Publication:
Juvenile Xanthogranuloma Presented with Buphthalmos and Corneal Clouding in Neonatal Period: A Case Report

Simple item page
dc.contributor.authorDERİCİOĞLU, VOLKAN
dc.contributor.authorSEVİK, MEHMET ORKUN
dc.contributor.authorERASLAN, MUHSİN
dc.contributor.authorsDERİCİOĞLU V., SEVİK M. O. , ERASLAN M., Dirican B., Yücelten D., Cinel L.
dc.date.accessioned2022-11-02T13:52:12Z
dc.date.available2022-11-02T13:52:12Z
dc.date.issued2022-05-01
dc.description.abstractAim: To report an ocular juvenile xanthogranuloma (JXG) case presented with buphthalmos, corneal cloudiness, and normal intraocular pressure (IOP) in the neonatal period and treated with Ahmed glaucoma valve (AGV) implantation. Background: JXG is a rare disorder predominantly seen in infants, but the neonatal presentation is extraordinary. Although spontaneous hyphema is a common presenting sign in JXG, buphthalmos and corneal opacity in the neonatal period were reported only in one case, which had high IOP values at presentation. Case presentation: Sixteen-day-old male patient presented with buphthalmos, diffuse corneal clouding, and 11 mm Hg of IOP value in the right eye. IOP increased to 28 mm Hg three weeks later, and spontaneous hyphema developed, which did not respond to antiglaucomatous medications and topical corticosteroids. AGV was implanted, and the IOP decreased to 13 mm Hg postoperatively. In the follow-ups, numerous firm yellowish nodules were noticed on the patient’s skin during the examination under general anesthesia. Histopathological examination of the skin nodules was compatible with the diagnosis of JXG. Lens subluxation and phacodonesis were developed during the follow-up and were managed with pars plana lensectomy. After a silent period of 3 months, epithelial ingrowth was determined around the side port entrance. Unfortunately, the ingrowth did not respond to cryotherapy and resulted in phthisis bulbi. Pathological evaluation of the enucleated phthisic eye revealed posterior segment involvement. Conclusion: Ocular JXG can be present with buphthalmos, corneal opacity, and normal IOP values without any skin lesions in the neonatal period. Neonatal presentation of JXG may be associated with limited medical therapy response and aggressive disease course. Clinical significance: This case report introduces the second ocular JXG case, which presented with buphthalmos and corneal cloudiness, and the third pathologically proven posterior segment involvement of JXG in the literature.
dc.identifier.citationDERİCİOĞLU V., SEVİK M. O. , ERASLAN M., Dirican B., Yücelten D., Cinel L., "Juvenile Xanthogranuloma Presented with Buphthalmos and Corneal Clouding in Neonatal Period: A Case Report", Journal of Current Glaucoma Practice, cilt.16, sa.2, ss.128-131, 2022
dc.identifier.doi10.5005/jp-journals-10078-1369
dc.identifier.endpage131
dc.identifier.issn0974-0333
dc.identifier.issue2
dc.identifier.startpage128
dc.identifier.urihttps://avesis.marmara.edu.tr/api/publication/f11f8f2b-b9a5-4b8e-a617-2b6a0fb99d6f/file
dc.identifier.urihttps://hdl.handle.net/11424/282790
dc.identifier.volume16
dc.language.isoeng
dc.relation.ispartofJournal of Current Glaucoma Practice
dc.rightsinfo:eu-repo/semantics/openAccess
dc.subjectTıp
dc.subjectCerrahi Tıp Bilimleri
dc.subjectGöz Hastalıkları ve Cerrahisi
dc.subjectSağlık Bilimleri
dc.subjectMedicine
dc.subjectSurgery Medicine Sciences
dc.subjectEye Diseases and Surgery
dc.subjectHealth Sciences
dc.subjectKlinik Tıp (MED)
dc.subjectKlinik Tıp
dc.subjectOFTALMOLOJİ
dc.subjectClinical Medicine (MED)
dc.subjectCLINICAL MEDICINE
dc.subjectOPHTHALMOLOGY
dc.subjectOftalmoloji
dc.subjectOphthalmology
dc.subjectAhmed glaucoma valve
dc.subjectBuphthalmos
dc.subjectCase report
dc.subjectHistopathology
dc.subjectJuvenile xanthogranuloma
dc.subjectNeonatal
dc.subjectPosterior segment involvement
dc.subjectSecondary glaucoma
dc.subjectAhmed glaucoma valve
dc.subjectBuphthalmos
dc.subjectCase report
dc.subjectHistopathology
dc.subjectJuvenile xanthogranuloma
dc.subjectNeonatal
dc.subjectPosterior segment involvement
dc.subjectSecondary glaucoma.
dc.titleJuvenile Xanthogranuloma Presented with Buphthalmos and Corneal Clouding in Neonatal Period: A Case Report
dc.typearticle
dspace.entity.typePublication
local.avesis.idf11f8f2b-b9a5-4b8e-a617-2b6a0fb99d6f
local.indexed.atSCOPUS
relation.isAuthorOfPublicationf6d1a5dd-1e4c-45ec-b996-df0e77c46f5a
relation.isAuthorOfPublicationdca036e3-6583-4f3b-a8d5-0abfacb51e74
relation.isAuthorOfPublication22668ace-5f6d-45b8-ab01-b2839b2a5566
relation.isAuthorOfPublication.latestForDiscoveryf6d1a5dd-1e4c-45ec-b996-df0e77c46f5a

Files

Original bundle
Now showing 1 - 1 of 1
Thumbnail Image
Name:
File.pdf
Size:
12.4 MB
Format:
Adobe Portable Document Format
Download

Collections

Research Outputs