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Open Access Highly Accessed Case report

Old-age-onset subconjunctival juvenile xanthogranuloma without limbal involvement

Mo-Sae Kim, So-Ah Kim and Ho-Seok Sa*

Author Affiliations

Department of Ophthalmology, Asan Medical Center, University of Ulsan, College of Medicine, 388-1 Pungnap-2-dong, Songpa-gu, 138-736 Seoul, Korea

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BMC Ophthalmology 2014, 14:24  doi:10.1186/1471-2415-14-24

Published: 6 March 2014

Abstract

Background

Juvenile xanthogranuloma (JXG) is a benign idiopathic cutaneous granulomatous tumor occurring primarily in infants less than 1 year old, and less commonly found in older children and adults. To date, however, there have been no reports of patients aged >50 years with cornealscleral JXG without limbal involvement. We describe here a 58-year-old woman with subconjunctival JXG without limbal involvement.

Case presentation

A 58-year-old female was referred for evaluation of a subconjunctival mass in her left eye, found incidentally 2 weeks earlier. Examination revealed a protruding yellow-orange subconjunctival mass just below the 6-o’clock limbus of her left eye, measuring 6.0 × 4.5 mm, but not extending into the cornea. The overlying conjunctival epithelium was intact, and a feeding vessel was observed between the mass and the episclera. The subconjunctival lesion was excised under local anesthesia, by dissecting the mass from the overlying conjunctiva and underlying sclera. The conjunctiva was reattached to the sclera without creating a bare area. Hematoxylin and eosin-stained sections showed that the mass was a mixed inflammatory lesion containing dense infiltrations of epithelioid histiocytes with foamy cytoplasm, lymphocytes, and plasma cells, as well as multinucleated Touton giant cells with the characteristic circumferential ring of nuclei. Immunohistochemical staining showed that the lesion was positive for the macrophage marker CD68 and negative for the Langerhans cell markers S-100 protein and CD1a, indicating that the lesion was a xanthogranuloma. The patient has been followed up for 12-months without recurrence.

Conclusions

JXG can occur as a solitary subconjunctival mass even in older adults, and immunohistochemistry is useful in differential diagnosis. Simple excision with careful dissection may be effective for subconjunctival JXG.

Keywords:
Juvenile; Xanthogranuloma; Subconjunctival