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Xanthogranulomatous disease in the lacrimal gland
  1. J A Sivak-Callcott1,
  2. W K Lim2,
  3. L L Seah2,
  4. J Oestreicher3,
  5. D Rossman3,
  6. N Nijhawan3,
  7. J Rootman4,5,
  8. V White5,
  9. H J Williams6,
  10. W W L Chang6,
  11. A DiBartolomeo7,*,
  12. D Howarth8
  1. 1Department of Ophthalmology, West Virginia University, Morgantown, WV, USA
  2. 2Singapore National Eye Centre, Singapore
  3. 3Department of Ophthalmology, University of Toronto, Toronto, Ontario, Canada
  4. 4Department of Ophthalmology and Visual Sciences, University of British Colombia, Vancouver, BC, Canada
  5. 5Department of Pathology, University of British Colombia, Vancouver, BC, Canada
  6. 6Department of Pathology, West Virginia University, Morgantown, WV, USA
  7. 7Department of Rheumatology, West Virginia University, Morgantown, WV, USA
  8. 8Department of Pathology, University of Toronto, Toronto, Ontario, Canada
  1. Correspondence to: Jennifer A Sivak-Callcott MD, West Virginia University Eye Institute, PO Box 9193, Morgantown, WV 26506, USA; jsivakhsc.wvu.edu

https://doi.org/10.1136/bjo.2004.063578

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    We report three cases of adult lacrimal gland xanthoganulomatous disease that demonstrate the spectrum of this disorder and provide insight into immune dysfunction.

    Case 1

    A 23 year old asthmatic female had 1 year of bilateral, painless, lacrimal gland masses from polyclonal B cell (CD 20+) infiltration (fig 1A, B) The patient was asymptomatic for 18 months after external beam radiation (25 Gy in 10 fractions). While 7 months pregnant, painless lacrimal gland enlargement recurred, as firm, yellow, nodular masses (fig 1C). A second biopsy showed foamy histiocytes, Touton giant cells, and lymphoid infiltrate without necrobiosis (fig 1D). The orbital masses have remained stable 2 years after corticosteroids and surgical debulking. Systemic involvement included breast MALT type lymphoma 4 years after presentation.

    Figure 1

     (A) T1 weighted axial magnetic resonance imaging (MRI) showing bilateral lacrimal gland enlargement. (B) Initial lacrimal gland biopsy showing a dense lymphocytic infiltrate with preserved ducts (haematoxylin and eosin stain, original magnification 4×). (C) Clinical photograph of yellow, recurrent mass, right orbit, 2 years after radiation treatment. (D) Second lacrimal gland biopsy showing Touton giant cells, foamy histiocytes, lymphocytes, and plasma cells (haematoxylin and eosin stain, original magnification 60×). (E) Clinical photograph of patient showing improvement 1 year after treatment.

    Case 2

    A 49 year old Brunei …

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    Footnotes

    • ↵* Published posthumously.

    • None of the authors have any financial interests regarding this paper.