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A male patient with proptosis and palatal perforation: A diagnostic challenge
Peyman Mottaghi1, Sam Mirfendereski2, Farshad Riahi2
1Department of Rheumatology, Isfahan University of Medical Sciences, Isfahan, Iran.
Abstract:
Granulomatosis with polyangiitis (Wegener's granulomatosis), affects many systems and can show up in a variety of ways. Sometimes, only a few organs are affected. Rare complications of this vasculitis syndrome include a retroorbital pseudotumor with proptosis due to chronic tissue inflammation, as well as necrosis with subsequent palate perforation. We present a 59-year-old male who had multiple ophthalmologic evaluations for chronic pain, redness, and proptosis in the right eye. During the workup for eye problems, he underwent surgery for chronic right nasal obstruction and bloody discharge. After 2 weeks, he noticed pain and ulceration in his hard palate that rapidly transformed into a perforation of the palate and was finally referred to a rheumatologist due to increased erythrocyte sedimentation rate. A CT scan was requested for the patient, which showed an orbital mass and palatal perforation. A biopsy of the mass behind the eye showed that it was inflamed across the membrane and had fibrinoid necrosis, which is a sign of necrotizing vasculitis.
Insights
Granulomatosis with polyangiitis can cause rare eye and palate complications. This case highlights necrotizing vasculitis presenting as proptosis and palatal perforation.
Area of Science:
- Rheumatology
- Ophthalmology
- Otorhinolaryngology
Background:
- Granulomatosis with polyangiitis (Wegener's granulomatosis) is a systemic vasculitis affecting multiple organs.
- Clinical manifestations can be diverse, sometimes involving only a few organs.
- Rare complications include orbital pseudotumor and palatal necrosis.
Observation:
- A 59-year-old male presented with chronic right eye pain, redness, and proptosis.
- Nasal obstruction and discharge led to surgery, followed by palate ulceration and perforation.
- Elevated erythrocyte sedimentation rate prompted rheumatologic referral.
Findings:
- CT scan revealed an orbital mass and palatal perforation.
- Biopsy of the retroorbital mass showed inflammation and fibrinoid necrosis.
- Histopathology confirmed necrotizing vasculitis.
Implications:
- This case underscores the varied presentations of Granulomatosis with polyangiitis.
- Early recognition of rare complications like proptosis and palatal perforation is crucial.
- Multidisciplinary evaluation is essential for diagnosing and managing systemic vasculitis.
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