In Vivo Confocal Microscopy and Anterior Segment Optic Coherence Tomography Findings in Ocular Ochronosis

Purpose. To report clinical and in vivo confocal microscopy (IVCM) findings of two patients with ocular ochronosis secondary due to alkaptonuria. Materials and Methods. Complete ophthalmologic examinations, including IVCM (HRT II/Rostock Cornea Module, Heidelberg, Germany), anterior segment optical...

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Main Authors: Elif Demirkilinc Biler, Suzan Guven Yilmaz, Melis Palamar, Pedram Hamrah, Afsun Sahin
Format: Article
Language:English
Published: Wiley 2015-01-01
Series:Case Reports in Ophthalmological Medicine
Online Access:http://dx.doi.org/10.1155/2015/592847
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author Elif Demirkilinc Biler
Suzan Guven Yilmaz
Melis Palamar
Pedram Hamrah
Afsun Sahin
author_facet Elif Demirkilinc Biler
Suzan Guven Yilmaz
Melis Palamar
Pedram Hamrah
Afsun Sahin
author_sort Elif Demirkilinc Biler
collection DOAJ
description Purpose. To report clinical and in vivo confocal microscopy (IVCM) findings of two patients with ocular ochronosis secondary due to alkaptonuria. Materials and Methods. Complete ophthalmologic examinations, including IVCM (HRT II/Rostock Cornea Module, Heidelberg, Germany), anterior segment optical coherence tomography (AS-OCT) (Topcon 3D spectral-domain OCT 2000, Topcon Medical Systems, Paramus, NJ, USA), corneal topography (Pentacam, OCULUS Optikgeräte GmbH, Wetzlar, Germany), and anterior segment photography, were performed. Results. Biomicroscopic examination showed bilateral darkly pigmented lesions of the nasal and temporal conjunctiva and episclera in both patients. In vivo confocal microscopy of the lesions revealed prominent degenerative changes, including vacuoles and fragmentation of collagen fibers in the affected conjunctival lamina propria and episclera. Hyperreflective pigment granules in different shapes were demonstrated in the substantia propria beneath the basement membrane. AS-OCT of Case 1 demonstrated hyporeflective areas. Fundus examination was within normal limits in both patients, except tilted optic discs with peripapillary atrophy in one of the patients. Corneal topography, thickness, and macular OCT were normal bilaterally in both cases. Conclusion. The degenerative and anatomic changes due to ochronotic pigment deposition in alkaptonuria can be demonstrated in detail with IVCM and AS-OCT. Confocal microscopic analysis in ocular ochronosis may serve as a useful adjunct in diagnosis and monitoring of the disease progression.
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spelling doaj-art-99fd92cb017b4a049fabcf3214e046072025-08-20T02:20:16ZengWileyCase Reports in Ophthalmological Medicine2090-67222090-67302015-01-01201510.1155/2015/592847592847In Vivo Confocal Microscopy and Anterior Segment Optic Coherence Tomography Findings in Ocular OchronosisElif Demirkilinc Biler0Suzan Guven Yilmaz1Melis Palamar2Pedram Hamrah3Afsun Sahin4Ege University Faculty of Medicine, Department of Ophthalmology, 35040 Izmir, TurkeyEge University Faculty of Medicine, Department of Ophthalmology, 35040 Izmir, TurkeyEge University Faculty of Medicine, Department of Ophthalmology, 35040 Izmir, TurkeyDepartment of Ophthalmology, Tufts Medical Center, Boston, MA, USAOsmangazi University Faculty of Medicine, Department of Ophthalmology, Eskişehir, TurkeyPurpose. To report clinical and in vivo confocal microscopy (IVCM) findings of two patients with ocular ochronosis secondary due to alkaptonuria. Materials and Methods. Complete ophthalmologic examinations, including IVCM (HRT II/Rostock Cornea Module, Heidelberg, Germany), anterior segment optical coherence tomography (AS-OCT) (Topcon 3D spectral-domain OCT 2000, Topcon Medical Systems, Paramus, NJ, USA), corneal topography (Pentacam, OCULUS Optikgeräte GmbH, Wetzlar, Germany), and anterior segment photography, were performed. Results. Biomicroscopic examination showed bilateral darkly pigmented lesions of the nasal and temporal conjunctiva and episclera in both patients. In vivo confocal microscopy of the lesions revealed prominent degenerative changes, including vacuoles and fragmentation of collagen fibers in the affected conjunctival lamina propria and episclera. Hyperreflective pigment granules in different shapes were demonstrated in the substantia propria beneath the basement membrane. AS-OCT of Case 1 demonstrated hyporeflective areas. Fundus examination was within normal limits in both patients, except tilted optic discs with peripapillary atrophy in one of the patients. Corneal topography, thickness, and macular OCT were normal bilaterally in both cases. Conclusion. The degenerative and anatomic changes due to ochronotic pigment deposition in alkaptonuria can be demonstrated in detail with IVCM and AS-OCT. Confocal microscopic analysis in ocular ochronosis may serve as a useful adjunct in diagnosis and monitoring of the disease progression.http://dx.doi.org/10.1155/2015/592847
spellingShingle Elif Demirkilinc Biler
Suzan Guven Yilmaz
Melis Palamar
Pedram Hamrah
Afsun Sahin
In Vivo Confocal Microscopy and Anterior Segment Optic Coherence Tomography Findings in Ocular Ochronosis
Case Reports in Ophthalmological Medicine
title In Vivo Confocal Microscopy and Anterior Segment Optic Coherence Tomography Findings in Ocular Ochronosis
title_full In Vivo Confocal Microscopy and Anterior Segment Optic Coherence Tomography Findings in Ocular Ochronosis
title_fullStr In Vivo Confocal Microscopy and Anterior Segment Optic Coherence Tomography Findings in Ocular Ochronosis
title_full_unstemmed In Vivo Confocal Microscopy and Anterior Segment Optic Coherence Tomography Findings in Ocular Ochronosis
title_short In Vivo Confocal Microscopy and Anterior Segment Optic Coherence Tomography Findings in Ocular Ochronosis
title_sort in vivo confocal microscopy and anterior segment optic coherence tomography findings in ocular ochronosis
url http://dx.doi.org/10.1155/2015/592847
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