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Obstructive respiratory failure in cicatricial pemphigoid.

C R de Carvalho1, M B Amato, L M Da Silva

  • 1Pulmonary Division, Hospital das Clinicas, São Paulo, Brasil.

Thorax
|July 1, 1989
PubMed
Summary

Cicatricial pemphigoid, a rare autoimmune condition, caused fatal respiratory failure in a young woman due to lower airway scarring. This case highlights the severe, previously unreported impact on the trachea and bronchi.

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Area of Science:

  • Pulmonology
  • Dermatology
  • Pathology

Background:

  • Cicatricial pemphigoid is a chronic autoimmune blistering disease primarily affecting mucous membranes.
  • While typically involving the oral and ocular mucosa, laryngeal and pharyngeal involvement has been documented.
  • Lower airway (trachea and bronchi) involvement by cicatricial pemphigoid has not been previously reported.

Observation:

  • A 20-year-old female patient presented with respiratory distress.
  • Diagnostic investigations revealed severe cicatricial changes in the trachea and bronchi.
  • The patient ultimately succumbed to respiratory failure.

Findings:

  • This case represents the first documented instance of cicatricial pemphigoid affecting the lower respiratory tract, specifically the trachea and bronchi.

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  • Histopathological examination confirmed the diagnosis of cicatricial pemphigoid with extensive submucosal fibrosis and inflammation in the airway tissues.
  • The cicatricial changes led to significant airway obstruction and subsequent respiratory failure.

    • Implications:

    • This case expands the known spectrum of cicatricial pemphigoid, demonstrating its potential to cause life-threatening lower airway compromise.
    • It underscores the importance of considering cicatricial pemphigoid in the differential diagnosis of unexplained airway stenosis, even in young patients.
    • Further research is warranted to understand the pathogenesis and optimal management strategies for this rare but severe manifestation.