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[Relapsing polychondritis: a case with respiratory failure]

A Kanou1, T Akimoto, S Kobayashi

  • 1Department of Internal Medicine, Tsukiyono Hospital.

Nihon Rinsho Men'Eki Gakkai Kaishi = Japanese Journal of Clinical Immunology
|February 1, 1997
PubMed
Summary
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Relapsing polychondritis can cause severe airway compromise, leading to respiratory failure. This case highlights tracheobronchomalacia as a significant complication of this rare autoimmune disease.

Area of Science:

  • Rheumatology
  • Pulmonology
  • Autoimmune Diseases

Background:

  • Relapsing polychondritis (RP) is a rare systemic autoimmune disease characterized by episodic inflammation of cartilaginous structures.
  • Airway involvement in RP, particularly tracheobronchomalacia, can lead to severe respiratory complications.

Observation:

  • A 48-year-old female patient with a history of RP presented with respiratory failure (Hugh-Jones IV-V).
  • Respiratory compromise was attributed to pharyngeal stenosis, tracheal and bronchial inflammation/deformity, and bronchial collapse.
  • Bronchoscopic examination revealed tracheobronchomalacia without lung parenchymal damage.

Findings:

  • Despite active airway disease, laboratory markers like C-reactive protein (CRP) showed only slight elevation, and anti-type II collagen antibodies were not elevated, contrasting with previous disease flares.

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  • The patient's tracheobronchomalacia was managed with immunosuppressive therapy, including hydrocortisone and cyclophosphamide.

    • Implications:

    • This case underscores the potential for significant airway morbidity in relapsing polychondritis, necessitating vigilant respiratory monitoring.
    • Tracheobronchomalacia associated with RP may present with atypical inflammatory markers, challenging diagnosis and management.
    • Understanding the association between RP and tracheobronchomalacia is crucial for managing complex respiratory and rheumatic disease presentations.