Cases reported "Polychondritis, Relapsing"

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1/40. A new role for magnetic resonance imaging in the diagnosis of laryngeal relapsing polychondritis.

    BACKGROUND: Relapsing polychondritis involving the upper airway is a rare cause of airflow obstruction and hoarseness. The diagnosis of relapsing polychondritis depends on clinical signs, characteristic findings on cartilage biopsy, and response to treatment. Delays in diagnosis and treatment can increase the morbidity and mortality of the disease. methods: We present a case report of primary laryngeal relapsing polychondritis. RESULTS: Serologic testing, direct laryngoscopy, and endoscopic biopsy could not establish the diagnosis of relapsing polychondritis. magnetic resonance imaging (MRI) examination demonstrated findings consistent with the histopathological diagnosis obtained on open biopsy. Follow-up MRI after treatment showed resolution of the initial findings. CONCLUSION: This case demonstrates the usefulness of MRI in the diagnosis and management of relapsing polychondritis involving the upper airway.
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ranking = 1
keywords = airway, obstruction
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2/40. Endobronchial stenting for severe airway obstruction in relapsing polychondritis.

    Airway complications of relapsing polychondritis (RP), including tracheobronchial stenosis, can be fatal. This paper describes a life-saving technique (placement of multiple metallic endobronchial stents under conscious sedation) to prevent life-threatening airway closure in a 50-year-old woman with RP. Using fluoroscopic and bronchoscopic guidance, a tracheal stent and three endobronchial metallic stents were deployed in the central airways, with good functional outcome. There were no complications. In critical airway compromise caused by RP, the insertion of endobronchial stents can result in improved symptoms, pulmonary function, and a return to daily activities, without the use of tracheotomy and mechanical ventilation.
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ranking = 11.126615685701
keywords = airway obstruction, airway, obstruction
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3/40. Management of airway manifestations of relapsing polychondritis: case reports and review of literature.

    STUDY OBJECTIVE: To report the first series of patients with severe airway manifestations of relapsing polychondritis (RP) that were managed successfully with self-expandable metallic stents, and to review the literature. DESIGN: Retrospective review of medical records, and current clinical follow-up. SETTING: Tertiary care referral hospital. patients: All patients with airway manifestations of RP that were managed with self-expandable metallic stents at our institution. RESULTS: All five patients (four women and one man; age, 40 to 69 years old) had severe airway manifestations, and three of them required mechanical ventilation. spirometry with flow-volume curves showed severe combined obstructive and restrictive ventilatory defects. bronchoscopy revealed dynamic collapse of the proximal airways. diagnosis was made 8 months to 13 years after the first symptom of the disease. Pharmacotherapy included prednisone, methotrexate, cyclosporine, and dapsone. A total of 17 self-expandable metallic stents of varying sizes were placed using flexible bronchoscope from 4 to 19 years after the first symptom. The overall outcome was favorable in four patients. Three patients have survived without ventilatory support 16 to 18 months following the first stent placement, and the fourth patient survived for 20 months without ventilatory support before she died. The fifth patient, who was receiving mechanical ventilation, died in 1 week probably due to persistent dynamic collapse of the airways distal to the stents. CONCLUSION: Self-expandable metallic tracheobronchial stents should be considered in the management of airway manifestations of RP, especially in patients who require mechanical ventilation.
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ranking = 4.768067234497
keywords = airway
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4/40. pulmonary fibrosis with intractable pneumothorax: new pulmonary manifestation of relapsing polychondritis.

    Relapsing Polychondritis is a rare disease which causes the repetitive inflammation of cartilage and connective tissues. Although the large airway is sometimes involved and the stenosis of them often influences the prognosis of the patients, there have been few reports concerning the manifestation of the peripheral lung. A 60-year-old man with pulmonary fibrosis was admitted to a regional hospital due to sudden deafness, and then he suffered from relapsing polychondritis. During the steroid therapy, he also suffered from bilateral pneumothoraces. His computed tomogram revealed many bilateral bullae, emphysematous changes, and fibrotic changes in bilateral lungs. The mechanism of generating peripheral pulmonary manifestations is also discussed.
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ranking = 0.4768067234497
keywords = airway
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5/40. Anesthetic management of a patient with relapsing polychondritis--a case report.

    Relapsing polychondritis is a rare multisystem disorder of uncertain etiology. It is characterized by recurrent and progressive destruction of both elastic and hyaline cartilages as well as connective tissue. Respiratory tract involvement is associated with high mortality and morbidity. General anesthesia may be required in these patients for tracheostomy, bronchoscopy, nasal reconstruction, aortic valve replacement, and recent tracheobronchial stenting which they usually sustain. tracheostomy was once the most likely surgical procedure in relapsing polychondritis. However, this procedure is only effective in patients with upper subglottic involvement. In cases of extensive tracheobronchial involvement, tracheostomy is ineffective because the distant tracheal collapse below the tracheostomy is still unresolved. We would like to report a case of relapsing polychondritis with tracheobronchial involvement, who underwent an emergent tracheostomy. tracheostomy with continuous positive airway pressure (CPAP) effectively improved her airway collapse. Herein, we also discuss the anesthetic management after review of the current literature.
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ranking = 0.95361344689939
keywords = airway
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6/40. Metallic tracheal stents: complications associated with long-term use in the upper airway.

    The use of metallic airway stents for the treatment of benign airway stenosis is increasingly advocated; however, the long-term safety and efficacy of these devices has not been established. Three case studies involving late but significant proximal tracheal stent complications are reported: 1 related to proximal mechanical stent failure and 2 related to obstructing granulation tissue and stenosis at the proximal stent orifice. Placement of these stents in close proximity to the cricotracheal junction is thought to be primarily responsible for these complications as a result of the excess shearing forces created at the stent-mucosal interface by the differential motion of the stent relative to the rigid subglottic airway and the more distensible trachea. Although the use of metallic stents remains appropriate in cases in which there is a defined and relatively short-term end point for treatment, caution is urged in using them in the proximal trachea for long-term management of benign airway disease.
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ranking = 3.8144537875976
keywords = airway
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7/40. A case of relapsing polychondritis involving placement of an expandable metallic stent.

    Relapsing polychondritis is a relatively uncommon disease characterized by recurrence of progressive inflammation of cartilaginous structures. Laryngotracheal involvement occurs in 50-70% of cases, and occasionally results in acute airway obstruction. We reported a case of relapsing polychondritis with severe tracheal stenosis that was treated by the placement of two expandable metallic stents. A 58-year-old man was hospitalized with fever and breathing difficulties. A tracheostomy was performed and a silicone T-tube was placed. A diagnosis of relapsing polychondritis was made based on the biopsy from auricular and tracheal cartilages. Two expandable metallic stents were placed in the trachea to bilateral main bronchus. This dilated the airway lumen and resulted in the dyspnea disappearing completely. However, 18 months later a sudden massive hemorrhage occurred through the tracheostoma, and he died of respiratory failure. autopsy showed a tracheoinnominate artery fistula. We discuss the management of relapsing polychondritis with severe tracheal stenosis.
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ranking = 2.9008556022876
keywords = airway obstruction, airway, obstruction
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8/40. Obstructive sleep apnoea/hypopnoea as the initial presentation of relapsing polychondritis.

    Relapsing polychondritis (RP) is a relatively rare rheumatological condition, which can affect the upper airway and prove potentially life threatening. We report the first case of RP presenting initially as the obstructive sleep apnoea/hypopnoea syndrome, discuss management and review the extant literature.
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ranking = 0.4768067234497
keywords = airway
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9/40. Relapsing polychondritis: reversible airway obstruction is not always asthma.

    Relapsing polychondritis (RP) is a rare disease characterized by recurrent inflammation of cartilaginous and other proteoglycan-rich tissues. Respiratory tract involvement is a common cause of morbidity and mortality in RP. We describe a patient whose clinical features at onset of disease were typical of asthma. Later, the patient developed symptoms and signs characteristic of RP. tracheobronchomalacia necessitated airway support by stenting. The possibility that airway obstruction in the initial stages of RP is due to airway inflammation and that early, aggressive immunosuppressive treatment of RP may delay or prevent irreversible cartilaginous destruction and airway collapse are discussed.
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ranking = 13.550664564539
keywords = airway obstruction, airway, obstruction
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10/40. Relapsing polychondritis: an unusual cause of painful auricular swelling.

    Auricular pain, redness, and swelling are usually the result of infectious cellulitis. However, relapsing polychondritis is another cause of this symptom complex and is the result of an autoimmune phenomenon. It presents with inflammation and destruction of both articular and nonarticular cartilage, with the external ear and joint cartilage most often involved. Although usually indolent with multiple acute exacerbations, relapsing polychondritis can be rapidly fatal if the airway or cardiovascular system is involved. We present a case of relapsing polychondritis initially mistaken for auricular cellulitis. The clinical manifestations and course of relapsing polychondritis are reviewed, along with the emergency department management.
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ranking = 0.4768067234497
keywords = airway
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