Adult Orbital Xanthogranuloma: A Case Report
Keywords:
Adult orbital xanthogranuloma, xanthogranulomatous disease, orbit, eyelid swelling, non-Langerhans cell histiocytosis, corticosteroid therapyAbstract
Objectives: To report a rare case of adult orbital xanthogranuloma (AOX).
Methods: We describe a 39-year-old Thai woman with progressive bilateral eyelid swelling and for 5 years and
yellowish discoloration of the left lower eyelid for 2 years.
Results: An earlier biopsy of the left lacrimal gland at another hospital showed dacryoadenitis. She received
oral prednisolone with good initial response, but symptoms recurred after discontinuation. At presentation, she had
bilateral eyelid swelling, yellowish discoloration of the left lower eyelid, bilateral lacrimal glands enlargement, and
right eye proptosis, without infection. Orbital computed tomography (CT) demonstrated mildly enhancing infiltrative
soft tissue in both periorbital regions and lacrimal glands, without bony destruction. A repeat biopsy of the yellowish
lesion revealed foamy macrophages and small lymphoid cells. No systemic involvement was identified. Clinical and
histopathological findings supported a diagnosis of AOX, the rarest subtype of adult orbital xanthogranulomatous
disease (AOXGD AXDO). Oral corticosteroid therapy was initiated.
Conclusions: AOX is a rare orbital disorder that should be considered in the differential diagnosis of orbital
disease. Systemic evaluation is essential due to potential associations in other subtypes. Although various treatment
approaches have been reported, no standard therapy exists. Further studies are required to clarify disease mechanisms
and management.
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The Thai Journal of Ophthalmology (TJO) is a peer-reviewed, scientific journal published biannually for the Royal College of Ophthalmologists of Thailand. The objectives of the journal is to provide up to date scientific knowledge in the field of ophthalmology, provide ophthalmologists with continuing education, promote cooperation, and sharing of opinion among readers.
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