economics

Key Points Eventration and permanent phrenic nerve injury are indications for

diaphragmatic plication in symptomatic patients. Thoracotomy, laparotomy, and minimally invasive approaches

have all been described and successfully used. Flag, accordion, and invagination techniques using nonabsorbable

suture have been described. Timing of surgery must allow recovery of reversible phrenic nerve

injury. Unfortunately, outcomes and related measures can be only anec-

dotally described.

symptoms. In 1947, Bisgard3 described precisely the tech-nique of plication employed to treat a 6-week-old baby. Since these fi rst publications, many studies were devoted to this subject in both France (Quenu and Herlemont,4 Perrotin and Moreaux,5 Dor and colleagues6) and the United States (Michelson,7 McNamara and associates8). These studies allowed evaluation of the various surgical techniques (plica-tion by a thoracic or an abdominal approach, with excision followed by suturing), as well as the indications for surgery. The more recent works by Revillon and Fekete,9 Donzeau-Gouche and colleagues,10 Wright and colleagues,11 Graham and colleagues,12 and Ribet and Linder13 confi rmed the improvement of respiratory functions with surgical treat-ment. Plication by the transthoracic approach has gradually replaced the abdominal approach. Since the appearance of video-assisted technology, several authors have reported their experience on plication using this tool. We initially described a technique of VATS in 1996 (Mouroux et al, 1996).14 More recently, Httl and associates15 reported a technique of plica-tion by video-assisted laparoscopy (Httl et al, 2004).

HISTORICAL READINGS

Bisgard JD: Congenital eventration of the diaphragm. J Thorac Surg 16:484-491, 1947.

Donzeau-Gouche, Personne CL, Lechien J, et al: Eventrations diaphrag-matiques de ladultes: A propos de vingt cas. Ann Chir Thor Cardio-vasc 36:87-90, 1982.

Dor J, Richelme H, Aubert J, Boyer R: Lventration diaphragmatique. J Chir 97:399-432, 1969.

Graham DR, Kaplan D, Evans CC, et al: Diaphragm plication for unilateral diaphragmatic paralysis: A 10-year experience. Ann Thorac Surg 49:248-252, 1990.

Httl TP, Wichmann MW, Reichart B, et al: Laparoscopic diaphragmatic plication. Surg Endosc 18:547-551, 2004.

McNamara JJ, Paulson DL, Urschel HC, Razzuk MA: Eventration of diaphragm. Surgery 64:1013-1021, 1968.

Michelson E: Eventration of the diaphragm. Surgery 49:410-420, 1961.Morrison JMW: Eventration of diaphragm due to unilateral phrenic

nerve paralysis. Arch Radiol Electrother 28:72-75, 1923.Mouroux J, Padovani B, Poirier NC, et al: Technique for the repair of

diaphragmatic eventration. Ann Thorac Surg 62:905-907, 1996.Perrotin J, Moreaux J: Chirurgie du Diaphragme VIII. Les ventrations.

Paris, Masson, 1965, pp 221-262.Quenu J, Herlemont P: Du traitement chirurgical de lventration dia-

phragmatique. J Chir 69:101-121, 1953.Revillon Y, Fekete CN: Eventration diaphragmatique chez lenfant. Ann

Chir 36:71-74, 1982.

1431

PLICATION OF THE DIAPHRAGMchapter

119 Jrme MourouxNicolas VenissacFrancesco LeoDaniel PopMarco Alifano

The goal of diaphragmatic plication is tightening and lowering of the diaphragm. It is a corrective surgery from a both mor-phologic and a functional point of view and may be applied to the treatment of diaphragmatic eventration and paralysis. The indications and the possible functional benefi ts have been described by many surgical teams. The procedure is widely performed in pediatric patients and, in particular, in newborns with congenital eventration or acquired diaphrag-matic paralysis. On the other hand, some confusion exists in the classifi cation of diseases causing diaphragmatic elevation in adults that might suitable for surgical treatment. Further-more, the use of thoracotomy (which has become the pre-ferred approach in recent years) for a functional surgery is a deterrent to both patients and physicians. Nevertheless, the possibility of performing the procedure by video-assisted tho-racic surgery (VATS), with the obvious adaptation of the methods of plication to this approach, unquestionably has led to a new interest in these pathologies and their surgical treat-ment. This chapter provides an overview of the diseases involved (classifi cation, epidemiology, etiology, and anatomi-coclinical consequences), as well as surgical techniques, indi-cations, and results of diaphragmatic plication.

HISTORICAL NOTEWood1 is classically credited with having introduced in 1916 the idea of wrinkling the diaphragm in order to reduce the dimensions of the cupola. In 1923, Morrison2 performed the fi rst successful repair of an eventration, and he described the surgical principles that are still used. He plicated the diaphragm of a 10-year-old girl with immediate relief of

Surgical Techniques

Ch119-F06861.indd 1431Ch119-F06861.indd 1431 1/21/2008 3:47:18 PM1/21/2008 3:47:18 PM

Section 6 Diaphragm1432

Ribet M, Linder JL: Plication of the diaphragm for unilateral eventration or paralysis. Eur J Cardiothorac Surg 6:357-360, 1992.

Wood HG: Eventration of the diaphragm. Surg Gynecol Obstet 23:344, 1916.

Wright CD, Williams JG, Ogilvie CM, Donnelly RJ: Results of the diaphragm plication for unilateral diaphragmatic paralysis. J Thorac Cardiovasc Surg 90:195-198, 1985.

CLASSIFICATION AND EPIDEMIOLOGYThe radiologic fi nding of an elevated diaphragm has been named in various ways, with subsequent confusion. The medical debate has further contributed to these ambiguities, since the initial description of diaphragmatic eventration by Jean Louis Petit in 1774.16 Terms such as eventration, relax-ation, paralysis, and hernia have been frequently used as synonyms.7,17,18 Diaphragmatic eventration is an anomaly defi ned by the long-lasting or permanent elevation of an entire hemidiaphragm or a portion of it, without defects. The muscular insertions are normal, the normal apertures are sealed, and there is no interruption in the pleural or perito-neal layer. Eventration can be differentiated from hernia (with or without sac) or from rupture because these other conditions involve loss of continuity of one or more of the layers constituting the diaphragm. According to most authors, only congenital eventration needs to be considered as a disease, whereas all other conditions need to be regarded as a syndrome. On the other hand, the terms eventration and paralysis are often confused because paralysis may be the cause of an abnormal elevation of the diaphragm (with degen-erative changes in the muscular layer), whereas pure eventra-tion is not always associated with paralysis. In spite of several differences, both eventration and paralysis carry the same physiologic consequences and share most symptoms. Although the management of these two conditions may be substantially different, plication may constitute a treatment option in both cases.

FIGURE 119-1 A, Left diaphragmatic eventration with mediastinal contralateral shift. B, Same patient, 2 years after plication of the diaphragm.

A B

There are no radiologic boundaries allowing differentiation between a slight or insignifi cant diaphragmatic elevation and a relevant one. Nevertheless, an extreme elevation is mani-festly pathologic, especially if an abnormal mediastinal shift is present (Fig. 119-1).

The incidence of both eventration and paralysis is diffi cult to estimate. Among newborns, the reported incidence ranges from 1 in 1400 to 1 in 13,000 cases, but elevation of a hemi-diaphragm is 10 times more frequently related to phrenic paralysis than to a true congenital eventration.8,19 In adults, the study of Christensen,20 published in 1959, retrieved 38 cases among 107,778 examined persons. No further study was performed to allow actualization of these data. The male predominance of the condition (60%-80% of cases) and the preferential involvement of the left side are well-established characteristics.17,18

ETIOLOGY

Eventration and Paralysis in ChildrenIn the newborn, diaphragmatic elevation may be related to either a congenital eventration or an acquired diaphragmatic paralysis. The latter is by far the most frequent cause of ele-vation in children. In the series by Huault and coworkers19 dealing with 202 pediatric patients with diaphragmatic eleva-tion, a phrenic paralysis was observed in 190 cases, whereas a true eventration accounted for only 12 cases.

Congenital EventrationThe diaphragm originates from the union of several compo-nents. Its development begins in the 4th week of gestation and fi nishes during the third month. During the 4th week, the septum transversum appears. It is represented by a thick blade of mesoderm that is initially located on the level of occipital and upper cervical somites but is subject to a pro-


Francisco Reyna

Chapter 119 Plication of the Diaphragm 1433

gressive downward migration. During this descent, it is colo-nized by myogenous stem cells migrating from cervical somites. There is progressive fusion of the septum transver-sum with the esophageal dorsal mesentery, which in turn gives rise to the diaphragmatic crura. Communication between the thoracic and the abdominal cavity persists up to the 8th week because of the existence of the posterolaterally located pleuroperitoneal ducts. Closure of this communica-tion is achieved by the pleuroperitoneal membrane, which is initially formed by apposition of the two serosal layers (pleura cranially and peritoneum caudally) but secondarily is subject to colonization from myoblasts originating from a lateral mus-cular burrowing. At the beginning of the 6th week of gesta-tion, the initial structure of the diaphragm reaches the region of thoracic somites, and at the end of the 8th week it can be found at the level of the fi rst lumbar vertebra (L1).

Congenital eventration occurs secondarily to an abnormal myoblastic colonization of the diaphragm during fetal devel-opment. Macroscopically, the diaphragm has the appearance of a translucid membrane. Microscopic examination shows that the diaphragm consists of two serosal layers separated by rare muscular fi bers and fi brotic tissue. Peripheral inser-tions have a normal muscular aspect. Eventration may be total (usually on the left side) or partial (more frequently observed on the right). Partial eventrations are classifi ed into three different types: anterior, posterolateral, medial. Congenital eventration may be associated with other malformations.8,18,21

Diaphragmatic ParalysisIn the newborn, phrenic nerve injury is usually related to a surgical trauma, in most cases occurring during the repair of a congenital cardiovascular anomaly. In the series by Huault and coworkers,19 this cause accounted for 68% of the cases. The incidence of diaphragmatic paralysis after cardiac surgery ranges from 0.3% to 12.8%.22 The second cause of phrenic nerve injury in newborns is obstetric trauma (diffi cult deliv-ery, use of forceps, intrauterine malposition). In these cases, the identifi cation of an associated nerve injury (brachial plexus, recurrent laryngeal, sympathetic) may help in diagnosis.9

Diaphragmatic Elevation in AdultsThe previously described forms may manifest in adulthood and thus require consideration at the time of diagnostic workup. Apart from these cases, the eventrations are classi-cally divided into those with and without phrenic nerve involvement.6,17,18,23

Eventrations With Phrenic Nerve InvolvementThis group of eventrations can be classifi ed according to the level of involvement; such a classifi cation allows a better understanding of laboratory investigations useful in the workup. The levels of involvement are spinal cord (amyo-trophic lateral sclerosis, trauma, poliomyelitis), radicular (vertebral disc diseases, osteophytosis), and nerve. Phrenic nerve involvement at the cervical level is caused by noniatro-genic trauma (e.g., motor vehicle accident), iatrogenic trauma

(obstetric, vertebral or neck surgery, central venous access, locoregional analgesia, collapse therapy), and infection (herpes virus). Nerve involvement at the mediastinal level is caused by metastatic lymph nodes (especially from lung cancers), mediastinal tumors, tuberculous adenitis, noniatro-genic trauma (penetrating and nonpenetrating), and iatro-genic trauma (lung and cardiac surgery).

Eventrations Without Phrenic Nerve InvolvementEventrations without phrenic nerve involvement may result from thoracoabdominal trauma, disease in neighboring tissues (subphrenic abscess, atelectasis, pleural infection), or idio-pathic causes.

Despite an exhaustive workup, the cause of diaphragmatic elevation in adults often remains unexplained. In the study of Pielher and colleagues,24 the cause of eventration could not be found in 142 (57.5%) of 247 patients at initial workup, and only in 6 further cases was the etiology identifi ed during the subsequent follow-up. Neoplasms and cervical or thoracic surgery each accounted for 33% of causes among the 105 patients with a defi ned etiology of diaphragmatic elevation; in the remaining cases, noniatrogenic trauma, infection, or neurologic disease was responsible for the condition. At the time this work was performed, several diagnostic tools were not yet available; nevertheless, in spite of progress in imaging technology, the cause of eventration still sometimes remains uncertain.13

ANATOMICOCLINICAL CONSEQUENCES

ChildhoodElevation of the diaphragm reduces the lung volume. This condition is precariously tolerated because of the weakness of accessory muscles and the excessive mobility of the medi-astinum, which shifts during inspiration, causing contralateral lung compression. This situation is further worsened by the dorsal decubitus position (with diaphragmatic compression by abdominal viscera) and bronchial collapse caused by inherent softness.

Clinical manifestations may appear very precociously: acute respiratory distress, often necessitating a respiratory assistance, is a frequent feature. The need of mechanical ventilation varies between 13% and 72%, according to pub-lished series. It is more frequently necessary in children with phrenic paralysis (40%-72%)13,25-27 than in patients with con-genital eventration (13%-16%).9,25 In some cases, clinical manifestations appear less precociously, and dyspnea, recur-rent bronchitis or pneumonia, vomiting, postprandial suffoca-tion crises, or failure to thrive may constitute the presenting symptoms.

Occasionally, the elevation remains asymptomatic and is discovered only in adulthood.

AdulthoodElevation of the diaphragm is better tolerated in adults and sometimes is discovered only when chest radiography is per-formed for other reasons. Consequences of diaphragmatic elevation may be respiratory, digestive, or cardiac.6,17,18



Respiratory: The elevation of the diaphragm causes a decrease in lung volumes, which is responsible for a restrictive syndrome and a moderate hypoxemia. Clague and Hall28 showed that this syndrome was worsened by placing the patient in the supine position. This was con-

fi rmed in the study by Graham and colleagues.12 Ridyard and Stewart29 showed with ventilation-perfusion scanning that both ventilation and perfusion of the ipsilateral lower lobe are diminished in cases of unilateral diaphragmatic paralysis (Fig. 119-2).

A

B

C

D

Anterior view 3/4 Posterior left

Posterior view 3/4 Anterior right

349K

244K

FIGURE 119-2 Frontal (A) and lateral (B) radiographic views of an adult with elevated hemidiaphragm. C, Computed tomographic scan of the same patient. D, Ventilation-perfusion scan showing the absence of ventilation in the inferior middle right lung.



Digestive: Elevation of the diaphragm is accompanied by ascension of abdominal organs. On the right side, the liver is primarily involved. In partial eventrations, it insinuates itself into the deformation, giving rise to the classic appearance of a bun on chest radiography. Sometimes, liver can be accompanied by colon or gastric antrum. In total eventration, the whole liver is pushed cranially, inducing the ascension of the pylorus with the lower portion of stomach. The right colic fl exure and the median portion of the transverse colon may become interposed between the liver and the diaphragm, resulting in the Chilaiditi syndrome (Fig. 119-3).30 On the left side, the stomach is primarily involved, and it assumes a reversed-U position, with the gastric fundus being pushed posterosu-periorly and the antrum anterosuperiorly. The median portion, which has a horizontal position, is occupied by a new air pocket with the potential for weakening the lower esophageal sphincter, leading to the possibility of a gastroesophageal refl ux. Ascension of colon, spleen, and kidney may be associated.

Cardiac: Cardiac symptoms are more frequent in left-sided eventration. In this case, cardiac shift (dextrocardia) may be responsible for arrhythmias.

As already stated, totally asymptomatic cases are possible. Oligosymptomatic presentations are also possible, including slight dyspnea, sometimes worsened by the supine position; epigastric or upper quadrant pain (spontaneous or induced by the fl exion); regurgitation; and painful eructations.6,7,17,18,23 Exceptional presentations include acute respiratory failure31 and diaphragmatic rupture.32

TECHNIQUES OF DIAPHRAGMATIC PLICATION

PrinciplesPlication of the diaphragm aims to provide a satisfactory tension to an abnormally fl ask-shaped dome, while at the same time lowering it. This procedure has obviously no impact on the mobility of the diaphragm in cases of phrenic paralysis. Nevertheless, in anticipation of restoring of phrenic nerve function, the preservation of distal branches of the nerve allows physiologic movement, in contrast to the exci-sion-suture techniques that do not allow this kind of recovery. Anatomic and histologic observations show that the central portion of the dome is more or less slimmed, with various degrees of atrophy or of rarefaction of the muscular fi bers, whereas the peripheral portion generally maintains a solid texture. Stitches will fi nd better support on this portion of the diaphragm at the time of the plication.6 Lowering of the cupola, while providing a more physiologic tension, allows re-expansion of the adjacent lung, diminution of the adverse effect of abdominal pressure, elimination of paradoxical movements and of mediastinal shift, and improvement of the actions of intercostal and accessory muscles.13

Conventional Procedures (Box 119-1)Plication Through an Open Transthoracic ApproachFlag Plication. This is the reference technique (Fig. 119-4). It is carried out through a more or less large postero-lateral thoracotomy. The location of the thoracotomy incision is variable according to different authors experience (from the 6th to 8th intercostal spaces). The lung and mediastinum

A

B

FIGURE 119-3 A, Chilaiditi syndrome: barium swallow showing interposition of the large bowel between the liver and the diaphragm. B, Computed tomographic scan of the same patient.



Accordion Plication. Other techniques of plication have been described. In particular, the technique by Schwartz and Filler33 is often employed by pediatric surgeons. In this tech-nique (Fig. 119-5), the redundant diaphragm is pulled in a radial direction, and pleats are created by the placement of full-thickness horizontal mattress nonabsorbable sutures in the anteromedial to posteromedial direction while avoiding injury to branches of the phrenic nerve. This type of plication gives the diaphragm an accordion appearance. In this manner, the diaphragm can be plicated with as many rows of sutures as necessary to tighten it. Some authors have also suggested buttressing the fi nal layers of the sutures with polytetrafl uoroethylene pledgets to prevent tearing out of the sutures.12,34

Plication Through an Open Transabdominal ApproachPlication by the abdominal approach (Fig. 119-6) is based on the same principles. It is performed through a median or transverse laparotomy.4,5,17 The diaphragm is grasped with two Babcock forceps, and a large fold is drawn downward. Transfi xing stitches are applied at the base of the fold, which is subsequently folded over and fi xed to the anterior portion of the hemidiaphragm. Inadvertent lung puncture with pneu-mothorax may complicate this technique. Pleuropulmonary adhesions may render the technique unsuitable and even dangerous. The abdominal approach is currently rarely employed. It can be useful in cases of associated abdominal disease or bilateral eventration.

Box 119-1 Surgical TechniquesPlication is carried out by the transthoracic approach in the absence of indication for an abdominal approach (bilateral or associated intra-abdominal disease).

Plication is technically feasible by VATS: the operation is bloodless and rapid, and the desired tension can be applied to the plicated diaphragm.

A BFIGURE 119-4 Flag plication. A, The pleat is created and fi xed with mattress sutures at the base. B, The pleat is folded down and fi xed at its top.

are examined to exclude unsuspected disease. Two Babcock forceps raise the slimmed cupola, creating a fold. The direc-tion of the plication (anteroposterior or transverse) is deter-mined by the grossly apparent axis of the eventration. Generally, plication is performed according to a transverse axis. The fold is fi xed at its base by a series of U-shaped, nonabsorbable stitches. The plicated area is subsequently folded onto the portion of the diaphragm that appears more weak and fi xed close to the costal insertion of the diaphragm by one or several rows of sutures. At the level of the weak-ened portion, the repaired diaphragm has thus a three-layer thickness.

Mechanical stapling of the base of the fold has been pro-posed to replace the U-shaped stitches. Another variant is represented by splitting of the plication after incision of the apex of the fold. Each of the two aspects of the fold is sub-sequently folded onto a side of the axis of the plication. This last technique unquestionably weakens the diaphragmatic dome.



A BFIGURE 119-5 Accordion plication. A, Mattress sutures are placed carefully to avoid major nerve fi bers and entry into the abdominal cavity. B, Completed repair.

FIGURE 119-6 Diaphragm plication by the abdominal approach. The pleat is created with mattress sutures at the base; then it is folded frontally and fi xed at the anterior circumference.

Plication by Minimally Invasive SurgeryPlication by Video-Assisted Thoracic SurgeryThe previously described techniques of plication, and in par-ticular the fl ag plication, require a wide surgical approach to allow creation and handling of the diaphragmatic fold. These methods are not compatible with a video-assisted approach. One of us (JM) developed a technique,14 inspired by the Bisgard operation,3 that has the advantage of being compatible with VATS. The diaphragm is invaginated and then stitched, using two superimposed layers of sutures.Technique. The intervention is performed with the use of general anesthesia and selective tracheobronchial intubation to allow single-lung ventilation. Gastric decompression is achieved by placement of a nasogastric tube. The patient is placed in lateral decubitus position as for standard postero-lateral thoracotomy, with the surgeon standing behind. The operating table is positioned with the head raised to decrease the abdominal pressure on the diaphragm. Two thoracoports (10 mm or 5 mm) are introduced (Fig. 119-7). The fi rst one is placed in the fi fth intercostal space on the posterior axillary line for the introduction of the 0-degree scope (port 1). The second one is inserted in fi fth intercostal space on the ante-rior axillary line (port 2). After exploration of the lung and mediastinum, a 4- to 5-cm thoracotomy is carried out at the level of the eighth or ninth intercostal space on the posterior axillary line. A retractor is not usually necessary. This mini-thoracotomy allows the introduction of conventional instru-ments (needle holder, forceps). An endoscopic Duval forceps, introduced through port 2, is used to grasp and push the apex



FIGURE 119-7 A, Position of the two thoracoscopic ports. A minithoracotomy is made over the ninth intercostal space (ICS) for suturing of the diaphragm. B, With the use of Duval forceps, the apex of the eventration is pushed down toward the abdomen. C, The newly created transverse fold of diaphragm is sutured with nonabsorbable material. D, Completed operation. (FROM MOUROUX J, PADOVANI B, POIRIER NC, ET AL: TECHNIQUE FOR THE REPAIR OF DIAPHRAGMATIC EVENTRATION. ANN THORAC SURG 62:905, 1996.)

of the eventration down into the abdomen. The invagination creates a transverse fold from the minithoracotomy to the cardiophrenic angle behind the phrenic nerve. This fold is closed by a fi rst suture line of nonabsorbable material (Sur-gipro 3.5, Tyco Healthcare, France), beginning at the periph-ery of the diaphragm closest to the minithoracotomy. The fi rst stitch is knotted, with the free end held with a forceps. A superfi cial continuous suture is performed to avoid injury to the subdiaphragmatic organs. Once at the cardiophrenic angle, the sutures are drawn tight while the Duval forceps used to push the diaphragm downward is removed. A row of return stitches is made along the same axis, and the suture is tied with the free end of the fi rst knot. During placement of the return stitches, the suture is followed by the assistant using a gained forceps (to avoid injury to the stitch) intro-duced through port 2. The tension applied in this manner facilitates grasping of the edges of the fold to be sutured. This

fi rst back-and-forth series of continuous suture places the excess diaphragm in the abdomen, and care is taken to avoid applying tension to the fi rst series of sutures.

A second back-and-forth series of continuous suture is carried out similarly, thus burying the fi rst series of suture lines: stitches are inserted through a more peripheral portion of diaphragm to obtain the desired tension of the diaphrag-matic dome.

At the end of the procedure, a chest tube is inserted through port 2 and connected to an underwater suction drainage system. The nasogastric tube is removed the next day. The chest tube is removed 3 to 5 days postoperatively. Breathing exercises are started on the fi rst postoperative day and continued for 1 month.Comments. As for conventional surgery, video-assisted dia-phragmatic plication carries the risk of late recurrence, thus justifying long-term follow-up. In our opinion, two technical



aspects of this technique would prevent recurrence: the fi rst back-and forth running suture allows for maintenance of the excess 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 on the diaphragmatic dome. For this reason, care needs to be taken to avoid applying tension to the fi rst series of sutures.

In our opinion, there are two contraindications to this technique: the existence of extended pleuropulmonary adhe-sions and the need to reinforce the diaphragm with synthetic material. These situation are rarely predictable preopera-tively. The level and extent of the thoracotomy incision and the optimal technical aspects of diaphragmatic repair can be decided only after exploration through port 1 and, if neces-sary, port 2.Variations. Since our initial work, several authors have reported their experience with both adults and children and suggested technical variations.35-42 Hwang and associates35 performed the diaphragmatic plication by a four-port thora-coscopic approach: an 11.5-mm port for a 0-degree 10-mm rigid thoracoscope is placed in the fi fth intercostal space in the midaxillary line; two 5-mm ports are inserted in the eighth and ninth intercostal spaces in the posterior axillary line; and an 11.5-mm port is placed in the sixth intercostal space in the anterior axillary line. On the other hand, Lai and Paterson36 used a 7-cm anterior thoracotomy at the level of the xyphoid cartilage and a posterior thoracoscopic port introduced in the auscultatory triangle. Moon and associates37 used four thoracoports and carry out plication with the use of endoscopic linear staplers.

Since the fi rst successful correction of eventration by video-assisted thoracoscopy in a baby weighing 3 kg,39 the technique has gained popularity among pediatric surgeons. Various authors have described modifi cations to the tech-nique, concerning either the caliber of the thoracoports or the method employed to fi x the plication (Hines, 2003).40-42 These limited surgical series testify to the interest in the technique and the possibility of its implementation in children.

Laparoscopic Diaphragmatic PlicationHttl and associates15 have recently reported a laparoscopic technique of diaphragmatic plication. They treated three patients who had a left-sided diaphragmatic paralysis second-ary to cardiovascular surgery. In their technique, the plication is done with the patient in a 30-degree reversed Trendelen-burg position under general anesthesia after single-lumen endotracheal intubation. The surgeon is positioned between the legs of the patients, and the two assistants are placed one at each side of the patient. Through a paraumbilical incision, a 30- to 45-degree angle laparoscope is introduced via a 10-mm port. Three additional working ports (two of 10 mm and one of 5 mm) are placed in a semicircle in the right or left middle and upper abdomen under visual control. The left hepatic lobe is mobilized along the left triangular ligament. Subsequently, three retention stitches are placed transcuta-neously. By application of extracorporeal traction on these

sutures, the diaphragmatic dome is reduced and an intra-abdominal fold is created. This fold is used for the laparo-scopic diaphragmatic plication with 12 to 15 nonabsorbable U-type sutures that are than tied extracorporally and placed inside the abdomen using the knot pusher. The line of the plication runs from the left dorsal portion of the diaphragm to the ventral medial portion. In the experience of the authors, two minimal splenic injuries occurred, neither requiring splenectomy. No pneumothorax was noted, and satisfactory results were observed on long-term follow-up (at 40, 72, and 84 months).

INDICATIONS FOR PLICATION (Box 119-2)

ChildhoodCongenital EventrationKnowledge about treatment of congenital eventration in chil-dren is mainly derived from case reports and relatively small retrospective series.13,17,25 Patients were usually symptomatic, and in most cases they presented with respiratory distress. There are no data comparing surgical treatment to conserva-tive management, and the timing of operation with respect to the onset of symptoms is usually not stated. In these patients, if there is no evidence of phrenic nerve injury, spon-taneous recovery is unlikely; therefore, surgical indication is probably indicated in every symptomatic patient. These babies are often severely ill because of the frequently associ-ated comorbidities, and diaphragm repositioning may help restore partial function in a hypoplastic lung.13,17,25 Little is known about the management of eventration with few or no symptoms. Although conservative management is probably suffi cient, some authors advocate routine plication to maxi-mize development of the ipsilateral lung.25

Phrenic Nerve InjuryManagement of phrenic nerve injury in children (postpartum or postsurgical) has been much more extensively studied than management of congenital eventration. The condition is usually suspected because of respiratory distress, failure to thrive, or, in operated patients, diffi culty in weaning from mechanical ventilation. If nerve injury is suspected, confi rma-tion is obtained by chest radiography, fl uoroscopy, and/or ultrasonography of the chest.13,22,25,26,41,43-45

Box 119-2 IndicationsChildhoodIndication for plication exists in every symptomatic child with congeni-tal eventration.

In asymptomatic patients with congenital eventration, there are insuffi cient data to provide a level of evidence or grade of recom-mendation as to whether plication is indicated.

AdulthoodPlication is indicated in adults with long-lasting, symptomatic dia-phragmatic elevation.



The policies at various centers differ in terms of the indica-tions for and timing of surgery. In the retrospective surgical series by Tsugawa and associates,25 including 50 patients aged 4 days to 7 years with diaphragmatic elevation of miscella-neous origin (but secondary to phrenic nerve injury in most cases), respiratory distress was the indication for surgery. Ventilatory support was necessary in 10 of their patients for 2 to 6 weeks before plication. The number of cases managed conservatively during the time frame of the study (1971-1996) is not stated. This information can be derived from the retrospective experience (1996-2000) of Joho-Arreola and colleagues,22 who reported on 43 pediatric patients with dia-phragmatic paralysis complicating cardiac surgery. Twenty-nine patients underwent plication because of failure to wean from mechanical ventilation or respiratory distress. Among the 14 patients treated conservatively, the mean assisted ventilation time after cardiac surgery was relatively short (5 days), but some patients were mechanically ventilated for several weeks (up to 49 days). Patients ultimately treated by plication received mechanical ventilation for a longer period (mean, 13 days) before the decision for plication was made. Similarly, in the retrospective series by deVries Reilingh and coworkers,26 18 consecutive patients with obstetric injury to the phrenic nerve were evaluated between 1986 and 1997. All required resuscitation immediately after birth, and 14 of them received intubation and mechanical ventilation. Thir-teen of the 18 patients were ultimately treated by plication (at an average of 100 days postpartum), and in the remaining 5 patients, spontaneous clinical and radiologic recovery was observed within 1 month.

Generally, in the published reports, conservative manage-ment is always attempted before surgery is contemplated in children with phrenic nerve injury. There is general agree-ment that surgery must be performed after stabilization of the clinical condition by gastric decompression, administra-tion of supplemental oxygen, and, if necessary, mechanical ventilation, but the optimal timing of plication is not known. In fact, conservative treatment would permit restoration of diaphragmatic function if the phrenic nerve is not transected, but the time required may be very long (weeks or months), exposing patients to the unacceptable risks associated with prolonged mechanical ventilation. Most authors have pro-posed that observation and ventilation not be prolonged beyond a period of 2 weeks,13,43 so as to allow extubation and improved ventilation, with diaphragm plication being of course indicated only if there is no other primary cause of respiratory distress. If phrenic nerve injury is recognized during the initial cardiac or mediastinal surgery, immediate plication must be performed (Simansky et al, 2002).44

AdulthoodExperience with surgical treatment of eventration in adult-hood is much more limited. Most of the experience is derived from case reports, a small number of retrospective series, and a few prospective studies.11-14,46-49 Controversies exist about indications and the optimal timing of surgery; in this context, consideration of the natural history of diaphragmatic eleva-tion is of paramount importance.

Information about spontaneous evolution of nontraumatic diaphragmatic paralysis can be derived from the large retro-spective study by Pielher and colleagues24 involving 247 patients. The cause of paralysis could be identifi ed at initial evaluation in 105 patients but remained obscure in the remaining 142 subjects, who were followed-up for a mean of 8.7 years with no attempt at surgical repair. The cause of paralysis became evident in only 6 of these patients during the follow-up. In the remaining 136 cases, the leading symptom (exertional dyspnea) improved in only 34%; improvement in the other manifestations, cough and chest wall pain, was observed in 78% and 82% of cases, respec-tively. On chest radiography, the diaphragm returned to a normal position in only 12 of 131 patients who had this examination available.

Efthimiou and colleagues47 studied the evolution of post-surgical diaphragmatic paralysis. In a prospective observa-tional study enrolling 100 consecutive patients over a 6-month period, they reported a 32% incidence of unilateral paralysis among patients receiving ice/slush topical hypothermia during cardiac surgery, compared with 2% among those not receiving topical hypothermia. All of these patients could be treated conservatively, and paralysis regressed within 1, 6, and 12 months in 25%, 56%, and 69% of cases, respectively. At 2-year follow-up, the paralysis had regressed in all but one patient. Electromyography showed the absence of nervous conduction in all patients within 1 week after cardiac surgery but progressive reappearance of conduction in those patients who experienced restoration of diaphragmatic function. Obviously, in these patients, the phrenic nerve had suffered a thermal injury but had not been transected.

In the experience of Deng and associates,48 derived from a retrospective analysis of a prospectively collected database of patients undergoing high free right internal mammary artery harvesting, the incidence of right-sided diaphragmatic paralysis was 4%. In this setting, the phrenic nerve can be either thermally injured (by the proximity of electrocautery dissection) or completely transected. Management included immediate diaphragmatic plication (i.e., during the sternot-omy for cardiac surgery), if phrenic nerve transection was identifi ed intraoperatively, or a middle-term observation for postoperatively evidenced paralysis. Conservative manage-ment was adopted for the fi rst 3 months after cardiac surgery, after which plication was recommended in the absence of spontaneous regression of paralysis (apparently without regard to the presence of symptoms). Among the 26 patients with postoperative diaphragmatic paralysis, spontaneous regression was observed in 14 cases, and the remaining 12 patients were fi nally operated on.

Information about indications for plication can be derived from some retrospective surgical series evaluating the outcome of adult patients with diaphragmatic eventration treated by surgery.11-13,43,46,49 All of these studies included patients with diaphragmatic paralysis secondary to various conditions and idiopathic forms. In almost all instances, the indication for surgery was the presence of respiratory symp-toms (mainly dyspnea or orthopnea, but also cough and chest wall pain) or, less commonly, digestive symptoms (dyspepsia or meteorism) that interfered with patients normal activi-



ties. The mere presence of an elevated diaphragm on chest radiography was not considered an indication for operation (with some exceptions). Because of the retrospective charac-ter of these surgical series, conservative management was not evaluated, and the number (and relative proportion) of patients treated by a nonoperative approach in the same time frames in the various institutions was not stated.

It is generally believed that there is no indication for surgi-cal treatment of diaphragmatic eventration if the condition is secondary to a neoplastic disease or if there are no symp-toms. In our opinion, if a neoplastic origin is excluded on clinical and radiologic grounds, surgery must be considered on the basis of the clinical presentation and the timing of onset of symptoms. If the patient is symptomatic and the diaphragmatic eventration is long-lasting (>2 years), surgery is usually indicated; the operation is planned after clinical conditions have been optimized (e.g., treatment of respira-tory infections, appropriate weight loss). If the patient is symptomatic but the diaphragmatic eventration is recent, a period of observation (18-24 months) before surgery is advo-cated. During this period, physiotherapy is performed and possible issues of excessive weight addressed. In these patients, serial diaphragmatic electromyographies may be suggested to determine possible recovery of phrenic nerve function.50,51 It is noteworthy that spontaneous resolution of a recent eventration is possible.

If the patient has no or few symptoms, strict follow-up is performed, so that surgery may be promptly if even slight deterioration of respiratory function occurs. If signifi cant respiratory impairment is already present, a modest chest trauma or a pulmonary infection could precipitate adverse clinical conditions and necessitate mechanical ventilation.49

Finally, diaphragmatic plication is not contraindicated in patients with ventilatory support. In fact, experience has shown that plication done under these circumstances

can wean the patient from mechanical ventilation if the causes of respiratory distress are identifi ed and treated (Fig. 119-8).31,43,49

Recently, Smyrniotis and colleagues proposed, in order to decrease the risks of respiratory complications, to perform simultaneous prophylactic diaphragmatic plication during a major abdominal operation in patients with phrenic nerve palsy.52

RESULTS (Box 119-3)

ChildhoodPostoperative OutcomeSeveral studies have evaluated the outcome of pediatric patients treated by diaphragmatic plication, usually for phrenic nerve injury. They are summarized in Table 119-1.

A BFIGURE 119-8 A, Radiograph of a 59-year-old woman who experienced right-sided eventration secondary to a blunt chest trauma apparently received 10 years previously. A second blunt trauma (fall down stairs) caused rib fractures and precipitated respiratory failure requiring mechanical ventilation. Tracheostomy was performed to facilitate weaning, but it was complicated by a tracheoesophageal fi stula. Staged diaphragmatic plication to allow weaning and repair of the fi stula was planned. B, Radiograph of same patient 4 years after the plication of the diaphragm.

Box 119-3 ResultsChildhoodDiaphragmatic elevation secondary to phrenic nerve injury in children may be satisfactorily managed by plication; in almost all instances, weaning from respiratory support is possible, often with only a short delay.

Mortality is generally related to the underlying disease and not to the operation itself.

Long-term outcome is determined by the associated comorbidities because the operation allows a permanent improvement in respiratory function.

AdulthoodDiaphragmatic plication in nonventilated adult patients carries a low morbidity and very low, if any, mortality. Functional results are fully satisfactory in almost all cases.



TABLE 119-1 Outcome of Plication in Children

Weaning From Study Operative Follow-up Respiratory Radiologic ClinicalAuthor Period Design N Deaths* (Yr) Support Improvement Improvement

Tonz et al45 1983-1992 Retrospective 11 0 3.2 (mean) 11/11 10/11 9/9

Tzugawa et al25 1971-1996 Retrospective 25 5 1-25 20/20 20/20

deVries Reilingh 1986-1997 Retrospective 14 0 9/9 14/14et al26

deLeeuw et al44 1985-1997 Retrospective 68 4 49/50

Simansky et al43 1988-2000 Retrospective 10 3 7/7

Hines41 Retrospective 5 0 2/2 5/5 5/5

Joho-Arreola 1996-2000 Retrospective 29 8 1 13/21 et al22

*No deaths were related to plication.Not taking into account operative mortality.From Alifano M: Plication for eventration. In Ferguson M (ed): Diffi cult Decisions in Thoracic Surgery. New York, Springer, 2007, pp 356-364.

These studies aimed to evaluate operative mortality, impact of the procedure on weaning the patient from respiratory support, and, in some cases, improvement in clinical and/or radiologic presentation. In the retrospective series by Tsugawa and associates25 dealing with 25 children with phrenic nerve injury treated by thoracotomy plication, weaning from respi-ratory support (mechanical ventilation or supplemental oxygen) was possible with only a short delay (0-6 days) in 15 of 17 patients; the other 2 patients underwent repeat plica-tion, which was successful in one instance. In the same study, 25 additional patients underwent plication for congenital eventration, including 4 who were mechanically ventilated before the operation. Weaning was possible in all 25 cases, 1 to 61 days postoperatively.

Similar results were reported in the retrospective study by Simansky and colleagues.43 Among the 10 children with post-surgical phrenic nerve injury causing respiratory failure who underwent open plication, 7 could be weaned from mechani-cal ventilation (within 8 days in 6 cases), but the remaining 3 died despite a radiologically successful plication, mainly because of intractable underlying cardiac disease. No deaths were reported in the series by Tonz and coworkers,45 who operated on 11 of 25 patients with postsurgical phrenic nerve injury because of failure to wean from mechanical ventilation or respiratory distress after extubation. The remaining patients could be managed conservatively. Weaning was pos-sible in all the cases (within 1 week in all but two cases), and respiratory distress was managed successfully.

A more consistent experience, albeit retrospective, can be drawn from the study by deLeeuw and colleagues,44 also dealing with postsurgical phrenic nerve paralysis. In their experience, 40% of 170 children with this condition under-went open plication. Respiratory insuffi ciency was the indica-tion in almost all cases, with most patients being mechanically ventilated at the time of plication. The median time to fi nal extubation after plication was 4 days, with a range of 1 to 65 days. Multivariate analysis showed that the independent factors associated with a longer time to extubation were

bilateral paralysis and a longer interval from the initial opera-tion to diagnosis. There were four in-hospital deaths, but none of these was considered related to the procedure. As in all of the pediatric series described here, all of the deaths were considered secondary to the underlying disease.

Further evidence that the plication per se is not associated with mortality or major morbidity was provided by the expe-rience of deVries Reillingh and associates,26 who performed the operation by open approach in 13 patients with phrenic nerve injury resulting, in almost all cases, from an obstetric trauma (with no associated cardiac or pulmonary malforma-tions). Respiratory distress necessitating mechanical ventila-tion was present in most cases. Dramatic improvement was observed in all of the patients, with discontinuation of mechanical ventilation within a few days and return of arte-rial gas values to normal in all cases.

A small series of diaphragmatic plication in children by VATS was recently published.41 The authors reported on fi ve children, weighing 3.2 to 13.2 kg, with congenital or postsur-gical diaphragmatic eventration causing respiratory insuffi -ciency or recurrent respiratory infections. Satisfactory clinical and radiologic results were observed in all of the cases. In particular, weaning from mechanical ventilation could be achieved within 3 days in both of the patients who underwent surgery for this indication.

Long-Term OutcomeIn some surgical series of pediatric patients, information about long-term follow-up is available. Tonz and coworkers45 reported no late deaths related to diaphragmatic paralysis and good radiologic results in 10 of 11 patients. No children had respiratory symptoms at late follow-up. Similarly, Tsugawa and colleagues25 observed fully satisfactory clinical and radio-logic results in all of the patients who are available at follow-up after plication for either phrenic nerve injury or congenital eventration. On the other hand, in the study by Joho-Arreola and associates,22 6 of 21 patients had elevated diaphragm 1



year postoperatively; the percentage of patients with respira-tory symptoms in that study is not stated.

Overall, diaphragmatic elevation secondary to phrenic nerve injury in children may be satisfactorily managed by plication; in almost all instances, weaning from respiratory support is possible, often with only a short delay. Mortality is generally related to underlying disease and not to the opera-tion itself. Similarly, long-term outcome is determined by the associated comorbidities because the operation allows a per-manent improvement in respiratory function.

AdulthoodBecause adults with unilateral diaphragmatic elevation usually present with a mild respiratory insuffi ciency, weaning from mechanical ventilation is a rare indication for plication. In the recent prospective study by one of us (JM), plication by VATS was performed for this indication in only two patients, and both were successfully weaned within 1 week.49 On the contrary, only one among the four mechanically ventilated patients in the series by Simanski and colleagues43 involving patients with phrenic nerve injury could be weaned.

When the operation is performed because of less severe respiratory symptoms (or because of digestive problems), satisfactory results are uniformly observed (Table 119-2). In the aforementioned retrospective study by Simansky and colleagues,43 all seven nonventilated patients experienced an improvement in American Thoracic Society (ATS) dyspnea score of 2 or 3 levels at 3-month re-evaluation. At long-term follow-up (11-114 months), all seven were completely asymptomatic from a respiratory point of view.

In the experience of Graham and coworkers12 dealing with 17 patients treated by plication via thoracotomy between 1979 and 1989, an improvement was observed in all patients in both subjective (dyspnea score) and objective measure-ments. In particular, the operation resulted in signifi cant improvement in terms of postoperative forced vital capacity (FVC), total lung capacity (TLC), diffusing capacity for carbon monoxide (DLCO), partial pressure of oxygen (PO2), and partial pressure of carbon dioxide (PCO2). These satisfac-tory results were still present in all of the six patients who could be reassessed at long-term follow-up (>5 years).12 In the retrospective study by Ribet and Linder,13 9 of 11 patients

TABLE 119-2 Outcome of Plication in Adults (Nonventilated Patients)

Study

Operative Follow-up

Improvement

Author Period Design N Deaths (Yr) Clinical Radiologic Functional

Wright et al11 Retrospective 7 0 0.3-4 7/7 7/7 7/7

Graham et al12 1979-1989 Retrospective 17 0 5-7 6/6 6/6 6/6

Ribet and Linder13 1968-1988 Retrospective 11 0 8.5 (mean) 9/11 6/11 5/5

Simansky et al43 1988-2000 Retrospective 7 0 7.3 (mean) 7/7 7/7 7/7

Higgs et al46 1983-1990 Retrospective 19 0 7-14 (n = 15) 14/15 14/15 15/15

Mouroux et al49* 1992-2003 Prospective 10 0 6.3 (mean) 10/10 10/10 10/10

*Operated by video-assisted thoracic surgery.From Alifano M: Plication for eventration. In Ferguson M (ed): Diffi cult Decisions in Thoracic Surgery. New York, Springer, 2007, pp 356-364.

were persistently asymptomatic after the operation (follow-up of 3 months to 18 years), 1 patient was mildly dyspneic, and 1 had persistent digestive symptoms. Of note, chest radiographs showed a persistently elevated (though at a lesser extent) diaphragm in 5 of these cases. In this study, only fi ve patients had both preoperative and postoperative functional assessment, and an improvement in both FVC and forced 1-second expiratory volume (FEV1) was observed in all of those cases.

In the prospective study at Nice University Hospital dealing with 12 adult patients treated by video-assisted plica-tion for diaphragmatic elevation of miscellaneous origin (posttraumatic in most instances),49 all of the patients expe-rienced a complete disappearance of symptoms shortly after the operation, and no radiologic relapse was observed at a follow-up of 64.4 46 months. A signifi cant improvement in both FEV1 and FVC was observed at late spirometry in all of the cases.

SUMMARYPlication of the diaphragm is a simple and feasible technique. It is frequently employed in pediatric surgery. In the adult population, the indications are still debated. The availability of minimally invasive surgery can, in the future, increase interest in the plication technique.

COMMENTS AND CONTROVERSIESRestoration of size and contour of the diaphragm by plication is simplistic in its appeal and performance. However, it is an uncom-mon operation, and reports of its approaches, techniques, indica-tions, and outcomes are few and mostly anecdotal. In the extreme, for the infant with congenital eventration or neonatal phrenic nerve damage, this procedure is well described and very successful in permitting weaning from ventilator support. In the asymptomatic adult with phrenic paralysis or a large eventration, the role of plica-tion has not been defi ned. In any patient, time must be allowed for return of phrenic nerve and diaphragmatic function. All reversible causes of restrictive and obstructive respiratory impairment must be addressed (e.g., smoking, asthma, obesity). Only then plication is considered.

T. W. R.



KEY REFERENCES

Dor J, Richelme H, Aubert J, Boyer R: Lventration diaphragmatique. J Chir 97:399-432, 1969.

This article describes all techniques of plication.

Graham DR, Kaplan D, Evans CC, et al: Diaphragm plication for uni-lateral diaphragmatic paralysis: A 10-year experience. Ann Thorac Surg 49:248-52, 1990.

This report confi rms that diaphragmatic plication is a safe and effective procedure for adult patients with dyspnea resulting from unilateral diaphragmatic paralysis.

Hines MH: Video-assisted diaphragm plication in children. Ann Thorac Surg 76:234-236, 2003.

The fi rst short series (fi ve patients) of plication by VATS in children.

Httl TP, Wichmann MW, Reichart B, et al: Laparoscopic diaphragmatic plication. Surg Endosc 18:547-551, 2004.

Description (with very good photographs) and results of diaphragmatic plication by laparoscopy.

Mouroux J, Padovani B, Poirier NC, et al: Technique for the repair of diaphragmatic eventration. Ann Thorac Surg 62:905-907, 1996.

The fi rst description (with schemas) of the plication by VATS.

Simansky DA, Paley M, Refaely Y, Yellin A: Diaphragm plication fol-lowing phrenic nerve injury: A comparison of paediatric and adult patients. Thorax 57:613-616, 2002.

Presents the results of diaphragmatic plication of ventilated versus nonventilated pediatric and adult patients.


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