Ann Thorac Surg 2009;88:e20-e21. doi:10.1016/j.athoracsur.2009.06.093
© 2009 The Society of Thoracic Surgeons
Case Reports
Pleuropulmonary Complications of Rheumatoid Arthritis: A Thoracic Surgeon's Challenge
Natasha Rueth, MD,
Rafael Andrade, MD,
Shawn Groth, MD,
Jonathan D'Cunha, MD, PhD,
Michael Maddaus, MD*
Department of Surgery, Division of Thoracic and Foregut Surgery, University of Minnesota, Minneapolis, Minnesota
Accepted for publication June 23, 2009.
* Address correspondence to Dr Maddaus, MMC195, 420 Delaware St SE, Minneapolis, MN 55455 (Email: madda001{at}umn.edu).
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Abstract
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Approximately 1% of patients with rheumatoid arthritis have chronic lung disease develop, which can lead to complications, including pneumothorax and bronchopleural fistula. Given the inflammatory changes found, along with the immunosuppressant regimen used in management, these complications are often recalcitrant to initial surgical maneuvers. Our goal in reviewing these patients is to demonstrate the escalation of therapeutic interventions that may be needed to ensure successful resolution of this challenging disease process.
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Introduction
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Rheumatoid arthritis (RA) is a systemic illness with potential for pleuropulmonary complications in patients who may have diffuse pulmonary nodules develop. These nodules have a penchant to become necrotic and rupture, leading to a pneumothorax developing, or more severely leading to a bronchopleural fistula (BPF) (Fig 1) [1]. In addition, the lung is often inflamed, with a thick rind and noncompliant parenchyma, hindering diagnostic attempts and rendering the thoracic cavity resistant to standard therapeutic maneuvers.
The current literature describes simple management of an RA-induced pathology using standard techniques (ie, video-assisted thoracoscopic surgery [VATS] and pleurodesis) [1, 2]. However, the pleuropulmonary complications of rheumatoid arthritis often recur and frequently require repeated interventions. When this happens, their hospital course and subsequent management is anything but simple, and a paucity of literature guides thoracic surgeons in managing recalcitrant cases. To illustrate the decision-making paradigm used when complications persist, we present a series of 3 patients with recurrent rheumatoid arthritis disease, along with our approach to long-term care.
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Case Reports
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Patient 1
Our first patient is a 58-year-old man who underwent standard talc pleurodesis for rheumatoid effusions 4 years prior. He subsequently presented to our institution with an empyema. We performed VATS exploration and noted diffuse rheumatoid nodular disease. After exploration, his pneumothorax resolved and he was discharged home.
Weeks later he returned again with empyema, and consequently he underwent a thoracotomy and decortication of the inflammatory rind. Intraoperatively we noted a small BPF associated with his rheumatoid nodular disease; because of this we developed an Eloesser flap for drainage and dressing changes (Fig 2).
After 7 weeks of outpatient wound care, we closed the skin edges, but his empyema recurred again within weeks. Surgically we again evacuated his empyema, while noting a persistent BPF. We placed a chest tube, and he was discharged with the tube for drainage purposes, allowing the BPF to spontaneously heal with time. The tube was subsequently removed as an outpatient (2 to 3 cm of retraction weekly) allowing the tract to close.
At 2 years of follow-up, his BPF had resolved, his wound has fully healed, and he has maintained excellent pulmonary functional status.
Patient 2
We performed a standard VATS pleurodesis on a 45-year-old man with a previously drained rheumatoid effusion but with persistent pneumothorax. Despite an initial resolution, the patient returned to the clinic with shortness of breath and imaging consistent with recurrence of his pneumothorax (Fig 3).
We operatively drained his chest and again performed talc pleurodesis. Having learned from patient 1 as previously described, we attempted to treat this patient similarly, with tube thoracostomy for chest drainage, appropriate manipulation of immunosuppressive therapy, and time. However, this treatment repeatedly failed for this patient; attempts at chest tube removal resulted in recurrent pneumothoraxes.
When repeat talc pleurodesis and outpatient tube thoracostomy drainage proved unsuccessful, we turned to primary closure of the air leak, application of a synthetic sealing agent, and parietal pleurectomy. Initially successful, the patient returned after 3 weeks with a symptomatic pneumothorax again. Currently, he has been managed as an outpatient with a chest tube, having a persistent leak.
Patient 3
Finally, we managed a 37-year-old woman with recurrent pleural space infections, cavitated rheumatoid nodules, and nonhealing BPFs. Similarly with our first 2 patients, this patient had a protracted course requiring multiple debridements, explorations, attempts at pleurodesis, and chest drainage procedures. Her disease is bilateral, and her course consists of repeated failure, despite multiple treatment attempts; we have resorted to bilateral Eloesser flaps for chronic chest drainage with no attempts to close her cutaneous marsupialization.
These patients represent an escalation of resistance to the standard management of thoracic rheumatoid arthritis complications, and demonstrates the progression of surgical techniques used in their care.
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Comment
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Extra-articular pathology is common in patients with RA; approximately 1% of patients have pleuropulmonary disease [1], which is usually asymptomatic. However, if nodular cavitation or necrosis occurs, then complications can develop that may be resistant to standard care.
Review of the current literature demonstrates the significance of our series. The reported success rate for VATS with pleurodesis as management of spontaneous pneumothorax is 97% in the absence of rheumatoid disease; long-term recurrence rates range from 5% to 9% [3]. Clearly, these results can not be expected when the underlying lung is diseased, necrotic, and fibrotic (as with RA), thus exemplifying the need for a more persistent treatment algorithm.
Regarding management of rheumatoid nodule pulmonary disease, Saravena [2] describes treatment of a pneumothorax with associated BPF. With VATS drainage and pleurodesis [2], the author attained an uncomplicated resolution. Other authors have described fatalities associated with the pulmonary complications of RA, despite intervention [4, 5]. Herein, we demonstrate the progressive depth of involvement that may be needed to minimize the pulmonary impact of this disease process.
In summary, unlike simple BPFs in the setting of apical blebs, BPFs associated with rheumatoid nodules may be dramatically resistant to VATS pleurodesis. Perhaps the standard approach to spontaneous pneumothorax (VATS pleurodesis) should be reconsidered in this setting. Rather than alter a virgin pleural space with potentially inadequate treatments, it may be more appropriate to consider an early aggressive approach (such as thoracotomy with direct closure or resection of the fistula) when the pleural space is still unaltered and the visceral pleural surface is without loculated adhesions. If these fail, then escalated therapy may be indicated, including surgical pleurectomy or Eloesser flaps. This way, one may have the highest chance of success dealing with this complex patient population.
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References
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- Adelman HM, Dupont EL, Flannery MT, Wallach PM. Case report: recurrent pneumothorax in a patient with rheumatoid arthritis Am J Med Sci 1994;308:171-172.[Medline]
- Saravana S, Gillott T, Abourawi F, Peters M, Campbell A, Griffith S. Spontaneous pneumothorax: an unusual presentation of rheumatoid arthritis Rheumatology (Oxford) 2003;42:1415-1416.[Medline]
- Doddoli C, Barlési F, Fraticelli A, et al. Video-assisted thoracoscopic management of recurrent primary spontaneous pneumothorax after prior talc pleurodesis: a feasible, safe and efficient treatment option Eur J Cardiothorac Surg 2004;26:889-892.[Abstract/Free Full Text]
- Kitamura A, Matsuno T, Narita M, Shimokata K, Yamashita Y, Mori N. Rheumatoid arthritis with diffuse pulmonary rheumatoid nodules Pathol Int 2004;54:798-802.[Medline]
- Kobayashi T, Satoh K, Ohkawa M, Satoh A. Multiple rheumatoid nodules with rapid thin-walled cavity formation producing pneumothorax J Thorac Imaging 2005;20:47-49.[Medline]
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Abstract
Introduction
