Juvenile Xanthogranuloma Presenting as Bilateral Non-Infiltrative Extraconal Superior Orbital Tumour in a 27 Year Old Nigerian Woman: Features, Management and Outcome

Introduction Orbital masses in adults are often caused by systemic diseases or are associated with systemic manifestations. Juvenile xanthogranuloma as a cause is rare and unreported in Africa. We present clinical features, management, and outcomes of bilateral orbital adult onset juvenile xanthogra...

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Main Authors: Okosa Michael Chuka, Anyiam Daniel Chukwuemeka Darlinton
Format: Article
Language:English
Published: SAGE Publishing 2012-01-01
Series:Clinical Medicine Insights: Case Reports
Online Access:https://doi.org/10.4137/CCRep.S10138
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spelling doaj-795a1f7649784db184f695276345f9982020-11-25T02:50:13ZengSAGE PublishingClinical Medicine Insights: Case Reports1179-54762012-01-01510.4137/CCRep.S10138Juvenile Xanthogranuloma Presenting as Bilateral Non-Infiltrative Extraconal Superior Orbital Tumour in a 27 Year Old Nigerian Woman: Features, Management and OutcomeOkosa Michael Chuka0Anyiam Daniel Chukwuemeka Darlinton1Department of Ophthalmology, Nnamdi Azikiwe University Teaching Hospital, Guinness Eye Centre, Onitsha, Nigeria, College of Health Sciences, Nnamdi Azikiwe University, Nnewi Campus.Department of Pathology, Nnamdi Azikiwe University Teaching Hospital, Nnewi Nigeria, College of Health Sciences, Nnamdi Azikiwe University, Nnewi Campus.Introduction Orbital masses in adults are often caused by systemic diseases or are associated with systemic manifestations. Juvenile xanthogranuloma as a cause is rare and unreported in Africa. We present clinical features, management, and outcomes of bilateral orbital adult onset juvenile xanthogranuloma. Case Presentation A 27 year old Nigerian woman presented with bilateral upper-lid lumps having lasted 5 months. These increased in size for about 1 month and stopped. Lid swelling was preceded by itchy eyes, redness of conjunctiva, and occasional mild pain. There were no visual or systemic symptoms. The lumps were firm, slightly mobile, not tender, and not attached to skin but rather to deeper structures. There was restriction on up-gaze but no proptosis or diplopia. Hematological, biochemical, and X-ray investigations were normal. Prednisolone tablets 10 mg daily for two weeks were not useful. Tissue biopsy was invaluable in diagnosis of this rare condition and disclosed juvenile xanthogranuloma. Partial surgical excision was done under lidocaine infiltration. No recurrence has occurred in 40 months of follow-up. No systemic disease has manifested. Conclusion Juvenile xanthogranuloma can present as bilateral superior orbital tumor in adults; functional and cosmetic aims were achieved by sub-total excision.https://doi.org/10.4137/CCRep.S10138
collection DOAJ
language English
format Article
sources DOAJ
author Okosa Michael Chuka
Anyiam Daniel Chukwuemeka Darlinton
spellingShingle Okosa Michael Chuka
Anyiam Daniel Chukwuemeka Darlinton
Juvenile Xanthogranuloma Presenting as Bilateral Non-Infiltrative Extraconal Superior Orbital Tumour in a 27 Year Old Nigerian Woman: Features, Management and Outcome
Clinical Medicine Insights: Case Reports
author_facet Okosa Michael Chuka
Anyiam Daniel Chukwuemeka Darlinton
author_sort Okosa Michael Chuka
title Juvenile Xanthogranuloma Presenting as Bilateral Non-Infiltrative Extraconal Superior Orbital Tumour in a 27 Year Old Nigerian Woman: Features, Management and Outcome
title_short Juvenile Xanthogranuloma Presenting as Bilateral Non-Infiltrative Extraconal Superior Orbital Tumour in a 27 Year Old Nigerian Woman: Features, Management and Outcome
title_full Juvenile Xanthogranuloma Presenting as Bilateral Non-Infiltrative Extraconal Superior Orbital Tumour in a 27 Year Old Nigerian Woman: Features, Management and Outcome
title_fullStr Juvenile Xanthogranuloma Presenting as Bilateral Non-Infiltrative Extraconal Superior Orbital Tumour in a 27 Year Old Nigerian Woman: Features, Management and Outcome
title_full_unstemmed Juvenile Xanthogranuloma Presenting as Bilateral Non-Infiltrative Extraconal Superior Orbital Tumour in a 27 Year Old Nigerian Woman: Features, Management and Outcome
title_sort juvenile xanthogranuloma presenting as bilateral non-infiltrative extraconal superior orbital tumour in a 27 year old nigerian woman: features, management and outcome
publisher SAGE Publishing
series Clinical Medicine Insights: Case Reports
issn 1179-5476
publishDate 2012-01-01
description Introduction Orbital masses in adults are often caused by systemic diseases or are associated with systemic manifestations. Juvenile xanthogranuloma as a cause is rare and unreported in Africa. We present clinical features, management, and outcomes of bilateral orbital adult onset juvenile xanthogranuloma. Case Presentation A 27 year old Nigerian woman presented with bilateral upper-lid lumps having lasted 5 months. These increased in size for about 1 month and stopped. Lid swelling was preceded by itchy eyes, redness of conjunctiva, and occasional mild pain. There were no visual or systemic symptoms. The lumps were firm, slightly mobile, not tender, and not attached to skin but rather to deeper structures. There was restriction on up-gaze but no proptosis or diplopia. Hematological, biochemical, and X-ray investigations were normal. Prednisolone tablets 10 mg daily for two weeks were not useful. Tissue biopsy was invaluable in diagnosis of this rare condition and disclosed juvenile xanthogranuloma. Partial surgical excision was done under lidocaine infiltration. No recurrence has occurred in 40 months of follow-up. No systemic disease has manifested. Conclusion Juvenile xanthogranuloma can present as bilateral superior orbital tumor in adults; functional and cosmetic aims were achieved by sub-total excision.
url https://doi.org/10.4137/CCRep.S10138
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AT anyiamdanielchukwuemekadarlinton juvenilexanthogranulomapresentingasbilateralnoninfiltrativeextraconalsuperiororbitaltumourina27yearoldnigerianwomanfeaturesmanagementandoutcome
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