Original Article
How pulmonary function changes after pectus excavatum correction surgery
Asian Cardiovascular & Thoracic Annals 0(0) 1–5 ß The Author(s) 2015 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav DOI: 10.1177/0218492315596464 aan.sagepub.com
Pinar Kuru1, Asli Dudakli2, Hakan Mursaloglu2, Hazal Arikan2, Aysu Oktay2 and Mustafa Yuksel3
Abstract Aim: We aimed to determine the effects of minimally invasive repair of pectus excavatum on pulmonary function and quality of life. Methods: Minimally invasive pectus excavatum repair was undertaken in 80 patients with a mean age of 16.91 4.37 years (range 7–30 years) and a mean Haller index of 4.07 1.39; 85% of the patients were male. They and their parents completed the Nuss Questionnaire Modified for Adults, and pulmonary function tests were performed on the patients before and 6 months after the operation. Results: The mean Nuss score was 31.06 6.78 before the operation and it increased to 37.1 8.31 (p ¼ 0.000) 6 months after the operation. Forced vital capacity decreased from 3.70 1.23 to 3.48 1.03 L (p ¼ 0.05) postoperatively. The percentage of expected forced vital capacity decreased from 83.21% 16.97% to 76.52% 20.98% (p ¼ 0.01). There was no significant change in forced expiratory volume in 1 s. The mean ratio of forced expiratory volume in 1 s to forced vital capacity was 86% preoperatively and it increased to 91% postoperatively (p ¼ 0.000). Conclusions: Minimally invasive pectus excavatum repair has a positive impact on the quality of life of pectus excavatum patients, but a negative impact on forced vital capacity. Follow-up studies are needed to assess the long-term changes in pulmonary function after this operation.
Keywords Body image; Funnel chest; Patient satisfaction; Quality of life; Minimally invasive surgical procedures, Respiratory function tests
Introduction Chest wall deformities are a common type of congenital deformity. Anterior chest wall deformities become prominent during childhood and may cause psychologic, orthopedic, and physiological problems, according to the severity. Medium and severe deformities can be corrected by surgical intervention during childhood, with good cosmetic, orthopedic, and functional outcomes.1 Pectus excavatum (PE) is the most common congenital chest wall deformity with an incidence of 1/300–400 live births. It is characterized by a posterior depression of the sternum and costal cartilages, however, the first the costal cartilages are usually in the normal position. PE is seen more often in Caucasians and males, and the severity varies. The asymmetric form is the most common, and the right side is generally more depressed, sometimes with sternal rotation.
PE usually becomes more prominent during the growth spurt, but in some cases, it becomes less severe. Scoliosis is a frequently associated anomaly, seen in a quarter of cases.2 PE and scoliosis are often found in patients with Marfan syndrome. Congenital cardiac anomalies and asthma may accompany PE. Although 1 Department of Neurology, Northwestern University Feinberg School of Medicine, Chicago, IL, USA 2 Marmara University School of Medicine, Istanbul, Turkey 3 Department of Thoracic Surgery, Marmara University School of Medicine, Istanbul, Turkey
Corresponding author: Pinar Kuru, MD, Northwestern University Feinberg School of Medicine, Department of Neurology, 303 E Chicago Ave. Ward Building 10-015., Chicago, IL, USA. Email: [email protected]
Downloaded from aan.sagepub.com at University of Sussex Library on August 30, 2015
2
Asian Cardiovascular & Thoracic Annals 0(0)
it is often asymptomatic, it can cause shortness of breath, easy fatigue, chest pain, and palpitation. Usually, there is no objective pathology in pulmonary function tests and cardiac evaluation, so this deformity is accepted as a cosmetic and psychosocial problem. In medium and severe forms of PE, the lungs and heart are displaced, resulting in exercise intolerance. These patients have round shoulders and a typical pectus posture. In severe PE, cardiorespiratory function problems can arise, with mild to moderate exercise intolerance, chest pain with effort, and recurrent respiratory tract infections.3 The aim of the surgery is to elevate the depressed part of the chest and return the sternum to the normal position. The mechanism depends on chronic pressure applied to the sternum with the aid of a shaped nickel-steel bar inserted retrosternally for a period of time. Minimally invasive repair of pectus excavatum (MIRPE) was described in 1987. To facilitate the technique, special equipment and a stabilizer to inhibit bar rotation were developed. Pulmonary function tests are commonly used to evaluate respiratory diseases, and spirometric tests assess pulmonary function. In a recent meta-analysis, it was reported that surgical correction of PE improved cardiovascular function, but pulmonary function test results did not change.4 Another study on pulmonary function reported similar results.5 However, in some case series, postoperative improvements in pulmonary function were noted.6,7 In another meta-analysis, early postoperative results indicated decreased pulmonary function, but amelioration was predominant in long-term follow-up.8 In a study comparing pulmonary outcomes of the Ravitch and Nuss techniques, pulmonary capacity decreased after the Ravitch operation and forced expiratory volume in the first second of exhalation (FEV1) was increased after the Nuss operation.9 In the present study, we aimed to investigate the effects of MIRPE on pulmonary function and quality of life in patients with PE.
Patients and methods This prospective follow-up study included 80 patients with PE who underwent MIRPE by the same surgeon (MY) between 2012 and 2014 in the Department of Thoracic Surgery, Marmara University Hospital. Cosmetic complaints were the main reason for attending the thoracic surgery outpatient clinic. A complete clinical history was taken, a physical examination was performed, including documenting photographs, radiographic imaging. Pulmonary function and laboratory tests were carried out before and 6 months after the operation in all patients. We also obtained a postoperative radiograph of the chest to confirm the placement of
the bar and to detect pneumothorax. The sample size calculated according to primary outcome and the power of the study was 93.9% with 80 patients. Marmara University School of Medicine Clinical Research Ethics Committee approved this study, and informed consent was obtained from each participant. MIRPE was performed under general anesthesia. To insert a curved bar under the sternum, a lateral incision was made on both sides of the chest. The bar (Tasarimmed; Medical Devices Manufacturing and Marketing, Inc., Istanbul, Turkey) was individually curved for each patient and used to elevate the depression of the chest wall. It was fixed to the ribs on both sides, and the incisions were closed primarily. To stabilize and fix the bar to the rib, a steel plate was used, which was not visible from the outside. At the end of the treatment, the bar was removed in a short surgical procedure. The Pectus Excavatum Evaluation Questionnaire (Nuss questionnaire; NQmA) is a disease-specific quality-of-life assessment tool for children with PE and their parents. The validity and reliability of the Turkish NQmA was confirmed by Bahadir and colleagues.10 Although the original parent form contained 13 items, as a result of the reliability and validity analyses, two items were excluded. The patient version of the NQmA includes 12 items and the parent version includes 11 items. The score for each items ranges from 1 to 4. Possible minimum and maximum scores are 12–48 for the patient form, and 11–44 for the parent form; higher scores indicate a better quality of life. The Turkish version of the NQmA has physical and psychosocial components. The patients and most of their parents completed the questionnaire before and 6 months after the surgery. Anteroposterior and lateral radiographs were used to calculate the Haller (pectus) index as a quantitative indicator of the severity of the chest wall deformity. The Haller index was calculated by dividing the width of the chest wall at its widest point by the distance between the posterior surface of the sternum and the anterior surface of the spine. A Haller index greater than 3.25 is one of the indications for surgery.11 The surgical indication in our study was mainly decided according to physical and laboratory examinations, age of the patient, and expectations. Pulmonary function tests were performed in the same laboratory on all patients before and 6 months after the surgery. Forced vital capacity (FVC), FEV1, percentage of predicted FEV1 (FEV1%), and the FEV1/FVC ratio were recorded. For descriptive data, mean standard deviation and percentages were used. The NQmA patient and parent forms and pulmonary function test results were analyzed using the dependent-samples t test to determine
Downloaded from aan.sagepub.com at University of Sussex Library on August 30, 2015
Kuru et al.
3
Table 1. Characteristics of 80 patients who underwent repair of pectus excavatum. Variable
No. of patients
Male Symmetric Associated anomaly or disease Scoliosis Haller index > 3.25 Age at operation (years) [range] Total hospitalization (days) [range] Postoperative hospital stay (days) [range] Haller index [range] Operative time (min) [range]
68 (85%) 53 (66.3) 32 (40%) 9 (11.3%) 58 (72.5%) 16.91 4.37 [7–30] 6.61 2.51 [3–18] 4.58 1.54 [2–10] 4.07 1.39 [1.29–9.5] 67.12 22.93 [30–150]
Table 2. Preoperative and postoperative pulmonary function test results in 80 patients who underwent repair of pectus excavatum. Test
Preoperative
Postoperative
p value
FVC (L) FVC% FEV1 FEV1% FEV1/FVC
3.7 1.23 83.21 16.97 3.12 1.04 83.67 19.55 0.86 0.14
3.48 1.03 76.52 20.98 3.19 1 83.33 22.67 0.91 0.07
0.05 0.01 0.53 0.89 0.00
FEV1: forced expiratory volume in 1st s; FEV1%: percentage of predicted forced expiratory volume in 1st s; FVC: forced vital capacity; FVC%: percentage of predicted forced vital capacity.
the statistical significance of the data before and after the operation. Pearson’s correlation coefficient and the Mann-Whitney U test were used to determine the relationship between quality-of-life scores and the demographic variables. In all analysis, p < 0.05 was accepted as significant.
Results The characteristics of the patients are given in Table 1. The majority of these patients were male (85%), 66.3% had a symmetric deformity, and 40% had an associated anomaly. The mean NQmA score before the operation was 31.06 6.78 and it increased to 37.1 8.31 (p ¼ 0.000) at 6 months after the operation. FVC and percentage of predicted FVC decreased postoperatively (Table 2). There was no significant change in FEV1 or FEV1%, but the mean FEV1/FVC ratio increased significantly postoperatively. Quality-of-life scores in both the patient and parent forms improved at 6 months postoperatively (Table 3, Table 4). There was no correlation between Haller index and preoperative pulmonary function tests, however, there was significant correlation between Haller index and postoperative percentage of predicted FVC (r ¼ 0.302 p ¼ 0.008), FEV1 (r ¼ 0.279, p ¼ 0.14), and FEV1% (r ¼ 0.323, p ¼ 0.04). There was a negative correlation between preoperative patient and parent quality-of-life scores and Haller index (r ¼ 0.258 p ¼ 0.026 and r ¼ 0.313 p ¼ 0.007, respectively), but there was no correlation with postoperative scores (r ¼ 0.208, p ¼ 0.085; r ¼ 0.172 p ¼ 0.193). There was no correlation between quality-of-life scores and pulmonary function
Table 3. Preoperative and postoperative Nuss questionnaire patient scores. Question stem
Preoperative
Postoperative
p value
Looks in general (P) How chest looks without shirt (P) Spending rest of life as chest looks now (P) Others make fun of him/her because of chest (P) Avoids doing things (P) Hides chest (P) Bothered because of the way chest looks (P) Feels shy/self-conscious because of chest (P) Feels bad about self (P) Has trouble exercising (PF) Chest causes shortness of breath (PF) Chest causes him/her to be tired (PF) Total
2.18 0.97 1.55 0.65 1.24 0.46 3.58 0.66 3.14 0.85 2.80 1.06 2.56 1.01 3.03 0.92 2.80 0.98 2.76 0.99 2.94 0.92 2.86 0.91 31.65 5.84
3.14 1.14 3.24 0.76 3.42 0.80 3.82 0.45 3.27 0.77 3.72 0.61 3.69 0.57 3.69 0.6 3.33 1.14 3.15 0.83 3.41 0.72 3.46 0.69 41.96 5.29
0.000 0.000 0.000 0.010 0.315 0.000 0.000 0.000 0.002 0.012 0.000 0.000 0.000
P: psychosocial; PF: physical functioning.
Downloaded from aan.sagepub.com at University of Sussex Library on August 30, 2015
4
Asian Cardiovascular & Thoracic Annals 0(0)
Table 4. Preoperative and postoperative Nuss questionnaire parent scores. Question stem
Preoperative
Postoperative
p value
Irritable (P) Frustrated (P) Sad/depressed (P) Restless (P) Isolated (P) Reluctant to be in public wearing bathing suit that would show the chest (P) How often parent is concerned about effects of the deformity on patient’s life (P) Have trouble exercising (PF) Have chest pain (PF) Have shortness of breath (PF) Feel tired (PF) Total
2.93 0.86 2.85 0.48 3 0.82 3.18 0.74 3.32 0.93 2.77 1.19 1.88 0.9 2.82 1 3.32 0.83 3.20 0.73 2.73 0.88 32.06 5.13
3.15 0.86 3.03 0.8 3.35 0.65 3.45 0.56 3.58 0.59 3.58 0.8 3.12 0.88 3.10 0.81 3.35 0.75 3.55 0.67 3.20 0.84 36.46 4.98
0.079 0.062 0.003 0.025 0.048 0.000 0.000 0.040 0.808 0.006 0.003 0.000
P: psychosocial; PF: physical functioning.
test results. There was also no relationship between a symmetric deformity and pulmonary function tests or quality-of-life scores. When a 3.25 cut-off level of the Haller index was considered, postoperative patient and parent total quality-of-life scores were lower (p ¼ 0.011 and p ¼ 0.015, respectively). The preoperative levels were also lower with weak significance (p ¼ 0.023; p ¼ 0.059). Postoperative pulmonary function results in patients with a higher Haller index were also significantly different: percentage of predicted FVC (p ¼ 0.019), FEV1 (p ¼ 0.04), and FEV1% (p ¼ 0.008). When sex was considered as a factor, there was no relationship with pulmonary function tests or quality-of-life scores.
Discussion When the demographic characteristics of the patients with PE in our series were considered, there were more symmetric cases than in previously reported studies. The most commonly encountered associated deformity, scoliosis,2 was also the leading deformity in the present study. Sigalet and colleagues12 reported that after MIRPE, there was a significant decrease in FVC and vital capacity, but FEV1 was not changed in the following 3 months. These results are in agreement with our findings. In the present study, FVC was decreased after 6 months, and there was no change in FEV, but FEV1/ FVC was increased. These findings indicate a restrictive pattern of lung function. Borowitz and colleagues13 reported that there was no significant change in pulmonary function tests at 6 and 12 months after the surgery. Lawson and colleagues14 found significant improvements in pulmonary function tests after removal of the bar, with subjective improvements in patients’ symptoms. Neviere and colleagues15 also reported an early postoperative decrease in pulmonary
function tests with improvement on long-term followup. Castellani and colleagues16 found similar results in the preoperative period and after removal of the bar. In a study by Johnson and colleagues,17 there was no improvement in exercise tolerance after surgical intervention. Kubiak and colleagues18 noted improved pulmonary function parameters after MIRPE, however, they did not suggested routine preoperative and postoperative pulmonary function tests. In a multicenter study, it was shown that the higher the Haller index in patients with PE, the worse the pulmonary function test results.19 Swanson and colleagues20 concluded that a Haller index above 3.6 was linked to pulmonary dysfunction. We found no significant relationship between Haller index and pulmonary function tests preoperatively, but there was negative correlation in the postoperative period. Quality of life of the patients were improved postoperatively as previously reported by Kuru and colleagues.1 We concluded the MIRPE has positive impact on PE patients’ quality of life but a negative impact on FVC 6 months after the bar placement. This can be interpreted as a limitation and restriction in lung capacity in the early postoperative period. Followup studies are needed to determine the long-term changes in pulmonary function after the operation, including test results under physical (exercise) stress. Funding The author(s) received no financial support for the research, authorship, and/or publication of this article.
Declaration of conflicting interests The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Downloaded from aan.sagepub.com at University of Sussex Library on August 30, 2015
Kuru et al.
5
References 1. Kuru P, Bostanci K, Ermerak NO, Bahadir AT, Afacan C and Yuksel M. Quality of life improves after minimally invasive repair of pectus excavatum. Asian Cardiovasc Thorac Ann 2015; 48: 112–119. 2. Waters P, Welch K, Micheli LJ, Shamberger R and Hall JE. Scoliosis in children with pectus excavatum and pectus carinatum. J Pediatr Orthop 1989; 9: 551–556. 3. Nuss D, Kelly RE Jr, Croitoru DP and Katz ME. A 10year review of a minimally invasive technique for the correction of pectus excavatum. J Pediatr Surg 1998; 33: 545–552. 4. Grozavu C, Ilias¸ M, Marin D, Pantile D, Dabelea C and Augustin T. Minimally invasive repair for pectus excavatum—aesthetic and/or functional? Chirurgia (Bucur) 2013; 108: 70–78. 5. Malek MH, Berger DE, Marelich WD, Coburn JW, Beck TW and Housh TJ. Pulmonary function following surgical repair of pectus excavatum: a meta-analysis. Eur J Cardiothorac Surg 2006; 30: 637–643. 6. Aronson DC, Bosgraaf RP, Merz EM, van Steenwijk RP, van Aalderen WM and van Baren R. Lung function after the minimal invasive pectus excavatum repair (Nuss procedure). World J Surg 2007; 31: 1518–1522. 7. O’Keefe J, Byrne R, Montgomery M, Harder J, Roberts D and Sigalet DL. Longer term effects of closed repair of pectus excavatum on cardiopulmonary status. J Pediatr Surg 2013; 48: 1049–1054. 8. Sigalet DL, Montgomery M, Harder J, Wong V, Kravarusic D and Alassiri A. Long term cardiopulmonary effects of closed repair of pectus excavatum. Pediatr Surg Int 2007; 23: 493–497. 9. Jayaramakrishnan K, Wotton R, Bradley A and Naidu B. Does repair of pectus excavatum improve cardiopulmonary function? Interact Cardiovasc Thorac Surg 2013; 16: 865–870. 10. Bahadir AT, Kuru P, Afacan C, Ermerak NO, Bostanci K and Yuksel M. Validity and reliability of the Turkish
11.
12.
13.
14.
15.
16.
17.
18.
19.
20.
version of the Nuss questionnaire modified for adults. Korean J Thorac Cardiovasc Surg 2015; 48: 112–119. Ha HI, Seo JB, Lee SH, Kang JW, Goo HW, Lim TH and Shin MJ. Imaging of Marfan syndrome: multisystemic manifestations. Radiographics 2007; 27: 989–1004. Sigalet DL, Montgomery M and Harder J. Cardiopulmonary effects of closed repair of pectus excavatum. J Pediatr Surg 2013; 38: 380–385. Borowitz D, Zallen G, Sharp J, et al. Pulmonary function and response to exercise following Nuss repair in patients with pectus excavatum. J Pediatr Surg 2003; 38: 544–547. Lawson ML, Mellins RB, Tabangin M, et al. Impact of pectus excavatum on pulmonary function before and after repair with the Nuss procedure. J Pediatr Surg 2005; 40: 174–180. Neviere R, Montaigne D, Benhamed L, et al. Cardiopulmonary response following surgical repair of pectus excavatum in adult patients. Eur J Cardiothorac Surg 2011; 40: 77–182. Castellani C, Windhaber J, Schober PH and Hoellwarth ME. Exercise performance testing in patients with pectus excavatum before and after Nuss procedure. Pediatr Surg Int 2010; 26: 659–663. Johnson JN, Hartman TK, Pianosi PT and Driscoll DJ. Cardiorespiratory function after operation for pectus excavatum. J Pediatr 2008; 153: 359–364. Kubiak R, Habelt S, Hammer J, Hacker FM, Mayr J and Bielek J. Pulmonary function following completion of minimally invasive repair for pectus excavatum (MIRPE). Eur J Pediatr Surg 2007; 17: 255–260. Lawson ML, Mellins RB, Paulson JF, Shamberger RC and Oldham K. Increasing severity of pectus excavatum is associated with reduced pulmonary function. J Pediatr 2011; 159: 256–261. Swanson JW, Avansino JR, Phillips GS, et al. Correlating Haller Index and cardiopulmonary disease in pectus excavatum. Am J Surg 2012; 203: 660–664.
Downloaded from aan.sagepub.com at University of Sussex Library on August 30, 2015
How pulmonary function changes after pectus excavatum correction surgery
Asian Cardiovascular & Thoracic Annals 0(0) 1–5 ß The Author(s) 2015 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav DOI: 10.1177/0218492315596464 aan.sagepub.com
Pinar Kuru1, Asli Dudakli2, Hakan Mursaloglu2, Hazal Arikan2, Aysu Oktay2 and Mustafa Yuksel3
Abstract Aim: We aimed to determine the effects of minimally invasive repair of pectus excavatum on pulmonary function and quality of life. Methods: Minimally invasive pectus excavatum repair was undertaken in 80 patients with a mean age of 16.91 4.37 years (range 7–30 years) and a mean Haller index of 4.07 1.39; 85% of the patients were male. They and their parents completed the Nuss Questionnaire Modified for Adults, and pulmonary function tests were performed on the patients before and 6 months after the operation. Results: The mean Nuss score was 31.06 6.78 before the operation and it increased to 37.1 8.31 (p ¼ 0.000) 6 months after the operation. Forced vital capacity decreased from 3.70 1.23 to 3.48 1.03 L (p ¼ 0.05) postoperatively. The percentage of expected forced vital capacity decreased from 83.21% 16.97% to 76.52% 20.98% (p ¼ 0.01). There was no significant change in forced expiratory volume in 1 s. The mean ratio of forced expiratory volume in 1 s to forced vital capacity was 86% preoperatively and it increased to 91% postoperatively (p ¼ 0.000). Conclusions: Minimally invasive pectus excavatum repair has a positive impact on the quality of life of pectus excavatum patients, but a negative impact on forced vital capacity. Follow-up studies are needed to assess the long-term changes in pulmonary function after this operation.
Keywords Body image; Funnel chest; Patient satisfaction; Quality of life; Minimally invasive surgical procedures, Respiratory function tests
Introduction Chest wall deformities are a common type of congenital deformity. Anterior chest wall deformities become prominent during childhood and may cause psychologic, orthopedic, and physiological problems, according to the severity. Medium and severe deformities can be corrected by surgical intervention during childhood, with good cosmetic, orthopedic, and functional outcomes.1 Pectus excavatum (PE) is the most common congenital chest wall deformity with an incidence of 1/300–400 live births. It is characterized by a posterior depression of the sternum and costal cartilages, however, the first the costal cartilages are usually in the normal position. PE is seen more often in Caucasians and males, and the severity varies. The asymmetric form is the most common, and the right side is generally more depressed, sometimes with sternal rotation.
PE usually becomes more prominent during the growth spurt, but in some cases, it becomes less severe. Scoliosis is a frequently associated anomaly, seen in a quarter of cases.2 PE and scoliosis are often found in patients with Marfan syndrome. Congenital cardiac anomalies and asthma may accompany PE. Although 1 Department of Neurology, Northwestern University Feinberg School of Medicine, Chicago, IL, USA 2 Marmara University School of Medicine, Istanbul, Turkey 3 Department of Thoracic Surgery, Marmara University School of Medicine, Istanbul, Turkey
Corresponding author: Pinar Kuru, MD, Northwestern University Feinberg School of Medicine, Department of Neurology, 303 E Chicago Ave. Ward Building 10-015., Chicago, IL, USA. Email: [email protected]
Downloaded from aan.sagepub.com at University of Sussex Library on August 30, 2015
2
Asian Cardiovascular & Thoracic Annals 0(0)
it is often asymptomatic, it can cause shortness of breath, easy fatigue, chest pain, and palpitation. Usually, there is no objective pathology in pulmonary function tests and cardiac evaluation, so this deformity is accepted as a cosmetic and psychosocial problem. In medium and severe forms of PE, the lungs and heart are displaced, resulting in exercise intolerance. These patients have round shoulders and a typical pectus posture. In severe PE, cardiorespiratory function problems can arise, with mild to moderate exercise intolerance, chest pain with effort, and recurrent respiratory tract infections.3 The aim of the surgery is to elevate the depressed part of the chest and return the sternum to the normal position. The mechanism depends on chronic pressure applied to the sternum with the aid of a shaped nickel-steel bar inserted retrosternally for a period of time. Minimally invasive repair of pectus excavatum (MIRPE) was described in 1987. To facilitate the technique, special equipment and a stabilizer to inhibit bar rotation were developed. Pulmonary function tests are commonly used to evaluate respiratory diseases, and spirometric tests assess pulmonary function. In a recent meta-analysis, it was reported that surgical correction of PE improved cardiovascular function, but pulmonary function test results did not change.4 Another study on pulmonary function reported similar results.5 However, in some case series, postoperative improvements in pulmonary function were noted.6,7 In another meta-analysis, early postoperative results indicated decreased pulmonary function, but amelioration was predominant in long-term follow-up.8 In a study comparing pulmonary outcomes of the Ravitch and Nuss techniques, pulmonary capacity decreased after the Ravitch operation and forced expiratory volume in the first second of exhalation (FEV1) was increased after the Nuss operation.9 In the present study, we aimed to investigate the effects of MIRPE on pulmonary function and quality of life in patients with PE.
Patients and methods This prospective follow-up study included 80 patients with PE who underwent MIRPE by the same surgeon (MY) between 2012 and 2014 in the Department of Thoracic Surgery, Marmara University Hospital. Cosmetic complaints were the main reason for attending the thoracic surgery outpatient clinic. A complete clinical history was taken, a physical examination was performed, including documenting photographs, radiographic imaging. Pulmonary function and laboratory tests were carried out before and 6 months after the operation in all patients. We also obtained a postoperative radiograph of the chest to confirm the placement of
the bar and to detect pneumothorax. The sample size calculated according to primary outcome and the power of the study was 93.9% with 80 patients. Marmara University School of Medicine Clinical Research Ethics Committee approved this study, and informed consent was obtained from each participant. MIRPE was performed under general anesthesia. To insert a curved bar under the sternum, a lateral incision was made on both sides of the chest. The bar (Tasarimmed; Medical Devices Manufacturing and Marketing, Inc., Istanbul, Turkey) was individually curved for each patient and used to elevate the depression of the chest wall. It was fixed to the ribs on both sides, and the incisions were closed primarily. To stabilize and fix the bar to the rib, a steel plate was used, which was not visible from the outside. At the end of the treatment, the bar was removed in a short surgical procedure. The Pectus Excavatum Evaluation Questionnaire (Nuss questionnaire; NQmA) is a disease-specific quality-of-life assessment tool for children with PE and their parents. The validity and reliability of the Turkish NQmA was confirmed by Bahadir and colleagues.10 Although the original parent form contained 13 items, as a result of the reliability and validity analyses, two items were excluded. The patient version of the NQmA includes 12 items and the parent version includes 11 items. The score for each items ranges from 1 to 4. Possible minimum and maximum scores are 12–48 for the patient form, and 11–44 for the parent form; higher scores indicate a better quality of life. The Turkish version of the NQmA has physical and psychosocial components. The patients and most of their parents completed the questionnaire before and 6 months after the surgery. Anteroposterior and lateral radiographs were used to calculate the Haller (pectus) index as a quantitative indicator of the severity of the chest wall deformity. The Haller index was calculated by dividing the width of the chest wall at its widest point by the distance between the posterior surface of the sternum and the anterior surface of the spine. A Haller index greater than 3.25 is one of the indications for surgery.11 The surgical indication in our study was mainly decided according to physical and laboratory examinations, age of the patient, and expectations. Pulmonary function tests were performed in the same laboratory on all patients before and 6 months after the surgery. Forced vital capacity (FVC), FEV1, percentage of predicted FEV1 (FEV1%), and the FEV1/FVC ratio were recorded. For descriptive data, mean standard deviation and percentages were used. The NQmA patient and parent forms and pulmonary function test results were analyzed using the dependent-samples t test to determine
Downloaded from aan.sagepub.com at University of Sussex Library on August 30, 2015
Kuru et al.
3
Table 1. Characteristics of 80 patients who underwent repair of pectus excavatum. Variable
No. of patients
Male Symmetric Associated anomaly or disease Scoliosis Haller index > 3.25 Age at operation (years) [range] Total hospitalization (days) [range] Postoperative hospital stay (days) [range] Haller index [range] Operative time (min) [range]
68 (85%) 53 (66.3) 32 (40%) 9 (11.3%) 58 (72.5%) 16.91 4.37 [7–30] 6.61 2.51 [3–18] 4.58 1.54 [2–10] 4.07 1.39 [1.29–9.5] 67.12 22.93 [30–150]
Table 2. Preoperative and postoperative pulmonary function test results in 80 patients who underwent repair of pectus excavatum. Test
Preoperative
Postoperative
p value
FVC (L) FVC% FEV1 FEV1% FEV1/FVC
3.7 1.23 83.21 16.97 3.12 1.04 83.67 19.55 0.86 0.14
3.48 1.03 76.52 20.98 3.19 1 83.33 22.67 0.91 0.07
0.05 0.01 0.53 0.89 0.00
FEV1: forced expiratory volume in 1st s; FEV1%: percentage of predicted forced expiratory volume in 1st s; FVC: forced vital capacity; FVC%: percentage of predicted forced vital capacity.
the statistical significance of the data before and after the operation. Pearson’s correlation coefficient and the Mann-Whitney U test were used to determine the relationship between quality-of-life scores and the demographic variables. In all analysis, p < 0.05 was accepted as significant.
Results The characteristics of the patients are given in Table 1. The majority of these patients were male (85%), 66.3% had a symmetric deformity, and 40% had an associated anomaly. The mean NQmA score before the operation was 31.06 6.78 and it increased to 37.1 8.31 (p ¼ 0.000) at 6 months after the operation. FVC and percentage of predicted FVC decreased postoperatively (Table 2). There was no significant change in FEV1 or FEV1%, but the mean FEV1/FVC ratio increased significantly postoperatively. Quality-of-life scores in both the patient and parent forms improved at 6 months postoperatively (Table 3, Table 4). There was no correlation between Haller index and preoperative pulmonary function tests, however, there was significant correlation between Haller index and postoperative percentage of predicted FVC (r ¼ 0.302 p ¼ 0.008), FEV1 (r ¼ 0.279, p ¼ 0.14), and FEV1% (r ¼ 0.323, p ¼ 0.04). There was a negative correlation between preoperative patient and parent quality-of-life scores and Haller index (r ¼ 0.258 p ¼ 0.026 and r ¼ 0.313 p ¼ 0.007, respectively), but there was no correlation with postoperative scores (r ¼ 0.208, p ¼ 0.085; r ¼ 0.172 p ¼ 0.193). There was no correlation between quality-of-life scores and pulmonary function
Table 3. Preoperative and postoperative Nuss questionnaire patient scores. Question stem
Preoperative
Postoperative
p value
Looks in general (P) How chest looks without shirt (P) Spending rest of life as chest looks now (P) Others make fun of him/her because of chest (P) Avoids doing things (P) Hides chest (P) Bothered because of the way chest looks (P) Feels shy/self-conscious because of chest (P) Feels bad about self (P) Has trouble exercising (PF) Chest causes shortness of breath (PF) Chest causes him/her to be tired (PF) Total
2.18 0.97 1.55 0.65 1.24 0.46 3.58 0.66 3.14 0.85 2.80 1.06 2.56 1.01 3.03 0.92 2.80 0.98 2.76 0.99 2.94 0.92 2.86 0.91 31.65 5.84
3.14 1.14 3.24 0.76 3.42 0.80 3.82 0.45 3.27 0.77 3.72 0.61 3.69 0.57 3.69 0.6 3.33 1.14 3.15 0.83 3.41 0.72 3.46 0.69 41.96 5.29
0.000 0.000 0.000 0.010 0.315 0.000 0.000 0.000 0.002 0.012 0.000 0.000 0.000
P: psychosocial; PF: physical functioning.
Downloaded from aan.sagepub.com at University of Sussex Library on August 30, 2015
4
Asian Cardiovascular & Thoracic Annals 0(0)
Table 4. Preoperative and postoperative Nuss questionnaire parent scores. Question stem
Preoperative
Postoperative
p value
Irritable (P) Frustrated (P) Sad/depressed (P) Restless (P) Isolated (P) Reluctant to be in public wearing bathing suit that would show the chest (P) How often parent is concerned about effects of the deformity on patient’s life (P) Have trouble exercising (PF) Have chest pain (PF) Have shortness of breath (PF) Feel tired (PF) Total
2.93 0.86 2.85 0.48 3 0.82 3.18 0.74 3.32 0.93 2.77 1.19 1.88 0.9 2.82 1 3.32 0.83 3.20 0.73 2.73 0.88 32.06 5.13
3.15 0.86 3.03 0.8 3.35 0.65 3.45 0.56 3.58 0.59 3.58 0.8 3.12 0.88 3.10 0.81 3.35 0.75 3.55 0.67 3.20 0.84 36.46 4.98
0.079 0.062 0.003 0.025 0.048 0.000 0.000 0.040 0.808 0.006 0.003 0.000
P: psychosocial; PF: physical functioning.
test results. There was also no relationship between a symmetric deformity and pulmonary function tests or quality-of-life scores. When a 3.25 cut-off level of the Haller index was considered, postoperative patient and parent total quality-of-life scores were lower (p ¼ 0.011 and p ¼ 0.015, respectively). The preoperative levels were also lower with weak significance (p ¼ 0.023; p ¼ 0.059). Postoperative pulmonary function results in patients with a higher Haller index were also significantly different: percentage of predicted FVC (p ¼ 0.019), FEV1 (p ¼ 0.04), and FEV1% (p ¼ 0.008). When sex was considered as a factor, there was no relationship with pulmonary function tests or quality-of-life scores.
Discussion When the demographic characteristics of the patients with PE in our series were considered, there were more symmetric cases than in previously reported studies. The most commonly encountered associated deformity, scoliosis,2 was also the leading deformity in the present study. Sigalet and colleagues12 reported that after MIRPE, there was a significant decrease in FVC and vital capacity, but FEV1 was not changed in the following 3 months. These results are in agreement with our findings. In the present study, FVC was decreased after 6 months, and there was no change in FEV, but FEV1/ FVC was increased. These findings indicate a restrictive pattern of lung function. Borowitz and colleagues13 reported that there was no significant change in pulmonary function tests at 6 and 12 months after the surgery. Lawson and colleagues14 found significant improvements in pulmonary function tests after removal of the bar, with subjective improvements in patients’ symptoms. Neviere and colleagues15 also reported an early postoperative decrease in pulmonary
function tests with improvement on long-term followup. Castellani and colleagues16 found similar results in the preoperative period and after removal of the bar. In a study by Johnson and colleagues,17 there was no improvement in exercise tolerance after surgical intervention. Kubiak and colleagues18 noted improved pulmonary function parameters after MIRPE, however, they did not suggested routine preoperative and postoperative pulmonary function tests. In a multicenter study, it was shown that the higher the Haller index in patients with PE, the worse the pulmonary function test results.19 Swanson and colleagues20 concluded that a Haller index above 3.6 was linked to pulmonary dysfunction. We found no significant relationship between Haller index and pulmonary function tests preoperatively, but there was negative correlation in the postoperative period. Quality of life of the patients were improved postoperatively as previously reported by Kuru and colleagues.1 We concluded the MIRPE has positive impact on PE patients’ quality of life but a negative impact on FVC 6 months after the bar placement. This can be interpreted as a limitation and restriction in lung capacity in the early postoperative period. Followup studies are needed to determine the long-term changes in pulmonary function after the operation, including test results under physical (exercise) stress. Funding The author(s) received no financial support for the research, authorship, and/or publication of this article.
Declaration of conflicting interests The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Downloaded from aan.sagepub.com at University of Sussex Library on August 30, 2015
Kuru et al.
5
References 1. Kuru P, Bostanci K, Ermerak NO, Bahadir AT, Afacan C and Yuksel M. Quality of life improves after minimally invasive repair of pectus excavatum. Asian Cardiovasc Thorac Ann 2015; 48: 112–119. 2. Waters P, Welch K, Micheli LJ, Shamberger R and Hall JE. Scoliosis in children with pectus excavatum and pectus carinatum. J Pediatr Orthop 1989; 9: 551–556. 3. Nuss D, Kelly RE Jr, Croitoru DP and Katz ME. A 10year review of a minimally invasive technique for the correction of pectus excavatum. J Pediatr Surg 1998; 33: 545–552. 4. Grozavu C, Ilias¸ M, Marin D, Pantile D, Dabelea C and Augustin T. Minimally invasive repair for pectus excavatum—aesthetic and/or functional? Chirurgia (Bucur) 2013; 108: 70–78. 5. Malek MH, Berger DE, Marelich WD, Coburn JW, Beck TW and Housh TJ. Pulmonary function following surgical repair of pectus excavatum: a meta-analysis. Eur J Cardiothorac Surg 2006; 30: 637–643. 6. Aronson DC, Bosgraaf RP, Merz EM, van Steenwijk RP, van Aalderen WM and van Baren R. Lung function after the minimal invasive pectus excavatum repair (Nuss procedure). World J Surg 2007; 31: 1518–1522. 7. O’Keefe J, Byrne R, Montgomery M, Harder J, Roberts D and Sigalet DL. Longer term effects of closed repair of pectus excavatum on cardiopulmonary status. J Pediatr Surg 2013; 48: 1049–1054. 8. Sigalet DL, Montgomery M, Harder J, Wong V, Kravarusic D and Alassiri A. Long term cardiopulmonary effects of closed repair of pectus excavatum. Pediatr Surg Int 2007; 23: 493–497. 9. Jayaramakrishnan K, Wotton R, Bradley A and Naidu B. Does repair of pectus excavatum improve cardiopulmonary function? Interact Cardiovasc Thorac Surg 2013; 16: 865–870. 10. Bahadir AT, Kuru P, Afacan C, Ermerak NO, Bostanci K and Yuksel M. Validity and reliability of the Turkish
11.
12.
13.
14.
15.
16.
17.
18.
19.
20.
version of the Nuss questionnaire modified for adults. Korean J Thorac Cardiovasc Surg 2015; 48: 112–119. Ha HI, Seo JB, Lee SH, Kang JW, Goo HW, Lim TH and Shin MJ. Imaging of Marfan syndrome: multisystemic manifestations. Radiographics 2007; 27: 989–1004. Sigalet DL, Montgomery M and Harder J. Cardiopulmonary effects of closed repair of pectus excavatum. J Pediatr Surg 2013; 38: 380–385. Borowitz D, Zallen G, Sharp J, et al. Pulmonary function and response to exercise following Nuss repair in patients with pectus excavatum. J Pediatr Surg 2003; 38: 544–547. Lawson ML, Mellins RB, Tabangin M, et al. Impact of pectus excavatum on pulmonary function before and after repair with the Nuss procedure. J Pediatr Surg 2005; 40: 174–180. Neviere R, Montaigne D, Benhamed L, et al. Cardiopulmonary response following surgical repair of pectus excavatum in adult patients. Eur J Cardiothorac Surg 2011; 40: 77–182. Castellani C, Windhaber J, Schober PH and Hoellwarth ME. Exercise performance testing in patients with pectus excavatum before and after Nuss procedure. Pediatr Surg Int 2010; 26: 659–663. Johnson JN, Hartman TK, Pianosi PT and Driscoll DJ. Cardiorespiratory function after operation for pectus excavatum. J Pediatr 2008; 153: 359–364. Kubiak R, Habelt S, Hammer J, Hacker FM, Mayr J and Bielek J. Pulmonary function following completion of minimally invasive repair for pectus excavatum (MIRPE). Eur J Pediatr Surg 2007; 17: 255–260. Lawson ML, Mellins RB, Paulson JF, Shamberger RC and Oldham K. Increasing severity of pectus excavatum is associated with reduced pulmonary function. J Pediatr 2011; 159: 256–261. Swanson JW, Avansino JR, Phillips GS, et al. Correlating Haller Index and cardiopulmonary disease in pectus excavatum. Am J Surg 2012; 203: 660–664.
Downloaded from aan.sagepub.com at University of Sussex Library on August 30, 2015
