Journal of the Korean Ophthalmological Society 2005;46(10):1733-1736.
Published online October 31, 2005.
Subconjunctival Juvenile Xanthogranuloma Involving the Limbus Associated with Neurofibromatosis.
Young Woo Suh, Chung Hoon Oh, Hyo Myung Kim
1Department of Ophthalmology, Korea University College of Medicine, Seoul, Korea. hyomkim@ns.kumc.or.kr
2Department of Ophthalmology, KEPCO Medical Foundation, Han-Il General Hospital, Seoul, Korea.
신경섬유종증을 동반한 각막 가장자리를 침범하는 결막하 소아황색육아종증 1예
서영우1,오청훈2,김효명1
Department of Ophthalmology, Korea University College of Medicine1, Seoul, Korea Department of Ophthalmology, KEPCO Medical Foundation, Han-Il General Hospital2, Seoul, Korea
Correspondence:  Young-Woo Suh, M.D.1
Abstract
PURPOSE
To report a case of isolated subconjunctival juvenile xanthogranuloma invading the corneal limbus in a patient with neurofibromatosis. METHODS: A 2-year-old boy visited the hospital with a conjunctival yellow-brown, non-tender mass of the left eye, which had been found 2 months earlier. Anterior segment examination revealed a 9 x 7 mm-sized subconjunctival elevated mass with an ill-defined border, invading the supero-temporal limbus. Additional ophthalmic examinations did not show any abnormal findings. In the trunk and extremities, there were multiple soft nodules and brown macules. An excisional biopsy of the subconjunctival mass, histopathologic and immunohistochemical examinations were performed. RESULTS: The mass was adhered to the episcleral tissue, without scleral or conjunctival involvement. The histological examination showed a granulomatous lesion consisting of multiple lipid-laden macrophages and other inflammatory cells. The immunohistochemical study showed positive staining for CD68 and negative staining for S100 protein. The multiple soft nodules and brown macules in the trunk and extremities were identified as neurofibromas and caf -au-lait macules by a dermatologist, and the patient was diagnosed with neurofibromatosis. CONCLUSIONS: We report a rare case of subconjunctival juvenile xanthogranuloma involving the limbus, associated with neurofibromatosis.
Key Words: Juvenile xanthogranuloma;Neurofibromatosis


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