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Ann Thorac Surg 2005;79:308-312
© 2005 The Society of Thoracic Surgeons


Original article: General thoracic

Surgical Treatment of Diaphragmatic Eventration Using Video-Assisted Thoracic Surgery: A Prospective Study

Jérôme Mouroux, MD, Nicolas Venissac, MD, Francesco Leo, MD, Marco Alifano, MD*, Françoise Guillot, MD

Service de Chirurgie Thoracique, CHU de Nice, Hôpital Pasteur, Nice, France

Accepted for publication June 11, 2004.

* Address reprint requests to Dr Alifano, Chirurgia Toracica, Ospedale Maggiore, Largo B. Nigrisoli, 2, 40100 Bologna, Italy (E-mail: marcoalifano{at}yahoo.com).


    Abstract
 Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
 References
 
BACKGROUND: This study seeks to evaluate results regarding the repair of diaphragmatic eventration using video-assisted thoracic surgery (VATS).

METHODS: We performed a prospective observational study including patients referred to us for surgical treatment of diaphragmatic eventration during a 12-year period. Clinical, radiologic, and functional data were prospectively recorded. VATS was performed with two thoracoports and a 4-cm mini-thoracotomy. Diaphragmatic plication was performed using two nonresorbable running sutures from periphery to the cardio-phrenic angle. Follow-up data (clinical examination, chest roentgenogram, lung function tests at 3, 6, 12 months, and annually thereafter) were also prospectively recorded.

RESULTS: Twelve patients (4 male adults, mean age 57.7 ± 14.8 years) were operated on between 1992 and 2003. The left side was involved in 8 patients and the mean height of diaphragm elevation was 7.5 ± 1.8 cm. All patients experienced symptoms related to the disease; in 2 patients the operation was carried out to achieve weaning from mechanical ventilation. The etiologic mechanism could be identified in 11 out of 12 patients (trauma, n = 9; Charcot-Marie disease, n = 1; calcified para-aortic nodes, n = 1). Mean operative time, drainage output, and hospital stay were 77 ± 15 minutes, 0.8 ± 04 L, and 3.4 ± 0.7 days, respectively. No mortality was observed; 1 patient experienced postoperative pneumonia, which was treated using antibiotics. All patients experienced amelioration of symptoms and long-term lung function tests revealed a marked improvement of both the forced volume capacity and the forced expiratory volume at 1 second. No relapses were observed at follow-up chest roentgenogram.

CONCLUSIONS: Treatment using VATS is a safe and effective alternative to conventional surgery. Functional improvement persists at long-term follow-up.


    Introduction
 Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
 References
 
Diaphragmatic eventration is a rare anomaly defined by a permanent elevation of a hemidiaphragm without defects; the muscular insertions are normal, the normal apertures are sealed, and there is no interruption in the pleural or peritoneal layers. The terms eventration and paralysis are often confused: paralysis may be the instigator of eventration because of degenerative muscular changes after denervation, whereas eventration is not always associated with paralysis. Nevertheless both eventration and paralysis perpetuate the same consequences.

The incidence of diaphragmatic eventration is difficult to estimate. Christensen and associates identified 38 patients among 107,778 examined adults [1]. The first patient diagnosed with diaphragmatic eventration was reported by Petit in 1774, whereas Morrison published the first surgical repair in 1923 [2]. Because of this initial description of repair, several articles focused on the indication with regard to surgery, operative techniques, and results [3, 4]. Different kinds of intervention require a standard thoracotomy, which probably represents an excessively invasive operative approach for a functional surgery. For this reason we developed a minimally invasive technique and published the preliminary results of a prospective study in 1996 [5]. This study was continued and we herein report the results of a 12-year experience using the technique indicating the long-term functional results.


    Patients and Methods
 Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
 References
 
Patients
Between 1992 and 2003 12 patients were operated on for diaphragmatic eventration according to the technique of diaphragmatic plication developed by one of us (JM) and performed by video-assisted thoracic surgery (VATS) [5]. The first 3 patients in this series constituted the study population of our previous work [5].

The following clinical data were prospectively collected: age, sex, clinical history, symptoms, delay between diagnosis and treatment, side (location) of disease, degree of diaphragmatic elevation (Fig 1), presence of a "fixed" mediastinal shift (shift evident on both inspiratory and expiratory chest roentgenogram films), results of etiologic investigations, lung function tests (spirometry and arterial gas analysis), operative findings and procedures, duration of chest drainage, postoperative complications, and total hospital stay. With respect to etiologic investigations, a thorough history and physical examination were always performed. If available, previous roentgenogram films were reviewed. Cervico-thoracic and upper abdominal computed tomography (CT) scans were always carried out. Several patients underwent magnetic resonance imaging and/or phrenic electromyography recommended by a referring physician before hospitalization. A "Sniff test" was not performed because, in our opinion, the treatment will not change in the presence or absence of diaphragmatic paralysis. Postoperative follow-up included physical examination, chest roentgenogram, and spirometry at 3, 6, and 12 months postoperatively and annually thereafter.



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Fig 1. Left diaphragmatic eventration on inspiratory roentgenogram film. The two horizontal lines allow calculation of elevation, which is estimated by the length of the double-headed arrow.

 
Surgical Technique
Operations were executed according to a previously published technique [5]. General anesthesia with double-lumen intubation was employed. A naso-gastric tube was inserted to completely empty the stomach. The patient was positioned in the full lateral position indicated for standard postero-lateral thoracotomy. Two thoracoports (10 mm instruments were employed at the beginning of our experience, whereas 5 mm ports are currently used) were inserted in the fifth intercostals space on the posterior axillary line (for the camera) and on the mammary line (for the grasper), respectively. A 4 or 5 cm minithoracotomy was performed in the ninth intercostals space on the posterior axillary line. Rib retraction was generally not necessary and conventional surgical instruments were introduced through the thoracotomy.

An endoscopic Duval grasper introduced through the anterior thoracoport was used to grasp and invaginate the apex of the eventration downward into the abdomen, thus creating a transverse fold from the periphery to the cardio-phrenic angle behind the phrenic nerve. This fold is closed first using a suture line of nonresorbable material (Surgipro 3.5; Tyco Healthcare France SA, Plaisir, France) beginning at the periphery of the diaphragm closest to minithoracotomy. The first stitch was tied and the free end was held with forceps. A superficial continuous suture was performed to avoid injury to the abdominal organs. Once at cardiophrenic angle, the suture was drawn tight, while the grasper used to push the diaphragm downward was removed. A row of return stitches was created along the same axis and the suture was tied with the free end of the first knot. During the placement of these return stitches, the assistant kept the suture material taut using forceps introduced through the posterior thoracoport. The tension applied in this manner facilitates grasping of the edges of the fold to be sutured.

This first back-and-forth series of continuous sutures allows for maintenance of the excess of diaphragm within the abdomen and care was taken to avoid applying tension to this first series of sutures. A second back-and-forth series of continuous sutures was carried out similarly, thus burying the first series of suture lines: stitches are inserted through a more peripheral portion of diaphragm to obtain the desired tension of the diaphragmatic dome. Schematic display of the technique is provided in Fig 1 of our previous paper [5].

Simple running sutures are employed in all the steps of the repair. Pledgets are never used. Chest drainage is achieved using a single chest tube introduced through the anterior thoracoport.

Data Analysis
Data are expressed as means ± standard deviation. Two-tailed Student's t test was employed to compare preoperative and postoperative lung function tests. A value of p less than 0.05 was considered statistically significant.


    Results
 Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
 References
 
Twelve patients with diaphragmatic eventration were operated on during the period of the study. The patients consisted of 4 adult males and 8 adult females. Their mean age was 57.7 ± 14.8 years. All of the patients experienced symptoms related to the disease: dyspnea (n = 12), palpitations (n = 4), chest pain (n = 3), dyspepsia (n = 2), and recurrent pneumonia (n = 1). Diaphragmatic eventration was left-sided in 8 patients and the mean height of elevation was 7.5 ± 1.8 cm. "Fixed" mediastinal shift was observed in 5 patients and all of these patients experienced palpitations.

Six patients exhibited a history of blunt thoracic trauma and 1 patient had suffered a thoracic stab wound with section of the phrenic nerve. In 2 other patients phrenic nerve palsy represented a surgical complication (1 patient underwent surgery for thoracic outlet syndrome and 1 patient underwent neck lymph node dissection). One patient with a history of pulmonary tuberculosis exhibited calcified para-aortic nodes (Fig 2), whereas another patient exhibited Charcot-Marie disease. Overall an etiologic mechanism could be identified in 11 out of 12 patients.



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Fig 2. Thoracic computed tomography scan of the same patient indicated in Fig 1. Diaphragmatic eventration was evident for 20 years. This 63-year-old patient received a diagnosis of pulmonary tuberculosis 43 years previously. Calcified para-aortic nodes suggest an involvement of the phrenic nerve at this level.

 
All of the patients exhibited a moderate hypoxemia (mean PaO2 = 73.8 ± 7.2 mm Hg) and impairment of forced expiratory volume at 1 second (FEV1) (mean value 57% ± 12% of predicted) and forced volume capacity (FVC) (63% ± 12% of predicted). In 2 of the last 3 patients spirometry was performed in both the standing and lying position, however the third patient was examined in the sitting position because of an inability to maintain the lying position. In all of these patients there was a decrease in both FEV1 and FVC values with the patient in the lying or in the sitting position (Table 1).


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Table 1. Spirometry in the Standing and Lying (Patients 1 and 2) or Sitting (Patient 3) Positions for the Last 3 Patients of the Study
 
Two patients (59 and 80 years old, respectively) experienced decompensation of respiratory insufficiency requiring mechanical ventilation and they were both operated on to allow weaning. One of them experienced left-sided eventration of an unknown etiology. This patient had been admitted for infectious exacerbation (pneumonia) of respiratory insufficiency and required mechanical ventilation. Despite improvement of the pneumonia, weaning was not possible and diaphragmatic plication was planned. The other patient experienced right-sided eventration secondary to a blunt chest trauma sustained 10 years previously. A second blunt trauma (fall from stairs) caused rib fractures and precipitated respiratory failure requiring mechanical ventilation. Tracheostomy was performed to facilitate weaning, but it was complicated by tracheo-esophageal fistula. Staged diaphragmatic plication (to allow weaning) and repair of fistula were planned.

All patients underwent surgery. Rib retraction was never necessary in this series. Mean operative time, drainage output, and hospital stay were 77 ± 15 minutes, 0.8 ± 04 L, and 3.4 ± 0.7 days, respectively. A single patient experienced contralateral lobar pneumonia that was treated successfully with antibiotics. Weaning from mechanical ventilation was accomplished on postoperative days 4 and 6 in the 2 patients who underwent diaphragmatic plication for respiratory failure. The patient with tracheo-esophageal fistula could be successfully operated on (esophageal suture and tracheal sleeve resection) after 4 weeks.

When the study was completed (January 2004) mean follow-up was 64.4 ± 46 months. Six patients had been followed-up for more than 5 years. Shortly after the operation all of the patients experienced complete disappearance of symptoms and no radiologic relapse was observed. A significant improvement in both FEV1 (Fig 3) and FVC was observed at late spirometry (1 year, n = 10; 5 years, n = 6), whereas no significant change in arterial gas analysis was determined (Tables 2 and 3). No late recurrence was recorded.



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Fig 3. Preoperative and postoperative forced expiratory volume at 1 second (FEV1) values in 10 patients. Two patients were operated on less than 1 year before the completion of the study. *Identifies a patient treated with adjuvant chest radiotherapy for a thymoma diagnosed 3 years after surgical treatment of eventration. (%th = percent of predicted; S = surgery; t = time.)

 

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Table 2. Short-Term Functional Follow-Up: Preoperative and Postoperative (1 Year) Arterial Gas Analysis and Spirometry Values in 10 Patients (Two Patients Were Operated On Less Than 1 Year Before the Completion of the Study)
 

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Table 3. Long-Term Functional Follow-Up: Preoperative and Postoperative (5 Years) Arterial Gas Analysis and Spirometry Values in 6 Patients
 

    Comment
 Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
 References
 
Diaphragmatic eventration represents the atrophy and progressive distension of the diaphragmatic muscle. In adulthood both the presence of symptoms and the functional impairment are largely variable. Dyspnea is the most frequent symptom and is typically worsened by the lying position [6]. This position is also associated with a worsening with regard to respiratory function testing [6], as observed in all 3 patients in our series who underwent this functional assessment. Other clinical presentations included chest pain (either spontaneous or provoked by flexion), recurrent respiratory infections, palpitations caused by cardiac arrhythmias, and dyspepsia [7]. Decompensation of respiratory insufficiency has been reported [8]: in these patients diaphragmatic eventration represents a factor aggravating an underlying disease, as was noted in 2 patients from our series. Diaphragmatic eventration indicates different possible etiologies: trauma, neoplasms, infections, and degenerative diseases. Congenital diaphragmatic eventration is rarely recognized only in adulthood [9–11]. Etiologic investigations must be exhaustive and should take into account either the clinical history or previous chest x-rays. In our opinion cervico-thoracic and upper abdominal CT scans should be performed in every patient to exclude both a neoplastic disease (at cervical, mediastinal, or pulmonary level) involving the phrenic nerve and subdiaphragmatic abnormalities (abscess, Chilaiditi syndrome). Despite exhaustive investigations etiology may remain obscure, although phrenic nerve electromyography indicates its functional impairment.

It is generally believed that there is no indication regarding the surgical treatment of diaphragmatic eventration when the condition is secondary to a neoplastic disease or in the absence of symptoms [12]. In our opinion if a neoplastic origin is clearly excluded by clinical and radiologic data, surgical indication has to be discussed on the basis of clinical presentation and time frame regarding the onset of symptoms. If the patient is symptomatic and diaphragmatic eventration has been recognized for more than 2 years, surgery is generally indicated; the operation has to be planned once clinical conditions have been optimized (eg, respiratory infections treated, overweight issues solved, etc).

If the patient is symptomatic but the diaphragmatic eventration is recent, we advocate a period of observation (18–24 months) before suggesting surgery. During this period physiotherapy has to be performed and possible weight issues (overweight) addressed. In these patients a diaphragmatic electromyography with magnetic stimulation of the phrenic nerve may be suggested: this kind of examination is at least as accurate but less painful than standard electromyography [13]. It is noteworthy that spontaneous resolution of a recent eventration is possible, as we observed this occurrence in 2 patients (not included in the present series) referred to our institution during the study period. Serial chest roentgenogram photos are useful with regard to assessing possible improvements and, in particular, we use the empiric method illustrated in Fig 1 to evaluate evolution. Although this method may underestimate or overestimate the degree of elevation, depending on the affected side, this inconvenience is counterbalanced by its practical advantages.

Finally if the patient experiences no or few symptoms, they should still be strictly followed-up to promptly indicate the necessity for operation in the event of an even slight deterioration in respiratory function. In fact if considerable respiratory impairment is already present, a modest chest trauma or a pulmonary infection may precipitate clinical conditions and require mechanical ventilation (as occurred in 2 patients in our series).

Patients are often referred for surgery when some degree of respiratory insufficiency exists. All of our patients exhibited alterations with regard to the respiratory function test, which were, in some instances, very important. We think that there is no defined degree of alteration with regard to the respiratory function test precluding the possibility of surgery: accurate preoperative preparation provides the possibility of performing the operation with an acceptable operative risk.

Various surgical techniques have been proposed (excision and suture, diaphragmatic plication, prosthesis) to treat diaphragmatic eventration [7]. Diaphragmatic plication through standard thoracotomy is the most frequently employed technique. It carries a low morbidity and no mortality. Graham and associates treated 17 patients using thoracotomy and a functional improvement was still present at long-term follow-up [14]. Similar results were also reported by Ribet and Linder [15].

The technique of diaphragmatic plication may be performed using VATS. Since our initial publication many authors have reported their experience with the same or very similar techniques [16–19]. Van Smith and associates reported successful treatment of a newborn weighing 3 Kg [19].

In our experience diaphragmatic plication using VATS achieved results similar to those obtained using conventional surgery. Unfortunately the rarity of diaphragmatic eventration precludes the possibility of performing randomized studies to assess accurate comparisons. However in our 12-year experience performing the technique, we can verify its simplicity, low morbidity, and effectiveness. Clinical, functional, and radiologic improvement was observed in all of the patients and improvement still persists in all 6 patients with a long-term follow-up. As for conventional surgery, video-assisted diaphragmatic plication carries the risk of late recurrence, thus justifying a long-term survey. In our opinion two technical aspects of our technique would guarantee a safeguard against this risk: the first back-and-forth running suture allows for maintenance of the excess of diaphragm within the abdomen while achieving a favorable distribution of tension, whereas the second running suture involves more peripheral portions of the diaphragm with more resistant tissues and provides the desired tension of the diaphragmatic dome. Thus care should be taken to avoid applying tension to this first series of sutures.

Recent papers have suggested a fully thoracoscopic approach to repair diaphragmatic eventration [16, 18, 20–22]. However when using this approach four thoracoports are generally employed [18, 20–22], two of them often being of large caliber [18, 20]. We currently use a small working incision and two 5 mm ports, which renders our approach to be approximately as invasive as a four-thoracoport thoracoscopy. It is generally recognized that postoperative pain—the most troublesome problem after this type of surgery—mainly depends on the intraoperative injury to intercostals nerves by rib spreading or large caliber thoracoports. Hence as recently indicated [23] when determining the optimal surgical approach for minimal invasive repair of diaphragmatic eventration, the most important aspect is not the kind of incision created, but whether rib spreading can be avoided. Though our technique considers the possibility of applying a rib retractor if poor visualization is obtained, this was never necessary in our series.

We conclude that minimally invasive surgery has probably caused a renewed interest in diaphragmatic eventration and its treatment. The encouraging results observed in this study prompt us to suggest this technique as an alternative to conventional diaphragmatic plication through standard thoracotomy.


    References
 Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
 References
 

  1. Christensen P. Eventration of diaphragm Thorax 1959;14:311-319.
  2. Morrison JMW. Eventration of diaphragm due to unilateral phrenic nerve paralysis Arch Radiol Electrotherap 1923;28:72-75.
  3. Mc Namara JJ, Paulson DL, Urschel HC, Razzuk MA. Eventration of diaphragm Surgery 1968;64:1013-1021.[Medline]
  4. Wright CD, Williams JG, Ogilvie CM, Donnelly RJ. Results of the diaphragm plication for unilateral diaphragmatic paralysis J Thorac Cardiovasc Surg 1985;90:195-198.[Abstract]
  5. Mouroux J, Padovani B, Poirier NC, et al. Technique for the repair of diaphragmatic eventration Ann Thorac Surg 1996;62:905-907.[Abstract/Free Full Text]
  6. Clague HW, Hall DR. Effect of posture on lung volume: airway closure and gas exchange in hemidiaphragmatic paralysis Thorax 1979;34:523-526.[Abstract/Free Full Text]
  7. Dor J, Richelme H, Aubert J, Boyer R. L'éventration diaphragmatiqueJ. Chir 1969;97:399-432.
  8. Watanabe S, Shimokawa S, Fukueda M, et al. Large eventration of diaphragm in a elderly patient treated with emergency plication Ann Thorac Surg 1998;65:1776-1777.[Abstract/Free Full Text]
  9. Deslauriers J. Eventration of diaphragm Chest Surg Clin N Am 1998;8:315-360.[Medline]
  10. Mouroux J, Venissac N, Alifano M. Traitement chirurgical des paralysies et éventrations diaphragmatiques chroniques Rev Mal Respir 1999;16:3S87-8.
  11. Efthimiou J, Butler J, Benson MK, Westaby S. Bilateral diaphragm paralysis after cardiac surgery with topical hypothermia Ann Thorac Surg 1991;52:1005-1008.[Abstract]
  12. Pielher JM, Pairolero PC, Gracey DR, Bernatz PE. Unexplained diaphragmatic paralysis: a harbinger of malignant disease? J Thorac Cardiovasc Surg 1982;84:861-864.[Abstract]
  13. Similowski T, Mehiri S, Duguet A, Straus C, Derenne JP. Comparison of magnetic and electrical phrenic nerve stimulation in assessment of phrenic nerve conduction time J Appl Physiol 1997;82:1190-1199.[Abstract/Free Full Text]
  14. Graham DR, Kaplan D, Evans CC, Hind CRK, Donelly RJ. Diaphragm plication for unilateral diaphragmatic paralysis: a 10-year experience Ann Thorac Surg 1990;49:248-252.[Abstract]
  15. Ribet M, Linder JL. Plication of the diaphragm for unilateral eventration or paralysis Eur J Cardiothorac Surg 1992;6:357-360.[Abstract]
  16. Suzumura Y, Terada Y, Sonobe Met al. A case of unilateral diaphragmatic eventration treated by plication with thoracoscopic surgery Chest 1997;112:530-532.[Abstract/Free Full Text]
  17. Lai DTM, Paterson HS. Mini-thoracotomy for diaphragmatic plication with thoracoscopic assistance Ann Thorac Surg 1999;68:2364-2365.[Abstract/Free Full Text]
  18. Moon SW, Wang YP, Kim YW, Shin SB, Jin W. Thoracoscopic plication of diaphragmatic eventration using endostapplers Ann Thorac Surg 2000;70:299-300.[Abstract/Free Full Text]
  19. Van Smith C, Jacobs JP, Burke RP. Minimally invasive diaphragm plication in a infant Ann Thorac Surg 1998;65:842-844.[Abstract/Free Full Text]
  20. Hwang ZS, Shin JS, Cho YH, Sun K, Lee IS. A simple technique for the thoracoscopic plication of the diaphragm Chest 2003;124:376-378.[Abstract/Free Full Text]
  21. Hines MH. Video-assisted diaphragm plication in children Ann Thorac Surg 2003;76:234-236.[Abstract/Free Full Text]
  22. Abraham MK, Menon SS, Bindumole PS. Thoracoscopic repair of eventration of diaphragm Indian Pediatr 2003;40:1088-1089.[Medline]
  23. Shin JS. Plication for diaphragmatic eventrationA simple technique, not a simple problem. Chest 2004;125:1170-1171.[Free Full Text]



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