Ann Thorac Surg 2006;81:e24-e26
© 2006 The Society of Thoracic Surgeons
Case report
Long-Term Management of Polychondritis with Serial Tracheobronchial Stents
Takahiro Nakajima, MD,
Yasuo Sekine, MD, PhD,
Mio Yasuda, MD,
Kazuhiro Yasufuku, MD, PhD,
Akira Iyoda, MD, PhD,
Makoto Suzuki, MD, PhD,
Kiyoshi Shibuya, MD, PhD,
Toshihiko Iizasa, MD, PhD,
Takehiko Fujisawa, MD, PhD
*
Department of Thoracic Surgery, Graduate School of Medicine, Chiba University, Chiba, Japan
Accepted for publication February 3, 2006.
* Address correspondence to Dr Fujisawa, Department of Thoracic Surgery, Graduate School of Medicine, Chiba University 1-8-1 Inohana, chuo-ku, Chiba, 260-8670 Japan (Email: fujisawat{at}faculty.chiba-u.jp).
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Abstract
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A 56-year-old woman was referred to our hospital for airway compromise. Relapsing polychondritis was diagnosed by bronchial cartilage biopsy. Bronchoscopy showed tracheobronchial malacia due to the disappearance of bronchial cartilage as a result of chondritis. Stent insertion using a Dynamic stent (Rüsch; Kernen, Germany), Ultraflex stent (Boston Scientific International, Colombes, France) and TM stent (Fuji Systems Corp, Tokyo, Japan) was repeated for more than 7 years. Dramatic improvement of dyspnea was obtained each time. The patient survived during this period without oxygen inhalation or mechanical ventilation before she suddenly died due to suffocation caused by difficulty of sputum discharge.
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Introduction
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Relapsing polychondritis is a rare inflammatory disease of unknown cause. Although there are many clinical features, airway manifestation is considered one of the most important factors affecting prognosis. Various types of artificial stents have been reported to be efficacious for the treatment of tracheobronchial malacia due to relapsing polychondritis.
In June 1997, a 56-year-old woman noted a cough, throat pain, and discomfort. By February 1998, she was aware of hoarseness and 2 months later, she had arthralgia and chest pain develop. A chest computed tomographic scan showed stricture of both the main bronchi on inspiration (Fig 1A). In September 1998, a tracheostomy and tracheal biopsy were performed for airway stricture; the pathology revealed infiltration of inflammatory cells into the connective tissues, and degeneration and calcification of the cartilaginous tissues suggesting relapsing polychondritis.
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Fig 1. (A) Chest computed tomographic scan shows narrowing of both main bronchi on inspiration. (B) Bronchoscopy shows tracheobronchial edema, disappearance of tracheal cartilages, and osteomalacia.
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In February 1999, dyspnea and difficulty in sputum excretion were exacerbated. Bronchoscopic examination revealed an edematous larynx, narrowing of the glottis, tracheobronchial edema, disappearance of bronchial cartilage rings, and stricture of the tracheobronchus on inspiration (Fig 1B). To maintain the tracheal lumen, stent insertion was performed. Because the distance of the tracheobronchial malacia was very long, a Dynamic stent (Rüsch) was selected. Multi-planar reconstruction imaging was useful for selecting stent diameter (Fig 2A). Under general anesthesia, the Dynamic stent (Rüsch) was inserted by rigid fiber scope under fluoroscopic guidance (Fig 2B). We repeated bronchoscopic examination once a month regularly, and good patency of the central airway was confirmed for the initial 2 years. However, because the stent lumen became covered with dried sputum, the lumen had been gradually narrowing. The stent was replaced in April 2002 due to this deterioration. At this time, granulation had occurred in the bilateral main bronchi, which caused airway stenosis and suffocation. Even though ethanol injection was repeatedly performed for granulation tissue, stenosis of both bronchi worsened. We decided to insert two stents into each main bronchus. We selected an expandable metallic stent because the Dynamic stent (Rüsch) had already been inserted into each main bronchus and the lumen was quite small. In July 2003, a covered Ultraflex stent (Boston Scientific International, Colombes, France) was inserted into the right main bronchus and a noncovered Ultraflex stent was inserted into the left main bronchus. The patient suddenly suffocated in April 2005 due to narrowing of the Dynamic stent (Rüsch) caused by dried sputum and peripheral granulation tissue. On an emergency basis, we removed the Dynamic stent (Rüsch) and inserted a newly developed TM stent (Fuji Systems Corp), which is a silastic stent with special coating to avoid sputum adhesion. The TM stent (Fuji Systems Corp) remained in good condition without sputum retention; however, granulation around the metallic stent worsened (Fig 3). In particular, the covered Ultraflex stent filled with dried sputum and granulation tissue. The patient died suddenly in September 2005. The cause of death was suspected suffocation due to difficulty of sputum discharge.
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Fig 2. (A) Bronchoscopy shows the inside of the Dynamic stent (Rüsch; Kernen, Germany) after insertion. (B) Multiplanar reformation shows maintenance of tracheobronchial patency by the Dynamic stent 2 years after insertion.
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Fig 3. Internal lumen of the TM stent (Fuji Systems Corp, Tokyo, Japan), which is made from silicon with a special coating can be kept in good condition.
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Comment
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The fatal problem with relapsing polychondritis is airway manifestations [1], which are caused by malacia of trachea and bronchi due to chronic chondritis. Treatment strategy has shifted from positive airway pressure by mechanical ventilation to stent placement, and stent placement has dramatically prolonged patient survival [2]. There are many types of bronchial stents, such as the Dumon stent (Novatech, Plan de Grasse, France), expandable metallic stent, Z-stent (William Cook Europe, Bjaeverskov, Denmark), and the Dynamic stent (Rüsch). Although there are several reports of management using expandable metallic stents for relapsing polychondritis, we believe that there are no published reports of management using a silastic stent for this condition. When we choose a stent for benign disease, we must consider the benefits and faults of each stent. The advantages of an expandable metallic stent are the ease of placement by bronchoscopy under local anesthesia, strong expandability, rare migration, and preservation of mucociliary function because of epithelialization of the stent [3]. On the other hand, the expandable metallic stent is relatively easily broken, it is necessary to insert multiple stents for a case with a long stenotic stretch, and it is hard to remove the stent without complications. The Dumon stent is durable, easy to remove, and replaceable, but it is hard to fix; this stent tends to accumulate sputum in the prosthesis because of interference of airway mucociliary function [4]. In contrast, the Dynamic stent (Rüsch), which was developed by Freitag and colleagues [5], has a quasi-tracheobronchial formation indicating a "Y"-shaped silicon stent incorporating horseshoe-shaped steel struts. A flexible posterior membrane facilitates coughing. It is suitable for a case with long luminal narrowing. It can be removed and replaced; however, insertion is difficult and bio-film formation has occurred in cases with long-term placement [6]. The TM stent (Fuji Systems Corp), which is made from silicon with a special coating, has recently been developed in Japan. The conventional silicon stent has micropores that absorb moisture, and sputum is dried leading to adhesion to the internal lumen. On the other hand, the TM stent (Fuji Systems Corp) is treated with a special coating that covers the micropores to reduce absorption of moisture. Then adherence of dried sputum can be prohibited for a long time. We repeated bronchoscopic examination once a month during stent insertion. Although the Dynamic stent (Rüsch) was covered with dried sputum within 3 months, the TM stent (Fuji Systems Corp) could be kept clean for greater than 5 months. The insertion procedure for the TM stent (Fuji Systems Corp) is the same as for the Dumon stent, and the TM stent (Fuji Systems Corp) can be removed and replaced. Therefore, we believed that the TM stent (Fuji Systems Corp) was more appropriate than any other stent for such a patient who had lived a long time with her severe, benign diseases.
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References
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- Eng J, Sabanathan S. Airway complications in relapsing polychondritis Ann Thorac Surg 1991;51(4):686-692.[Abstract]
- Faul JL, Kee ST, Rizk NW. Endobronchial stenting for severe airway obstruction in relapsing polychondritis Chest 1999;116(3):825-827.[Abstract/Free Full Text]
- Sarodia BD, Dasgupta A, Mehta AC. Management of airway manifestations of relapsing polychondritiscase reports and review of literature. Chest 1999;116(6):1669-1675.[Abstract/Free Full Text]
- Dumon JF. A dedicated tracheobronchial stent Chest 1990;97(2):328-332.[Abstract/Free Full Text]
- Freitag L, Eicker R, Linz B, Greschuchna D. Theoretical and experimental basis for the development of a dynamic airway stent Eur Respir J 1994;7(11):2038-2045.[Abstract]
- Hosokawa Y, Tsujino I, Syoda T, Horikoshi A, Sawada S. Examination of expandable metallic stent removed at autopsy Respirology 2003;8(4):522-524.[Medline]
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Abstract
Introduction
