A complicated case of relapsing polychondritis: Case report

一例复杂的复发性多软骨炎病例报告

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Abstract

RATIONALE: Relapsing polychondritis (RP) is a rare inflammatory disease that presents clinically with severe dyspnea and respiratory distress. Differentiating it from amyloidosis can be diagnostically challenging. In cases of severe respiratory distress, timely bronchoscopic intervention, including laser tracheobronchoplasty and glucocorticoids (GCs) spraying, is crucial. Efficacy of this combined approach has not been reported to treat onset of RP. PATIENT CONCERNS: We report a complex case of RP in a 56-year-old male. Over the past 2 years, the patient experienced recurrent episodes of dyspnea, cough, and expectoration. Chest computed tomography (CT) scans indicated tracheal wall thickening, and airway stenosis and deformation. Recently, the patient presented with severe dyspnea and respiratory distress, leading to a definitive diagnosis of RP. DIAGNOSES: The diagnosis of RP was established based on the patient's medical history, CT scans, clinical signs (noting cartilage collapse and swelling in the ears, nasal collapse), and endoscopic findings (severe airway stenosis and collapse). INTERVENTIONS: The patient was treated with noninvasive ventilation, budesonide, GCs for anti-inflammatory effects, and moxifloxacin for anti-infective therapy. Due to worsening conditions, invasive ventilation was used for 4 days. An emergency bronchoscopic examination was performed, followed by sputum aspiration, laser tracheobronchoplasty for airway reshaping, and endotracheal intubation to stabilize oxygen saturation and alleviate symptoms. Cyclophosphamide was administered. OUTCOMES: The patient experienced significant relief from dyspnea, and no recurrence was observed within 1 month after the completion of treatment. LESSONS: When RP is exacerbated by infection, leading to progressive dyspnea and causing acute respiratory distress, it is difficult to distinguish based on CT scans. In situations where pathological results are not promptly available, endoscopic diagnosis and intervention are merited, we recommend performing laser tracheobronchoplasty early during bronchoscopy and spraying GCs to reduce mucosal edema.

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