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chapter
119
Jrme Mouroux
Nicolas Venissac
Francesco Leo
Daniel Pop
Marco Alifano
Key Points
Eventration and permanent phrenic nerve injury are indications for
injury.
Unfortunately, outcomes and related measures can be only anec-
dotally described.
HISTORICAL NOTE
Wood1 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
first 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
symptoms. In 1947, Bisgard3 described precisely the technique of plication employed to treat a 6-week-old baby. Since
these first 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 (plication 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 DonzeauGouche and colleagues,10 Wright and colleagues,11 Graham
and colleagues,12 and Ribet and Linder13 confirmed the
improvement of respiratory functions with surgical treatment. 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 plication 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 diaphragmatiques de ladultes: A propos de vingt cas. Ann Chir Thor Cardiovasc 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 diaphragmatique. J Chir 69:101-121, 1953.
Revillon Y, Fekete CN: Eventration diaphragmatique chez lenfant. Ann
Chir 36:71-74, 1982.
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Section 6 Diaphragm
ETIOLOGY
Eventration and Paralysis in Children
In 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 elevation in children. In the series by Huault and coworkers19
dealing with 202 pediatric patients with diaphragmatic elevation, a phrenic paralysis was observed in 190 cases, whereas
a true eventration accounted for only 12 cases.
Congenital Eventration
The diaphragm originates from the union of several components. Its development begins in the 4th week of gestation
and finishes 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-
FIGURE 119-1 A, Left diaphragmatic eventration with mediastinal contralateral shift. B, Same patient, 2 years after plication of the diaphragm.
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gressive downward migration. During this descent, it is colonized by myogenous stem cells migrating from cervical
somites. There is progressive fusion of the septum transversum 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 communication 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 muscular burrowing. At the beginning of the 6th week of gestation, 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 first lumbar vertebra (L1).
Congenital eventration occurs secondarily to an abnormal
myoblastic colonization of the diaphragm during fetal development. 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 fibers and fibrotic tissue. Peripheral insertions 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 classified
into three different types: anterior, posterolateral, medial.
Congenital eventration may be associated with other
malformations.8,18,21
Diaphragmatic Paralysis
ANATOMICOCLINICAL CONSEQUENCES
Childhood
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Adulthood
Elevation of the diaphragm is better tolerated in adults and
sometimes is discovered only when chest radiography is performed for other reasons. Consequences of diaphragmatic
elevation may be respiratory, digestive, or cardiac.6,17,18
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1434
Section 6 Diaphragm
B
244K
C
Anterior view
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.
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Posterior view
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1435
Principles
Plication of the diaphragm aims to provide a satisfactory
tension to an abnormally flask-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 excision-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 fibers,
whereas the peripheral portion generally maintains a solid
texture. Stitches will find 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
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1436
Section 6 Diaphragm
FIGURE 119-4 Flag plication. A, The pleat is created and fixed with mattress sutures at the base. B, The pleat is folded down and fixed at its
top.
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FIGURE 119-5 Accordion plication. A, Mattress sutures are placed carefully to avoid major nerve fibers and entry into the abdominal cavity.
B, Completed repair.
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The previously described techniques of plication, and in particular the flag 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 posterolateral 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 first one
is placed in the fifth intercostal space on the posterior axillary
line for the introduction of the 0-degree scope (port 1). The
second one is inserted in fifth intercostal space on the anterior 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 minithoracotomy allows the introduction of conventional instruments (needle holder, forceps). An endoscopic Duval forceps,
introduced through port 2, is used to grasp and push the apex
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1438
Section 6 Diaphragm
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.)
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sutures, the diaphragmatic dome is reduced and an intraabdominal fold is created. This fold is used for the laparoscopic 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).
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1440
Section 6 Diaphragm
The policies at various centers differ in terms of the indications 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 miscellaneous 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 (19711996) 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 diaphragmatic paralysis complicating cardiac surgery. Twentynine 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. Thirteen 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 management is always attempted before surgery is contemplated in
children with phrenic nerve injury. There is general agreement that surgery must be performed after stabilization of
the clinical condition by gastric decompression, administration 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 proposed 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
Adulthood
Experience with surgical treatment of eventration in adulthood 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 elevation is of paramount importance.
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FIGURE 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 fistula. Staged diaphragmatic
plication to allow weaning and repair of the fistula was planned. B, Radiograph of same patient 4 years after the plication of the diaphragm.
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Section 6 Diaphragm
Author
Tonz et al45
Operative
Deaths*
Follow-up
(Yr)
Weaning From
Respiratory
Support
Radiologic
Improvement
Clinical
Improvement
Retrospective
11
3.2 (mean)
11/11
10/11
9/9
Study
Period
Design
1983-1992
25
Tzugawa et al
1971-1996
Retrospective
25
1-25
20/20
20/20
deVries Reilingh
et al26
1986-1997
Retrospective
14
9/9
14/14
deLeeuw et al44
1985-1997
Retrospective
68
49/50
Simansky et al43
1988-2000
Retrospective
10
7/7
Retrospective
2/2
5/5
5/5
1996-2000
Retrospective
29
13/21
Hines
41
Joho-Arreola
et al22
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bilateral paralysis and a longer interval from the initial operation 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 experience 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 malformations). Respiratory distress necessitating mechanical ventilation 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 arterial 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 five
children, weighing 3.2 to 13.2 kg, with congenital or postsurgical diaphragmatic eventration causing respiratory insufficiency 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 Outcome
In 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 radiologic results in all of the patients who are available at followup 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
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Study
Period
Design
Retrospective
N
7
Operative
Deaths
Follow-up
(Yr)
Clinical
Radiologic
Functional
0.3-4
7/7
7/7
7/7
Graham et al
1979-1989
Retrospective
17
5-7
6/6
6/6
6/6
1968-1988
Retrospective
11
8.5 (mean)
9/11
6/11
5/5
1988-2000
Retrospective
7.3 (mean)
7/7
7/7
7/7
Higgs et al
1983-1990
Retrospective
19
7-14 (n = 15)
14/15
14/15
15/15
Mouroux et al49*
1992-2003
Prospective
10
6.3 (mean)
10/10
10/10
10/10
43
Simansky et al
46
year postoperatively; the percentage of patients with respiratory 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 operation itself. Similarly, long-term outcome is determined by the
associated comorbidities because the operation allows a permanent improvement in respiratory function.
Adulthood
Because adults with unilateral diaphragmatic elevation usually
present with a mild respiratory insufficiency, 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 measurements. In particular, the operation resulted in significant
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 satisfactory 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
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were persistently asymptomatic after the operation (followup 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 five
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 plication for diaphragmatic elevation of miscellaneous origin
(posttraumatic in most instances),49 all of the patients experienced 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 significant improvement in
both FEV1 and FVC was observed at late spirometry in all of
the cases.
SUMMARY
Plication 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.
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Section 6 Diaphragm
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 unilateral diaphragmatic paralysis: A 10-year experience. Ann Thorac
Surg 49:248-52, 1990.
This report confirms 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 first short series (five patients) of plication by VATS in children.
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