Asian Cardiovasc Thorac Ann 2006;14:e12-e13
© 2006 Asia Publishing EXchange Ltd
Post-Pneumonectomy Syndrome in an Adult Presenting With Positional Syncope
Cengiz Gebitekin, MD,
Ahmet S Bayram, MD
Thoracic Surgery Department, Uludag University Medical Faculty, Bursa, Turkey
For reprint information contact: Ahmet S Bayram, MD Tel: 90 224 442 9166 Fax: 90 224 442 8698 Email: asbayram2{at}yahoo.com, Uludag University, School of Medicine, GKDC-ABD Bursa 16059, Turkey.
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ABSTRACT
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Post-pneumonectomy syndrome is a rare late complication of pneumonectomy, which is due to extreme mediastinal shift and rotation into the pneumonectomy space. Recurrent syncope following a right pneumonectomy in a 21-year-old woman was treated by insertion of an expandable silicone prosthesis into the pneumonectomy space.
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INTRODUCTION
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Post-pneumonectomy syndrome is a rare late complication of pneumonectomy due to extreme mediastinal shift and rotation into the pneumonectomy space. The patient usually presents with progressive exertional dyspnea, stridor, or recurrent infections, due to compression of the main bronchus. We report an unusual presentation of gross mediastinal displacement following a right pneumonectomy.
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CASE REPORT
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A 21-year-old woman underwent a right pneumonectomy for bronchiectasis. Two years later, she presented with recurrent syncopal attacks. These were positional. As a result of gross mediastinal displacement to the pneumonectomy side, the venae cavae and right heart were compressed when the patient lay on her right side, and backfl ow to and outfl ow from the heart were decreased. Echocardiography and computed tomography showed severe mediastinal displacement into the right hemithorax and compression of the right side of the heart (Figure 1a and 1b
). Electrocardiographic monitoring demonstrated sinus rhythm with no change of rhythm during the syncopal attacks. The patient did not complain of any respiratory symptoms and bronchoscopy demonstrated only minimal posterior compression at the level of the carina. She was managed conservatively for a year, but underwent surgery after her symptoms deteriorated.
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Figure 1. Computed tomography scans 2 years after a right pneumonectomy. (a) There is mediastinal shift and rotation into the pneumonectomy space, compressing the right side of the heart, but (b) only minimal narrowing of the left main bronchus.
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The previous thoracotomy scar was re-opened and adhesions between the mediastinum and pleural space divided. An expandable prosthesis (Eurosilicone, La Peyrolière, France) fi lled with 200 mL of saline was inserted into the pneumonectomy space; a port to allow alteration of the volume was placed subcutaneously over the anterior third of the fi fth rib. Each week, 50 mL of saline was added via the subcutaneous injection port until a total volume of 600 mL was reached (Figure 2a). Overfi lling of the prosthesis and compression of the right heart led to recurrence of symptoms 3 months later, necessitating removal of 100 mL of saline. An additional 100 mL of saline was aspirated two months later due to further mild symptoms (Figure 2b). The patient is currently symptom-free 5 years after the procedure.
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Figure 2. Compression of the right side of the heart by the prosthesis (a) is relieved after withdrawing 200 mL saline (b).
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DISCUSSION
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Post-pneumonectomy syndrome is most often reported in children, but seen after approximately 1 in 640 pneumonectomies in adults.1 The two largest series in adults show that patients commonly present with progressive exertional dyspnea, but stridor and recurrent infections have also been noted.2–4 Various periods between pneumonectomy and the onset of symptoms of 4 months to 17 years have been reported.2,3 This condition and its treatment can be complicated by secondary malacia of the airway cartilage.2 Positional syncope is an unusual presentation of this rare complication of pneumonectomy.
The accepted operative treatment of post-pneumonectomy syndrome is to relocate the mediastinum to its normal position and then to hold it in place with a prosthetic implant placed in the pneumonectomy space.2,4 In those not suitable for operation or with airway malacia, an endobronchial stent may be employed to maintain patency of the main bronchus. Unfortunately, saline implants can be displaced or leak, requiring further intervention, and there is always a risk of infection when using any foreign material. Expandable implants, fi rst used in plastic surgery, have the advantage that their volume can be altered via a subcutaneous injection port to obtain slow and controlled displacement of the mediastinum and optimal relief of symptoms.
In our case, inserting the implant worked well to hold the mediastinum in its new position. It is quite diffi cult to estimate the total amount of saline necessary to relieve the patient’s symptoms; 400 mL was required in this case. Repeated injections and aspirations were necessary before the symptoms were completely ameliorated. This case demonstrates the long-term effi cacy of insertion of a prosthetic implant in which the volume may be varied via a subcutaneous injection port.
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REFERENCES
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- Jansen JP, Brutel de la Riviere A, Alting MP, Westermann CJ, Bergstein PG, Duurkens VA. Post-pneumonectomy syndrome in adulthood. Surgical correction using an expandable prosthesis. Chest 1992;101:1167–70.[Abstract/Free Full Text]
- Grillo HC, Shepard JA, Mathisen DJ, Kanarek DJ. Postpneumonectomy syndrome: diagnosis, management, and results. Ann Thorac Surg 1992;54:638–51.[Abstract]
- Valji AM, Maziak DE, Shamji FM, Matzinger FR. Postpneumonectomy syndrome: recognition and management. Chest 1998;114:1766–9.[Abstract/Free Full Text]
- Birdi I, Bughai M, Wells FC. Surgical correction of postpneumonectomy stridor by saline breast implantation. Ann Thorac Surg 2001;71:1704–6.[Abstract/Free Full Text]
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C. Soll, D. Hahnloser, T. Frauenfelder, E. W. Russi, W. Weder, and P. B. Kestenholz
The postpneumonectomy syndrome: clinical presentation and treatment
Eur. J. Cardiothorac. Surg.,
February 1, 2009;
35(2):
319 - 324.
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