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Ann Thorac Surg 2009;88:1112-1117. doi:10.1016/j.athoracsur.2009.05.027
© 2009 The Society of Thoracic Surgeons
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Original Articles: General Thoracic

Long-Term Follow-Up of the Functional and Physiologic Results of Diaphragm Plication in Adults With Unilateral Diaphragm Paralysis

Richard K. Freeman, MD*, Jaclyn Van Woerkom, RN, BSN, Amy Vyverberg, RN, BSN, Anthony J. Ascioti, MD

Department of Thoracic and Cardiovascular Surgery, St Vincent Hospital, Indianapolis, Indiana

Accepted for publication May 8, 2009.

* Address correspondence to Dr Freeman, 8433 Harcourt Rd, Indianapolis, IN 46260 (Email: rfreeman{at}corvascmds.com).

Presented at the Forty-fifth Annual Meeting of The Society of Thoracic Surgeons, San Francisco, CA, Jan 26–28, 2009.


   Abstract
Top
 Abstract
Introduction
 Patients and Methods
 Results
 Comment
 Discussion
 References
 
Background: Patients with lifestyle-limiting dyspnea attributable to unilateral diaphragm paralysis have been shown to experience a decrease in their dyspnea and an improvement in their pulmonary spirometry and functional status with diaphragm plication acutely after surgery. This investigation summarizes these patients' outcomes with long-term follow-up.

Methods: Adult patients undergoing plication of the hemidiaphragm for lifestyle-limiting dyspnea secondary to unilateral diaphragm paralysis were assessed preoperatively, 6 month after surgery and then annually using the Medical Research Council dyspnea score, pulmonary spirometry, activities of daily living questionnaire, and a chest radiograph. Patients with at least 48 months of follow-up were included in this investigation.

Results: Forty-one patients underwent plication of the hemidiaphragm through video-assisted thoracoscopy (30) or thoracotomy (11). Mean follow-up was 57 ± 10 months. Mean forced vital capacity, forced expiratory volume at 1 second, functional residual capacity, and total lung capacity all improved by 19%, 23%, 21%, and 19% (p < 0.005), respectively, when measured 6 months after surgery, as were mean Medical Research Council dyspnea scores (p < 0.0001). These mean values remained constant over the follow-up period. Four patients did not show improvement in their Medical Research Council dyspnea scores nor functional status despite improvements in their pulmonary spirometry values. Two of these patients had a body mass index greater than 35 kg/m2 and 3 had documented unilateral diaphragm paralysis for at least 4 years before plication.

Conclusions: Plication of the hemidiaphragm produces improvement for the vast majority of patients in pulmonary spirometry, dyspnea, and functional status that endures over long-term follow-up. Patients who are morbidly obese or who have longstanding unilateral diaphragm paralysis may not realize the same benefits of plication.


   Introduction
Top
 Abstract
 Introduction
Patients and Methods
 Results
 Comment
 Discussion
 References
 
Adult patients with lifestyle-limiting dyspnea attributable to unilateral diaphragm paralysis have been shown to experience a decrease in their dyspnea and an improvement in their pulmonary spirometry and functional status with diaphragm plication [1]. However, the longevity of these improvements is not well established. This investigation was designed to assess the long-term subjective and objective results of adult patients undergoing plication of the hemidiaphragm for symptomatic unilateral paralysis using serial pulmonary spirometry and dyspnea and daily living activity scales.


   Patients and Methods
Top
 Abstract
 Introduction
 Patients and Methods
Results
 Comment
 Discussion
 References
 
Between 2001 and 2007, patients at least 21 years of age who underwent plication of the hemidiaphragm through thoracotomy or video-assisted thoracoscopy for symptomatic unilateral paralysis were offered participation in this prospective investigation. This study was approved by our Institutional Review Board, with individual consent being waived. All patients received a standardized evaluation before consideration for surgery which included a history and physical examination, a chest radiograph, a fluoroscopic "sniff" test, pulmonary spirometry, administration of the Medical Research Council (MRC) dyspnea score and the Manchester respiratory activities of daily living questionnaire (Table 1 and Appendix) [2, 3]. Patients without a clear etiology for their unilateral diaphragm paralysis also underwent a neurologic consultation and computed tomographic imaging or magnetic residence imaging study of the chest and brain. All patients undergoing diaphragm plication were also required to participate in a pulmonary rehabilitation program under the supervision of a pulmonary physician beginning before surgery. Excluded from participation in this investigation were patients with an upper motor neuron or malignant etiology for their diaphragm paralysis, patients with bilateral diaphragm paralysis, patients with acute respiratory failure/mechanically ventilated patients, and patients with symptomatic diaphragm paralysis not undergoing plication.


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  		Table 1 The Medical Research Council Dyspnea Scale
 
Patients with lifestyle-limiting dyspnea (MRC dyspnea score ≥ 3) from unilateral diaphragm paralysis present for at least 6 months were offered diaphragm plication. When possible, this was performed utilizing video-assisted thoracoscopic techniques, as we have previously described [1]. When using thoracoscopy, three 10-mm ports were utilized in the midclavicular and midaxillary lines of the eighth intercostals space as well as midway between the spine and the medial border of the scapula in the sixth intercostals space. Whether performing thoracoscopy or a thoracotomy, the uncut hemidiaphragm was plicated with a series of six to eight parallel U stitches using contralateral single-lung ventilation. After transecting the inferior pulmonary ligament, sutures were placed beginning medially on the diaphragm progressing laterally until the hemidiaphragm was nearly flat and taut (Fig 1). If performing the procedure thoracoscopically, the Endostitch (Ethicon Endo-Surgery, Cincinnati, OH) was used for intracorporeal suture placement. After surgery, patients' pleural spaces were drained with a chest tube. Patients were discharged home the day after their chest tube was removed when adequate analgesic could be maintained with oral medications and a diet tolerated.


Figure 1
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  		Fig 1. Method of diaphragm plication (video-assisted or thoracotomy). (Reprinted from Freeman et al [1] with permission.)

 
All patients were evaluated 6 months after diaphragm plication and then annually using the MRC dyspnea score, pulmonary spirometry, the activities of daily living questionnaire, a chest radiograph, review of any interim hospitalizations, and assessment of their ability to work. Patients with at least 48 months of follow-up were included in this analysis.

Statistical Analysis
Continuous data are expressed as the mean ± SD of the mean except where otherwise indicated. Differences between categorical variables were evaluated by Fisher's exact test. Differences between continuous variables were measured by the two-tailed Student t test. Statistical significance was accepted as p less than 0.05.


   Results
Top
 Abstract
 Introduction
 Patients and Methods
 Results
Comment
 Discussion
 References
 
During the study period, 52 patients underwent plication of the hemidiaphragm for symptomatic paralysis within our practice. Forty-one of these patients were eligible for inclusion in this investigation. The etiologies of diaphragmatic paralysis as well as patient demographics are listed in Table 2. The most common cause of diaphragmatic paralysis in this series was idiopathic. Patients underwent unilateral diaphragm plication at a mean of 8 ± 6 months (range, 6 to 60) after diagnosis through thoracotomy (11) or video-assisted thoracoscopy (30). Mean length of stay for the entire group was 3 ± 4 days (range, 2 to 10). Morbidities observed included pneumonia (2), atrial fibrillation (2), prolonged ileus (1), and deep venous thrombosis (1). No deaths occurred in this series of patients.


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  		Table 2 Patient Demographics
 
Mean follow-up for patients after diaphragm plication was 57 ± 10 months (range, 49 to 80). As seen in Table 3, mean forced vital capacity, forced expiratory volume at 1 second, functional residual capacity, and total lung capacity measured 6 months after surgery all increased significantly when compared with preoperative mean values. Mean MRC scores and daily living activity scores also showed significant improvement when compared with preoperative mean values. Included within the daily living activity increases was the return to work of 31 (91%) of 34 patients who, before plication, expressed such a desire. No patient in this series experienced a dehiscence of their plication during the follow-up period.


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  		Table 3 Functional Status at Six-Month Follow-Up for Patients Treated With Diaphragm Plication for Unilateral Diaphragm Paralysis
 
The improvements in mean pulmonary spirometry, MRC dyspnea score, and daily living activity scores represent significant improvements in 37 patients (90%). Four patients in this series did not realize a significant subjective improvement in their functional status described by MRC and daily living activities scores despite significant improvements in pulmonary spirometry. Two of these patients had a body mass index greater than 35. Three of these patients also had been known to have a paralyzed hemidiaphragm longer than 4 years before plication.

When mean pulmonary spirometry values, MRC dyspnea score, and daily living activity scores from the initial postoperative assessment at 6 months are compared with the subsequent annual mean values, the improvement described remains constant (Table 3). The stability of the observed pulmonary spirometry improvements are also seen at the individual level in all 41 patients. The 4 patients who did not experience an improvement in their MRC or daily living activity scores also showed stability in these subjective measures over the follow-up period at a significantly lower level than the other 37 patients in this series.


   Comment
Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
Discussion
 References
 
Plication of the hemidiaphragm has become the accepted treatment for pediatric patients with significant respiratory impairment because of unilateral diaphragmatic paralysis [4, 5]. Modern series have found significant improvement in patients' respiratory status after diaphragmatic plication with little associated morbidity. Some have recommended and subsequently shown the benefit of including plication even earlier in the treatment algorithm of such patients [6].

The use of diaphragm plication in the treatment of adult patients with unilateral diaphragm paralysis, however, has remained uncommon. Although not all adult patients with unilateral diaphragm paralysis are symptomatic, many are left with lifestyle-limiting dyspnea despite medical therapy and pulmonary rehabilitation. Such dyspnea is multfactorial in origin and results from significant alterations in respiratory physiology including atalectasis with ventilation/perfusion mismatch, shift of the mobile mediastinum to the contralateral side, worsening of pulmonary spirometry, paradoxical movement of the affected hemidiaphragm, and the preferential redistribution of pulmonary blood flow to the contralateral lungs [7, 8].

We have previously reported our series of unilateral diaphragm plications in symptomatic adult patients. We found these patients received significant benefits from diaphragm plication including decreased dyspnea, improved pulmonary spirometry, a trend toward less frequent respiratory-associated hospitalizations, and frequent return to work. While the results of such an investigation encourage physicians involved in these patients' care to consider plication surgery, this series remains small in absolute number and provided relatively brief follow-up. The purpose of this investigation was to assess the long-term subjective and objective outcomes of what is the largest series of adult patients undergoing diaphragm plication for chronic dyspnea attributable to unilateral diaphragm paralysis.

In this report, 41 patients underwent unilateral diaphragm plication, the majority by video-assisted thoracoscopy. We achieved a minimum follow-up period of 4 years in all patients, with a mean follow-up period of 57 months. All 41 patients experienced significant objective improvement in their pulmonary spirometry after diaphragm plication. Thirty-seven patients (90%) also recorded significant improvements in their dyspnea after plication. Both of these effects appeared to be sustained over the entire follow-up period. A return to work rate of 91% was also observed after plication.

Of the 4 patients who did not report significant improvements in their dyspnea score, 2 were, by definition, morbidly obese, having a body mass index of greater than 35. Three of these same patients also had a delay of at least 4 years between the diagnosis of unilateral diaphragm paralysis and plication. Although it seems intuitive that these two factors may influence the outcome of patients undergoing diaphragm plication, the power of this investigation is not sufficient to allow such conclusions.

Three previous reports of unilateral diaphragm plication in adult patients exist in which extended follow-up is also available. The first, by Higgs and coworkers [9], followed 19 patients for a mean of 10 years with annual pulmonary spiromtery and found that initial improvements persisted. No attempt at quantifying dyspnea was made. Mouroux and colleagues [10] similarly followed 12 adult patients to a mean of 64 months and arrived at a similar conclusion. Again, no measurement of subjective dyspnea was made. In 2007, Versteegh and colleagues [11] reported their series of 22 patients undergoing plication. These patients were followed to a mean of 49 months with annual pulmonary spirometry and the American Thoracic Society Dyspnea Scale, again with no degradation of these measures found over time. The actual follow-up period varied, however, and could be as little as 14 months for inclusion in the investigators' report. Some patients who had undergone bilateral diaphragm plication were also included, making the results more difficult to interpret.

In contrast to the above reports, this investigation combined a minimum follow-up period of 48 months for a patient to be included, achieving a mean of 57 months. Unlike the reports by Higgs and coworkers [9] and Mouroux and associates [10], a dyspnea scale was used in all of our patients in an attempt to quantify any change related to diaphragm plication. The number of patients included in this report is also more than double the number of patients undergoing unilateral diaphragm plication in any of these other reviews. Lastly, the majority of patients in this series underwent plication using video-assisted thoracoscopy.

In conclusion, this investigation demonstrates that adult patients with chronic dyspnea attributable to unilateral diaphragm paralysis received significant benefits from diaphragm plication. Such benefits include decreased dyspnea as measured by the MRC dyspnea score, improved pulmonary spirometry, and frequent return to work. These effects were independent of whether the plication was performed through thoracotomy or thoracoscopy. Furthermore, the improvements seen in pulmonary spirometry and dyspnea persisted over the entire follow-up period, which lasted as long as 6 and a half years in some patients. Morbid obesity and an excessive interval of time between diagnosis and plication may reduce the benefits of such treatment. Based on the results of this investigation, it is recommended that adult patients with unilateral diaphragm paralysis who have lifestyle-limiting symptoms of chronic dyspnea be considered for diaphragm plication.


   Appendix
 

Table

Reprinted from Yohannes AW, Greenwood YA, Connolly MNJ. Reliability of the Manchester respiratory activities of daily living questionnaire as a postal questionnaire. Age Ageing 2002;31:355–8, by permission of British Geriatrics Society.


   Discussion
Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
 Discussion
References
 
DR RAFAEL S. ANDRADE (Minneapolis, MN): Doctor Freeman, I commend you, this is a very nicely done paper; as you know, very little has been published that is of value or well done like this. I just have a couple of questions.

One, how did you define paralysis, or are you also doing patients with eventration? In our experience, as long as the diaphragm is elevated and the patient has dyspnea, it doesn't make a difference. And the second question, how many of these patients did you start with VATS and eventually convert to thoracotomy?

DR FREEMAN: Thank you, Dr Andrade, for your comments and also for your group's contribution to this area. As far as the second question, we converted in 3 obese patients.

We do our best, especially in this study, to try and separate eventration from paralysis and probably failed a couple of times. But we do that with history with a CT scan, which everyone gets, and with a sniff test.

DR ANDRADE: So what if you see a patient in clinic, and it may not be in yours, but who has bad dyspnea, is lifestyle-limiting, and on your fluoroscopy you can't confirm that this person has paralysis, would you reject that patient? I think the message should be that that patient should have surgery regardless of the fluoroscopy.

DR FREEMAN: You're absolutely right. We just did not include those people in this study. But you're absolutely right. We would offer that patient surgery.

DR ANDRADE: Thank you.

DR DANIEL L. MILLER (Atlanta, GA): I disagree with that because when you evaluate them with the sniff test and they have paradoxical motion and they're greater than 6 months from whatever event that caused there injury, then I would go ahead and do the plication.

But if there is paresis, there is no paradoxical movement of the diaphragm, in the majority of the time putting them through a rehab program with diaphragmatic breathing exercises they will get better and have full recovery of the diaphragm. However, if they're on CPAP or if they can't lay down flat, then they should plicated.

We see fewer injuries because of the off-pump cases, but we're seeing an increase after minimally invasive mitral valve surgery. So when patients come to you, do you send them through any type of diaphragmatic breathing exercises or other rehabilitation?

DR FREEMAN: Well, in deference to some of my cardiac partners that are here, I'll skip over the last part. I didn't go into it for the sake of time but all these patients have pulmonary rehabilitation before we ever considered them for surgery.

Your point is well taken about paradoxical motion, but I think when we look at these patients, that's only part of the pathophysiology. The other part is the chronic atelectasis and a very small hemithorax compared with what they started with. That being said, if they're in the office and they don't have significant symptoms, we're not going to offer them anything. They have to be symptomatic, and they have to have the findings.

DR MILLER: Thank you.

DR SETH D. FORCE (Atlanta, GA): The last patients who you just mentioned are the patients I want to talk about because the patients who I'm seeing in my office are the patients who have mild to moderate symptoms. They're not severely limited, but they do have shortness of breath. This issue is always are they feeling a difference from the paralyzed diaphragm or from atelectasis on that side?

What I usually do is get a CT scan and then a quantitative ventilation perfusion scan, and if I see either a matched defect or a mismatch at that level, I'll offer them surgery. But if I don't, then I won't, and I assume it's just from having a nonfunctioning diaphragm. Does that sound reasonable? Do you do anything like that with those types of patients, or how do you evaluate those patients for surgery?

DR FREEMAN: I think that's one of the benefits of an objective measure of their dyspnea, because if they don't have that much significant dyspnea, if it's not really lifestyle limiting, I probably wouldn't offer him surgery. I would offer that person pulmonary rehabilitation and see where we end up.

DR MICHAEL S. MULLIGAN (Seattle, WA): Quick question. This is a practical one. Usually these folks come in and they say I can't exercise; that's why I'm obese. And you reply, you're obese and the upward pressure on your diaphragm repair is going to cause it to fail. You hinted at the fact that you got into a little more trouble when the body mass index (BMI) really started to climb. Do you have a threshold BMI whereby the results are prohibitively threatened?

DR FREEMAN: What we found in our previous work was that those patients are not very amenable to thoracoscopic repair. You are right, it's a chicken and an egg problem. And we will usually offer them plication anyway, and the majority of them do get better.

DR JOSHUA R. SONETT (New York, NY): Did you exclude ventilated patients just from the study, or would you still plicate ventilated patients in order to try to palliate them, get them off the vent?

DR FREEMAN: We excluded them from the study. They're a very small number. And we have done it a couple of times, but it's a very unusual event, so I can't really give you a good answer.


   References
Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
 Discussion
 References
 

  1. Freeman RK, Wozniak TC, Fitzgerald EB. Functional and physiologic results of video-assisted thoracoscopic diaphragm plication in adult patients with unilateral diaphragm paralysis Ann Thorac Surg 2006;81:1853-1857.[Abstract/Free Full Text]
  2. Bestal JC, Paul EA, Garrod R, Jones PW, Wedzicha JA. Usefulness of the Medical Research Council dyspnea scale as a measure of disability in patients with chronic obstructive pulmonary disease Thorax 1999;54:581-586.[Abstract/Free Full Text]
  3. Yohannes AW, Greenwood YA, Connolly MNJ. Reliability of the Manchester respiratory activities of daily living questionnaire as a postal questionnaire Age Ageing 2002;31:355-358.[Abstract/Free Full Text]
  4. Langer JC, Filler RM, Coles J, Edmonds JF. Plication of the diaphragm for infants and young children with chronic nerve palsy J Pediatr Surg 1988;23:749-751.[Medline]
  5. Kizilcan F, Tanyel FC, Hicsonmez A, Buyukpamukcu N. The long term results of diaphragmatic plication J Pediatr Surg 1993;28:42-44.[Medline]
  6. DeVries TS, Koens BL, Vos A. Surgical eventration caused by phrenic nerve injury in the newborn J Pediatr Surg 1998;33:602-605.[Medline]
  7. Stevenson JG. Effects of unilateral diaphragm paralysis on branch pulmonary artery flow J Am Soc Echocardiogr 2002;15:1132-1139.[Medline]
  8. Schwartz MZ, Filler RM. Plication of the diaphragm for symptomatic phrenic nerve paralysis J Pediatr Surg 1978;13:259-263.[Medline]
  9. Higgs SM, Hussain A, Jackson M, Donnelly RJ, Berrisford RG. Long term results of plication for unilateral diaphragm paralysis Eur J Cardiothorac Surg 2002;21:294-297.[Abstract/Free Full Text]
  10. Mouroux J, Venissac N, Leo F, Alifano M, Guillot F. Surgical treatment of diaphragmatic eventration using video-assisted thoracic surgery: a prospective study Ann Thorac Surg 2005;79:308-312.[Abstract/Free Full Text]
  11. Versteegh MI, Braun J, Voigt PG, et al. Diaphragm plication in adult patients with diaphragm paralysis leads to long-term improvement of pulmonary function and level of dyspnea Eur J Cardiothorac Surg 2007;32:449-456.[Abstract/Free Full Text]



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 Ann. Thorac. Surg.Home page
S. S. Groth and R. S. Andrade
Diaphragm Plication for Eventration or Paralysis: A Review of the Literature
Ann. Thorac. Surg., June 1, 2010; 89(6): S2146 - S2150.
[Abstract] [Full Text] [PDF]

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