group. Following study investigated patient with defect with healthy ones. Fractional shortening, EF, LVED and RVED dimension were investigated at rest. Maximum HR, minimum HR, heart rate increase, maximum CI and SI were checked during exercise. There was no difference in received results between patients and healthy teenagers during echocardiographycal examination. Nevertheless CI value was lower in PE patients during exercise. They explain that no ability to improve CI during physical exertion may be the reason for dyspnea and fatigue seen in patients. They suspect that lower heart performance in affected group is caused by habitual lack of exercise. (3)

Comparison of qualitative and quantitative RV function showed Rajeev et. al hypothesizing that patients who have qualitative RV impairment, will have normal function of RV assessed quantitatively by CMR or echocardiographic examination. In other words RV function is not associated with qualitative impairment. Degree of RV dysfunction was assessed by qualitative echocardiography. There were measured tricuspid annular plane systolic excursion (TAPSE) and right ventricular fractional area change (RVFAC). RVEF was established by CMR. The analysis showed in general normal results. It did not meet indications for surgery therefore non patient underwent repairing procedure. The aim was to show importance of qualitative assessment, during evaluation of biventricular function. The quantitative echographic evaluation may be alternative and probably more accurate tool in RV function investigations to CMR derived imaging. It point that different examination modes may show inconsistent results, what can influence decision for surgery. (15- 16) Other researchers, who used CMR to assess right heart function claim that changes in RVEF depend on degree of compression, but severity of dysfunction cannot be predicted by Haller index. (23- 24).

Gürkan et al. performed echocardiographic assessment of RV function before and 1 month after surgery of PE. Doppler method was used. Following parameters were measured: RVEDD, TAPSE, pulsed tissue Doppler systolic velocity(S'), RV isovolumic acceleration (RV IVA) systolic pulmonary artery pressure, LVEF and myocardial performance indices of right and left ventricles (Tei index). Results after operation showed significant increase of RVEDD, TAPSE, S' and RV IVA. Tei indexes also highly improved. Obtained data show that chest decompression has positive impact on postoperative heart function. It can be accurately measured, before procedure as well as during postoperative monitoring, by quantitative echocardiographic method (14)

More and more studies concentrate on psychological aspect of PE repair. The importance of physical appearance very often is underestimated by pediatric surgeons. Many children lose their

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confidence due to abnormal chest appearance, which can be pointed out by peers. There is study which, except cardiopulmonary function, evaluated self- rated appearance. Pediatric patients filled questioner, asking about how they find their new look. The results are strongly pointing that impact on self- perception is much bigger than the physical effect. (25)

In early studies, most researches confirm changes in heart function before and after Nuss procedure PE surgery, especially the right heart. They have slightly different approach to parameters, which nevertheless lead to overlapping conclusion. Some paid more attention on changes in heart volume and therefore indexes. Other considered changes in mean amount of blood passing through heart. Despite approach, all mentions articles show more or less significant changes after surgery. Later findings do not differ much from previous studies. Lately performed investigations are probably more detailed and developed but conclusions are similar to those of 10 or more years.

Some studies point that right heart performance depends on its structural features (decrease space in the chest cavity) rather than on physiological function changes. (2, 22) However this statement refers to most patients, with exception of those with congenital heart diseases or malformations, which may affect its physiology.

Only very few analysis, suggest that PE deformity has small or no impact on heart function. Some claim that PE has stronger impact of appearance and thus self- confidence. It may cause lack of exercise due shame or low self esteem. After repair procedure, children or teenagers feel more brave and satisfied with their outlook, what make them to exercise more willingly (3, 25).

There are also works which analyzed not only right heart function but also the left heart parameters. That occurs in patients with severe deformities, when heart has not only enough space for function of right side but also it is too little space for work of left heart. (9)

Summarizing, it should be pointed that main cause of all abnormal parameters is decreased chest cavity space, caused by funnel chest. Heart has not enough area for full extend and therefore filling with blood is not complete as well. Problem of appearance cannot be underestimated. As most diagnosed patients are in adolescent age, look is frequently most important aspect. Due to mentioned, patients are not prone to exercise, moreover they avoid it. At one point it may be caused by exertion intolerance, but on the other hand children and teenagers want to avoid embarrassing questions of peers, regarding pathological chest. Decision of Nuss procedure should be based not only on indications listed in table 1, but also patients' and their parents' opinion and willing.

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RESEARCH METHODOLOGY AND METHODS.

The heterogeneous sample of 4 male and 2 female in age ranged between 11-17 years, who were scheduled for operative PE repair with Nuss procedure in Paediatric Surgery Department of Clinical Hospital of Lithuanian University of Health Sciences, was evaluated and compared with control subject in number of 6, matched with sex, age, height and weight. Studies were carried from January 2015 to January 2016. Parent or legal guardian of each patient signed consent about agreement that their child will participate in the research. PE patients were assessed with 2- dimensional echocardiography (2D ECHO) imaging before operation and 6 months after. Haller index was measured using CT scans as standard assessment tool before PE operation.

Transthoracic 2D ECHO imaging was performed at rest in all patients (study subjects and control group) with GE Vivid S6 Cardiovascular Ultrasound System®,by the same echo- cardiologist to exclude investigation variations. Studies were approved by LSMU Bioethical Center and all parents or legal guardians signed informed consent.

Following measurements were assessed: height, weight, body surface area (BSA), heart rate (HR), ejection fraction (EF), Haller index (HI), RVEDD, REVDV, RVESD, RVESV and TAPSE. In apical 4-chabmer view RVEDD, RVEDV, RVESD and RVESV were measured. TAPSE was evaluated in M Mode echocardiography as a difference between motion of annulus in diastole and systole. EF was assessed from 2D M mode-Teicholz method. Haller index (ration between anteroposterior and transverse distance of the chest) was measured from CT scan done as standard tool before procedure.

All data was analyzed in Microsoft Excel® and IBM SPSS Statistics®. Mean and standard deviation was calculated. The non- parametric Man-Withney-U test was performed to determine if data evaluated before and after surgery are significantly different. P <0.05 was believed to be statistically significant. To check correlation between test results Sperman analysis was used.

Nuss procedure is performed in minimally invasive mode. Two lateral incisions are made in order to insert metal bar. Stainless steel plate is introduced anteriorly to heart and lungs in pleural space, behind the sternum. The thoracoscope is used to monitor progression of procedure and is entered through the third incision. Bar usually stays in the body for 2-3 years, depending on healing process.

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RESULTS

The median values, minimal and maximal values of the right ventricular parameters are presented in the table 2. The end diastolic and systolic RV diameter and volume as well as TAPSE show significant increase after Nuss procedure (p<0,05). Percentage by which parameters increased was also evaluated. The highest change showed RVESD (increase by 30%), which had the strongest significance value (p= 0,00083). Haller index is 4,12 ± 1,13.

Table 2. Right ventricular parameters assessed by transthoracic echocardiography before and 6 months after Nuss repair of Pectus Excavatum and controls values.

RVEDD (mm) RVEDV (ml) RVESD (mm) RVESV (ml) TAPSE (mm) Before surgery (median) 26,00 126,50 17,75 35,50 19,50

After surgery (median) 28,00 136,00 22,50 42,50 22,50

Percentage change 8% 8% 27% 20% 15% P Value < 0,03 < 0,03 < 0,03 < 0,03 < 0,03 Minimal value - before surgery; - after surgery -21; -24; -112; -120; -15; -21; -30; -35; -18; -20; Maximal value - before surgery; - after surgery -30; -36; -135; -154; -22; -30; -43; -63; -24; -27; Controls (median) 26,50 131,50 23,00 41,50 23,50

Assessed data were compared between each other, using Spermaan function, in order to find correlation. The results are shown in table 3. The strongest and significant (p < 0,05) correlation with uphill trend was between preoperative values of Haller index and TAPSE (0,841). RVEDD and TAPSE before and after Nuss procedure demonstrate strong downhill correlation (respectively -0,77 and -0,779), but both correlation were not significant (respectively p=0,073 and p= 0,068) Graphical presentation of correlation between Haller index and TAPSE is in chart 5.

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Table 3. Correlation between received values.

Haller and TAPSE BO 0,841 p<0,05 Strong uphill linear correlation

RVEDD BO and TAPSE BO - 0,77 p=0,073 Strong downhill linear correlation RVEDD AO and TAPSE AO - 0,779 p=0,06 Strong downhill linear correlation BO= before operation; AO= after operation

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Chart 2.Changes of RVEDV after operation of PE (p<0,03).

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Chart 4. Changes of RVESV after operation of PE (p<0,03).

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DISCUSSION OF THE RESULTS

This study analyzed impact of PE on heart function and prospective changes after surgical repair with Nuss method. Heart function parameters were measured by echocardiographic imaging.

Most of studies show significant changes in heart parameters. Commonly they stress on RV dimension parameters, EF, as well as LV parameters. Many studies combined investigation of heart function with pulmonary performance. Many did exercise test.

All analyzed patients demonstrated significant changes in measured values. Haller index was >3 (severe) in 4 out of 6 patients. Increase of RVEDD in 2 out of 6 patients was more than 20%, RVEDV increased by more than 10 % in 2 out of 6 patients, RVESD in all patient increased by ≥ 20% and in 2 patients even ≥40 percent, RVESV in 1 patient increased by >45%, in the rest it was more than 15%. TAPSE changes were >20% in one subject, 3 of them had >10 %, 1 was < 5%. Comparing these results with controls, before Nuss procedure PE subjects had reduced systolic and diastolic RV diameters and volumes, as well as TAPSE. After repair, mentioned parameters increase to values comparable with normal. Probably it is caused by surgical decompression of chest, what in consequence increases space in the chest cavity.

Comparison of TAPSE and Haller index before operation showed significant strong uphill correlation (0,841; p<0,05). It was assumed that both parameters are proportional to certain extent. With higher value of Haller index, value of TAPSE will increase. Due to compression heart does not have enough space for horizontal extension, therefore it is compensated by more vertical work, what is reflected in increased TAPSE value. This may be related with mitral valve prolapse, which is common in patients with PE (25). Heart displacement to the right results in decrease space for its extension and because of this, TAPSE performance may affect mitral valve function, what can lead to its prolapse. As PE is congenital anomaly, effect of tricuspid valve performance on mitral valve is not direct, but it may be related as body develops.

Evaluation of correlation between RVEDD and TAPSE before and after surgery revealed strong downhill linear correlation, but did not meet conditions of significance (before operation p= 0,073; after operation p= 0,068). P value is close to significant, what may reflect insufficient amount of study object.

Obtained results corroborate with four other studies analyzing heart dimensions and volumes before and after repair in PE patients compared with age-matched controls. (8- 9, 2, 14).

Kowalewski et al. analyzed 3 groups of patients according to the degree of PE- mild, moderate and severe. They evaluated RVEDVI and RVESVI, which are proportional to volume itself. Results, showed increase in both values but first changed significantly. They also claimed that LVSVI also

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increased (in severe group) but it is not subject of this study. (8- 9) This statement agrees with our results. Despite the fact that we analyzed only volume of right ventricle, not its index, we can assume that as volume is related to index, increase of one will indirectly cause increase of the second.

Saleh et al. did study about right ventricular short and long dimensions at systole and diastole using CMR. RV short dimensions were lower in PE patient during diastole and systole (respectively 22,6 ± 7,0 mm and 18,9 ± 5,9 mm) in comparison to control (respectively 33,6 ± 4,6 mm and 26,7 ± 4,3 mm). Long dimension was bigger at end diastole (79,1 ± 9,6 mm) versus controls (73,3 ± 9 mm). The conclusion is that PE patients have decreased RV short axis and increased long axis. It is evidence of right heart compression by inward chest. In this study significant changes in RV volumes and diameters correspond with findings of Saleh et al. Even though imaging method is different, it was used as a reference to see similarity in the same data evaluated by different tools (2).

Gürkan et al. analyzed RV function by using echocardiography. They presented significant increase of RVEDD and TAPSE 1 month after surgery. This data agrees with those obtained in present study. Time of postoperative ECHO was shorter than we did what shows, direct and quick effect of repair procedure, on which there is no need to wait long. (14)

O'Keefe et al. analyzed patients' status before operation and 3 months after bar removal. They checked pulmonary function, cardiac echography and exercise tolerance. Obtained parameters showed improvement in cardiopulmonary performance. It shows positive outcome of repair, however it can be also related with growing factor, as bar is removed 2-3 year after repair. During this time, children grow and get stronger, what also has influence of cardiopulmonary function, so parameters after bar removal are not complete reflection of repair procedure results. (25) Gürkan et al. eliminated growing factor by analyzing heart function 1 month after surgery. (14) Evaluation of heart function 6 months after is also reliable. At this time patients finish rehabilitation after surgery and it is not enough time for significant changes in growth and strengthening of heart function, therefore growing factor in present study is eliminated as well.

Assessment of cardiopulmonary function after bar removal is important in evaluation of pectus recurrence. (25) Final results of the repair, rehabilitation and healing may be evaluated and eventual decision about re- repair may be made. This present study should be continued with analyzes 12 months after repair and post bar removal check. Then it would present complete following of heart changes during healing process.

Obtained results are different than some studies show. In other investigations, changes in RVEDD occurred but were not significant or there were no changes at all. (3) Rajeev et al. evaluated RV function using echocardiography for assessment of TAPSE and RVFAC, and CMR to check RVEF. They stand that quantitative echocardiographic assessment is as accurate in measuring RV function as the CMR and can be an alternative mode of assessment. Obtained results show normal

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TAPSE values, which are disagree with results obtained in this studies. The number of patients was similar to one in present study. They were referred due to dyspnea and previously present qualitative RV dysfunction. (15- 16) Tang et al. analyzed cardiopulmonary function before and 12 months after Nuss operation. Heart function parameters were assessed from echocardiography. RV measurement was RVEDD. The results showed significant increase of RVEDD in control group, but no change in PE subjects. It is explained by age- related growth, which was decided is eliminated in this study (22).

There are many factors which influence heart performance. Appearance is very important aspect in this anomaly. O'Keefe et al. took this feature under consideration by assessing patients' attitude toward the problem. They claim that it may be even more meaningful that the structural repair (25). Children or teenagers are less motivated to exercise if their body looks abnormal. They will not practice sports if it is accompanied by symptoms. It may lower their self-esteem and cause loose of confidence. Therefore, in this study, despite that Haller index of two analyzed subjects was less than 3, specialists decided to perform repair.

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CONCLUSIONS

1. In patients with PE, HI has influence of RV volume. Correlation of HI and TAPSE shows significant strong uphill trend (0,841, p< 0,05).

2. There is significant change in RH echocardiographic parameters 6 months after PE repair with Nuss method. Diameters and volumes during diastole and systole increased significantly (p<0,03). Highest change demonstrated RVESD (increase by 27%), the lowest in RVEDD and RVEDV, by 8%.

3. RV parameters comparing to control values, normalized after Nuss repair. Increase RVESD and TAPSE, were significant (respectively p< 0,01 and p< 0,05).

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PRACTICAL RECOMMENDATIONS

PE is pathology which needs long time follow up after repair. Assesment of heart was performed at the end of rehabilitation period, before growing factor may influence healing outcomes. It is suggested that additional evaluation should be made 12 months after repair and after bar removal. Regular analysis would show changes of heart functioning during healing process.

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LITERATURE LIST

1. Toepper A, Poleichtner S, Prothmann M, Zagrosek A, Schwenke C, von Knobelsdorff F, et al. Cardiac magnetic resonance in patients with pectus excavatum: impact of thoracic surgery on cardiac function- a follow up study. Journal of Cardiovascular Magnetic Resonance 2014; 16(Suppl 1):P96. 2. Saleh RS, Finn JP, Fenchel M, Moghadam AN, Krishnam M, Abrazado M, et al. Cardiovascular MRI in patients with pectus excavtum compared with normal controls. Journal of Cardiovascular Magnetic Resonance 2010, 12:73

3. Lesbo M, Tang M, Nielsen HH, Frøkiær J, Lundorf E, Pilegaard HK, et al. Compromised cardiac function in exercising teenagers with pectus excavatum. Interactive CardioVascular and Thoracic Surgery 13 (2011) 377-380

4. Park CH, Kim TH, Haam SJ, Lee S. Rib overgrowth may be a contributing factor for pectus excavatum: Evaluation of prepubertal patients younger than 10 years old. Journal of Pediatric Surgery 50 (2015) 1945–1948

5. Jaroszewski D, Notrica D, McMahon L, Steidley E and Deschamps C. Current Management of Pectus Excavatum: A review and update of Therapy and Treatment Recommendations. J Am Board Fam Med 2010;23:230 –239.

6. Oezcan S, Attenhofer Jost CH, Pfyffer M, Kellenberger C, Jenni R, Binggeli C, et al. Pectus excavatum echocardiography and cardiac MRI reveal frequesnt pericardial effusion and right-sided heart anomalies. Eur Heart J Cardiovasc Imaging. 2012; 13 (8): 673-679

7. Colombani PM. Preoperative assessment of chest wall deformities. Semin Thorac Cardiovasc Surg. 2009; 21:58-63.

8. Kowalewski J, Barcikowski S, Brocki M. Cardiorespiratory function before and after operation for pectus. Excavatum. Medium-term results. European Journal of Cardio-thoracic Surgery. 1998; 13: 275–279.

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12. Malek MH, Berger DE, Housh TJ, Marelich WD, Coburn JW and Beck TW. Cardiovascular Function Following Surgical Repair of Pectus Excavatum. A Metaanalysis. CHEST. 2006; 130:506– 516.

13. Guntheroth WG and Spiers PS. Cardiac Function Before and After Surgery for Pectus Excavatum. Am J Cardiol. 2007;99:1762–164.

14. Gürkan U, Aydemir B, Aksoy S, Akgöz H, Tosu AR, Öz D, et al. Echocardiographic assessment of right ventricular function before and after surgery in patients with pectus excavatum and right ventricular compression. Thorac Cardiovasc Surg. 2014 Apr 25;62(3):231-5.

15. Narayan RL, Vaishnava P, Castellano JM and Fuster V. Quantitative assessment of right ventricular function in pectus excavatum. J Thorac Cardiovasc Surg. 2012;143:e41-2.

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25. O'Keefe J, Byrne R, Montgomery M, Harder J, Roberts D, Sigalet DL. Longer term effects of closed repair of pectus excavatum on cardiopulmonary status. J Pediatr Surg. 2013 May;48(5):1049-54.

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ANNEX No. 1

TIRIAMOJO ASMENS TEVĖ/GLOBĖJŲ SUTIKIMO FORMA

Aš, motina/ tėvas ..., sutinku, kad mano vaikas dalyvaus Lietuvos sveikatos mokslų universiteto Medicinos fakulteto studentės Marta Maria Ejsmont atliekamame tyrime, skirtame nustatyti kardioechoskopinius pokyčius prieš ir po įdubos krūtinės operacijos.

Tėvas/ Mama/ Globėjas _____________________

(Parašas)

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