{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,18]],"date-time":"2026-08-18T16:31:59Z","timestamp":1787070719635,"version":"build-2736575974"},"reference-count":11,"publisher":"Ovid Technologies (Wolters Kluwer Health)","issue":"2","license":[{"start":{"date-parts":[[2025,5,1]],"date-time":"2025-05-01T00:00:00Z","timestamp":1746057600000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-sa\/4.0"}],"content-domain":{"domain":["lww.com","ovid.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2025,5]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Juvenile xanthogranuloma (JXG) is a rare, benign granulomatous disorder predominantly affecting young children, characterized by the presence of Touton giant cells. While typically presenting as cutaneous lesions, JXG can also involve the eyes, potentially leading to severe complications such as hyphema and glaucoma. Here, we present the case of an 11-month-old girl with multifaceted systemic and ocular manifestations of JXG. She presented with unilateral redness, watering, photophobia, and hyperpigmented nodules distributed across her body. Ocular examination revealed hyphema, heterochromia iridis, fibrinous reaction, and elevated IOP with signs of glaucomatous optic neuropathy. Despite initial treatment with topical steroids and antiglaucoma medications, the patient experienced recurrent hyphema. Further evaluation identified peripheral anterior synechiae and delicate vessels spanning the trabecular meshwork. Histopathological analysis of the skin nodules confirmed the diagnosis of JXG, with systemic involvement noted in the spleen and other organs. Multidisciplinary management, including chemotherapy, resulted in the regression of hyphema and the fibrinous reaction. This case underscores the importance of comprehensive evaluation and a multidisciplinary approach in managing JXG, highlighting the challenges faced by ophthalmologists in preventing complications such as rebleeding and refractory glaucoma.<\/jats:p>","DOI":"10.4103\/ojo.ojo_296_24","type":"journal-article","created":{"date-parts":[[2025,6,24]],"date-time":"2025-06-24T07:00:30Z","timestamp":1750748430000},"page":"235-238","update-policy":"https:\/\/doi.org\/10.1097\/lww.0000000000001000","source":"Crossref","is-referenced-by-count":0,"title":["Hyphaema, heterochromia and hyperpigmented cutaneous nodules"],"prefix":"10.4103","volume":"18","author":[{"given":"Nidhi","family":"Mamtani","sequence":"first","affiliation":[{"name":"Department of Ophthalmology, All India Institute of Medical Sciences, Nagpur, Maharashtra, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sagarika","family":"Snehi","sequence":"additional","affiliation":[{"name":"Department of Ophthalmology, All India Institute of Medical Sciences, Nagpur, Maharashtra, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Puja Hingorani","family":"Bang","sequence":"additional","affiliation":[{"name":"Department of Ophthalmology, All India Institute of Medical Sciences, Nagpur, Maharashtra, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kanishk","family":"Singh","sequence":"additional","affiliation":[{"name":"Department of Ophthalmology, All India Institute of Medical Sciences, Nagpur, Maharashtra, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rajesh","family":"Pattebahadur","sequence":"additional","affiliation":[{"name":"Department of Ophthalmology, All India Institute of Medical Sciences, Nagpur, Maharashtra, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Meghana","family":"Kandi","sequence":"additional","affiliation":[{"name":"Department of Ophthalmology, All India Institute of Medical Sciences, Nagpur, Maharashtra, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sachin","family":"Chaudhari","sequence":"additional","affiliation":[{"name":"Department of Pathology, All India Institute of Medical Sciences, Nagpur, Maharashtra, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pankaj","family":"Dwivedi","sequence":"additional","affiliation":[{"name":"Department of Paediatric Oncology, National Cancer Institute, Nagpur, Maharashtra, India"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"276","published-online":{"date-parts":[[2025,6,24]]},"reference":[{"key":"R1-20250628","doi-asserted-by":"crossref","first-page":"295","DOI":"10.1016\/j.ijporl.2011.11.010","article-title":"Nonlipidized juvenile xanthogranuloma: An unusual variant with a potential diagnostic pitfall","volume":"76","author":"Batista","year":"2012","journal-title":"Int J Pediatr Otorhinolaryngol"},{"key":"R2-20250628","doi-asserted-by":"crossref","first-page":"579","DOI":"10.1097\/00000478-200305000-00003","article-title":"Juvenile xanthogranulomas in the first two decades of life: A clinicopathologic study of 174 cases with cutaneous and extracutaneous manifestations","volume":"27","author":"Dehner","year":"2003","journal-title":"Am J Surg Pathol"},{"key":"R3-20250628","doi-asserted-by":"crossref","first-page":"1011","DOI":"10.1016\/0002-9394(65)92808-4","article-title":"Ocular lesions of juvenile xanthogranuloma. Nevoxanthoedothelioma","volume":"60","author":"Zimmerman","year":"1965","journal-title":"Am J Ophthalmol"},{"key":"R4-20250628","doi-asserted-by":"crossref","first-page":"399","DOI":"10.1159\/000515151","article-title":"Symmetrical facial giant plaque-type juvenile xanthogranuloma: Case report and review of the literature","volume":"13","author":"Itin","year":"2021","journal-title":"Case Rep Dermatol"},{"key":"R5-20250628","doi-asserted-by":"crossref","first-page":"197","DOI":"10.5114\/pdia.2014.40918","article-title":"Juvenile xanthogranuloma: A rare benign histiocytic disorder","volume":"31","author":"Szczerkowska-Dobosz","year":"2014","journal-title":"Postepy Dermatol Alergol"},{"key":"R6-20250628","doi-asserted-by":"crossref","first-page":"637","DOI":"10.1111\/pde.14174","article-title":"Juvenile xanthogranulomas: Examining single, multiple, and extracutaneous presentations","volume":"37","author":"So","year":"2020","journal-title":"Pediatr Dermatol"},{"key":"R8-20250628","doi-asserted-by":"crossref","first-page":"445","DOI":"10.1016\/S0190-9622(96)90437-5","article-title":"The risk intraocular juvenile xanthogranuloma: Survey of current practices and assessment of risk","volume":"34","author":"Chang","year":"1996","journal-title":"J Am Acad Dermatol"},{"key":"R9-20250628","doi-asserted-by":"crossref","first-page":"73","DOI":"10.1016\/j.ajoc.2016.12.003","article-title":"Intraocular juvenile xanthogranuloma of the iris in an adult patient","volume":"5","author":"Flockerzi","year":"2017","journal-title":"Am J Ophthalmol Case Rep"},{"key":"R10-20250628","doi-asserted-by":"crossref","first-page":"157","DOI":"10.4137\/CMO.S6686","article-title":"Neonatal systemic juvenile xanthogranuloma with an ominous presentation and successful treatment","volume":"5","author":"Fan","year":"2011","journal-title":"Clin Med Insights Oncol"},{"key":"R11-20250628","doi-asserted-by":"crossref","first-page":"130","DOI":"10.1002\/pbc.21523","article-title":"Treatment of juvenile xanthogranuloma","volume":"51","author":"Stover","year":"2008","journal-title":"Pediatr Blood Cancer"},{"key":"R12-20250628","doi-asserted-by":"crossref","first-page":"371","DOI":"10.1097\/00043426-200406000-00007","article-title":"Successful treatment of congenital systemic juvenile xanthogranuloma with Langerhans cell histiocytosis-based chemotherapy","volume":"26","author":"Nakatani","year":"2004","journal-title":"J Pediatr Hematol Oncol"}],"container-title":["Oman Journal of Ophthalmology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.ovid.com\/10.4103\/ojo.ojo_296_24","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,8,18]],"date-time":"2026-08-18T15:35:22Z","timestamp":1787067322000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.ovid.com\/10.4103\/ojo.ojo_296_24"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,5]]},"references-count":11,"journal-issue":{"issue":"2","published-print":{"date-parts":[[2025]]}},"URL":"https:\/\/doi.org\/10.4103\/ojo.ojo_296_24","relation":{},"ISSN":["0974-620X","0974-7842"],"issn-type":[{"value":"0974-620X","type":"print"},{"value":"0974-7842","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,5]]},"assertion":[{"value":"2024-09-14","name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-04-21","name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}}]}}