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In vivo confocal microscopy in goldenhar syndrome: a case report

BACKGROUND: Goldenhar Syndrome is characterized by malformations of multiple anatomical districts. Between these, bulbar dermoids are common and represent a significant clinical problem as they can affect both ocular function and aesthetic comfort. Histologic characterization of dermoids has been ex...

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Autores principales: Triolo, Giacinto, Ferrari, Giulio, Doglioni, Claudio, Rama, Paolo
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2013
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3853195/
https://www.ncbi.nlm.nih.gov/pubmed/24131730
http://dx.doi.org/10.1186/1471-2415-13-55
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author Triolo, Giacinto
Ferrari, Giulio
Doglioni, Claudio
Rama, Paolo
author_facet Triolo, Giacinto
Ferrari, Giulio
Doglioni, Claudio
Rama, Paolo
author_sort Triolo, Giacinto
collection PubMed
description BACKGROUND: Goldenhar Syndrome is characterized by malformations of multiple anatomical districts. Between these, bulbar dermoids are common and represent a significant clinical problem as they can affect both ocular function and aesthetic comfort. Histologic characterization of dermoids has been extensively performed; however, no reports exist describing in vivo confocal microscopy (IVCM) of these lesions. We aimed to (i) describe the in vivo confocal morphology of limbal dermoids in Goldenhar syndrome and (ii) compare these findings with standard light microscopy. CASE PRESENTATION: A 15-year-old Caucasian female affected by Goldenhar Syndrome showed a left, infero-temporal, limbal neoformation, with extension to the left orbital region. Prior to surgical removal, IVCM was performed with the Heidelberg Retina Tomograph II, Cornea Module, using the “section” modality. The IVCM sections showed structures resembling corneal epithelium and vascular structures. Surgical removal of the lesion was decided as it caused poor eyelid closure. After surgical removal, sectioning and standard optical microscopy were performed. The comparison between IVCM imaging and standard microscopy sections were highly correlated in the detection of the pilar and vascular structures. CONCLUSIONS: This study showed that IVCM may be a useful technique to study limbal dermoids, given its ability to detect typical microscopic features and its comparability to optical microscopy, which is the current standard.
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spelling pubmed-38531952013-12-07 In vivo confocal microscopy in goldenhar syndrome: a case report Triolo, Giacinto Ferrari, Giulio Doglioni, Claudio Rama, Paolo BMC Ophthalmol Case Report BACKGROUND: Goldenhar Syndrome is characterized by malformations of multiple anatomical districts. Between these, bulbar dermoids are common and represent a significant clinical problem as they can affect both ocular function and aesthetic comfort. Histologic characterization of dermoids has been extensively performed; however, no reports exist describing in vivo confocal microscopy (IVCM) of these lesions. We aimed to (i) describe the in vivo confocal morphology of limbal dermoids in Goldenhar syndrome and (ii) compare these findings with standard light microscopy. CASE PRESENTATION: A 15-year-old Caucasian female affected by Goldenhar Syndrome showed a left, infero-temporal, limbal neoformation, with extension to the left orbital region. Prior to surgical removal, IVCM was performed with the Heidelberg Retina Tomograph II, Cornea Module, using the “section” modality. The IVCM sections showed structures resembling corneal epithelium and vascular structures. Surgical removal of the lesion was decided as it caused poor eyelid closure. After surgical removal, sectioning and standard optical microscopy were performed. The comparison between IVCM imaging and standard microscopy sections were highly correlated in the detection of the pilar and vascular structures. CONCLUSIONS: This study showed that IVCM may be a useful technique to study limbal dermoids, given its ability to detect typical microscopic features and its comparability to optical microscopy, which is the current standard. BioMed Central 2013-10-16 /pmc/articles/PMC3853195/ /pubmed/24131730 http://dx.doi.org/10.1186/1471-2415-13-55 Text en Copyright © 2013 Triolo et al.; licensee BioMed Central Ltd. http://creativecommons.org/licenses/by/2.0 This is an open access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Case Report
Triolo, Giacinto
Ferrari, Giulio
Doglioni, Claudio
Rama, Paolo
In vivo confocal microscopy in goldenhar syndrome: a case report
title In vivo confocal microscopy in goldenhar syndrome: a case report
title_full In vivo confocal microscopy in goldenhar syndrome: a case report
title_fullStr In vivo confocal microscopy in goldenhar syndrome: a case report
title_full_unstemmed In vivo confocal microscopy in goldenhar syndrome: a case report
title_short In vivo confocal microscopy in goldenhar syndrome: a case report
title_sort in vivo confocal microscopy in goldenhar syndrome: a case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3853195/
https://www.ncbi.nlm.nih.gov/pubmed/24131730
http://dx.doi.org/10.1186/1471-2415-13-55
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