Refractory otitis media: an unusual presentation of childhood granulomatosis with polyangiitis

Pediatr Pulmonol. 2014 Mar;49(3):E21-4. doi: 10.1002/ppul.22746. Epub 2013 Jan 28.

Abstract

Childhood granulomatosis with polyangiitis (cGPA), previously known as Wegener's granulomatosis, is a rare, potentially fatal necrotizing vasculitis, the symptoms of which overlap with infection. We present a 16-year-old girl who, following 6 months of treatment for persistent middle ear effusion with progressive sensorineural hearing loss, developed rapidly progressing pneumonia, with pleural effusion, and multiple cavitatory lung lesions. Investigations demonstrated high titer c-ANCA and nasal septal biopsy confirmed the diagnosis of cGPA. This case highlights the difficulty in diagnosing cGPA and the potentially life-threatening consequences of failing to do so.

Keywords: Wegener's granulomatosis; atypical pneumonia; childhood vasculitis; chronic otitis media; hearing loss.

Publication types

  • Case Reports

MeSH terms

  • Adolescent
  • Antibodies, Antineutrophil Cytoplasmic / blood
  • Chronic Disease
  • Cyclophosphamide / therapeutic use
  • Diagnostic Errors*
  • Ear Diseases / diagnosis
  • Ear Diseases / drug therapy
  • Ear Diseases / etiology
  • Female
  • Granulomatosis with Polyangiitis / complications
  • Granulomatosis with Polyangiitis / diagnosis*
  • Granulomatosis with Polyangiitis / drug therapy
  • Hearing Loss, Mixed Conductive-Sensorineural / etiology
  • Humans
  • Immunosuppressive Agents / therapeutic use
  • Lung Diseases / diagnosis
  • Lung Diseases / drug therapy
  • Lung Diseases / etiology
  • Methylprednisolone / therapeutic use
  • Nose Diseases / diagnosis
  • Nose Diseases / drug therapy
  • Nose Diseases / etiology
  • Otitis Media / diagnosis*

Substances

  • Antibodies, Antineutrophil Cytoplasmic
  • Immunosuppressive Agents
  • Cyclophosphamide
  • Methylprednisolone