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Wegener's presenting as submandibular swelling

S Singh1, D Kaura, S Kumari

  • 1Department of Internal Medicine, Postgraduate Institute of Medical Education and Research, Chandigarh, India.

Sarcoidosis, Vasculitis, and Diffuse Lung Diseases : Official Journal of WASOG
|March 1, 1997
PubMed
Summary
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Bilateral submandibular gland swelling can be an early sign of limited Wegener's disease. Diagnosis involves specific antibodies and biopsy, with treatment including immunosuppression.

Area of Science:

  • Rheumatology
  • Immunology
  • Otorhinolaryngology

Background:

  • Wegener's disease, now known as granulomatosis with polyangiitis (GPA), is a systemic vasculitis.
  • Limited forms of GPA may present with unusual initial symptoms.
  • Submandibular gland involvement is an uncommon initial presentation of GPA.

Observation:

  • A patient presented with bilateral submandibular gland swelling as the primary symptom.
  • Diagnostic workup was initiated due to the unusual presentation.

Findings:

  • Diagnosis confirmed by positive anti-neutrophil cytoplasmic antibody (ANCA) testing.
  • Submandibular gland biopsy revealed dense inflammatory infiltrate with lymphocytes and non-caseating granulomas.
  • Transbronchial biopsy also showed ill-formed granulomas, supporting the diagnosis.

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Implications:

  • Highlights the importance of considering GPA in patients with isolated salivary gland enlargement.
  • Demonstrates the utility of ANCA testing and tissue biopsy in diagnosing limited GPA.
  • Successful management achieved with immunosuppressive therapy, indicating disease responsiveness.