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S. E. Coupland - One of the best experts on this subject based on the ideXlab platform.
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The epidemiology, clinical characteristics, histopathology and management of juvenile- and adult-onset corneoscleral limbus Xanthogranuloma
Graefe's Archive for Clinical and Experimental Ophthalmology, 2016Co-Authors: G. Kontos, S. Borooah, A. Khan, B. W. Fleck, S. E. CouplandAbstract:Purpose Adult-onset Xanthogranuloma (AOX) of the corneoscleral limbus is a rare inflammatory condition of unknown aetiology. Similar to limbal juvenile Xanthogranuloma (JXG), it presents as a growing mass at the corneoscleral junction. Limbal AOX and JXG can lead to sight-threatening complications if not managed in a timely manner. This systematic review summarises the main clinical and histopathological features of limbal AOX/JXG and discusses the management of this uncommon disease. Methods We performed a literature search in the MEDLINE database for all historical entries, using the search terms “limbus”, “limbal” and “Xanthogranuloma”, and retrieved all articles reporting on limbal Xanthogranuloma. After refining the search to articles relevant to limbal AOX, we were able to identify ten adult cases of limbal AOX and compare those with all reported cases of limbal JXG. Results Clinically, AOX usually presents as an isolated smooth, yellowish, dome-shaped nodule at the corneoscleral junction, similar to an ocular presentation of JXG, with which it also shares similar histopathological features. Conclusion Limbal JXG and AOX may represent the same disease entity. Diagnosis relies on the clinical presentation, pathology and immunohistochemical profile. Spontaneous regression is unlikely, and thus prompt surgical intervention should be considered to prevent sight-threatening complications. Xanthogranuloma should be included in the differential diagnosis of corneoscleral limbal masses in patients of all age groups.
Fenwick C Riley - One of the best experts on this subject based on the ideXlab platform.
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juvenile Xanthogranuloma of the corneoscleral limbus case report and review of the literature
Survey of Ophthalmology, 2004Co-Authors: Imtiaz A Chaudhry, Zohair Aljishi, Farrukh A Shamsi, Fenwick C RileyAbstract:Abstract Juvenile Xanthogranuloma is a benign histiocytic cutaneous disorder mainly occurring in infants which may affect the eye. Ocular manifestations usually present in the form of iris lesions with secondary hyphema and glaucoma, but juvenile Xanthogranuloma may also present as a corneoscleral limbal mass. We present the case of an 18-month-old female infant with ocular juvenile Xanthogranuloma occurring as a corneoscleral limbal mass without associated cutaneous or systemic findings. The limbal mass lesion was excised by lamellar dissection and histopathological studies revealed histiocytes admixed with lymphocytes, plasma cells, and eosinophils; foreign body and Touton giant cells present throughout the specimen confirmed the diagnosis of juvenile Xanthogranuloma. At 14-month follow-up, there was no recurrence of the limbal mass. The natural history of this uncommon ocular condition is discussed and the relevant literature reviewed. Juvenile Xanthogranuloma should be considered in the differential diagnosis of any corneoscleral limbal mass lesion, particularly in children.
Zena Lim-i-linn - One of the best experts on this subject based on the ideXlab platform.
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Juvenile Xanthogranuloma of the corneoscleral limbus
Cornea, 2005Co-Authors: Zena Lim-i-linnAbstract:Purpose: To report an 11-year-old Chinese girl with juvenile Xanthogranuloma of the limbus, occurring in isolation without dermatologic involvement, and to discuss the clinical and histologic features, pathogenesis, and treatment of juvenile Xanthogranuloma. Methods: Case report and review of medical literature. Results: A total excision and biopsy of the limbal lesion with lamellar graft was performed. Histologic examination showed a Xanthogranuloma. At 12-month follow-up, there was no recurrence of tumor. Conclusion: The diagnosis of juvenile Xanthogranuloma may be straightforward in cases that have concomitant cutaneous manifestations. However, in isolated ocular forms, histopathological examination is necessary. Unlike the more common iris lesions that are preferably treated nonsurgicilly, limbal lesions require surgical excision.
Geoffrey E Rose - One of the best experts on this subject based on the ideXlab platform.
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radiological and clinicopathological features of orbital Xanthogranuloma
British Journal of Ophthalmology, 2000Co-Authors: K A Miszkiel, S A A Sohaib, Geoffrey E Rose, Ian A Cree, I F MoseleyAbstract:Background—Orbital Xanthogranuloma, a diagnosis confirmed histologically, occurs rarely in adults and children. With its characteristic macroscopic appearance the adult form may be associated with a spectrum of biochemical and haematological abnormalities including lymphoproliferative malignancies. Method—The clinicopathological features and imaging appearances on computed tomography and magnetic resonance imaging of this condition are described in eight adults and a child. Results—Radiological evidence of proptosis was present in seven patients. In all nine patients an abnormal infiltrative soft tissue mass was seen, with increased fat in six cases. All patients had associated enlargement of extraocular muscles suggestive of infiltration and five had lacrimal gland involvement. Encasement of the optic nerve, bone destruction, and intracranial extension was present only in the child with juvenile Xanthogranuloma. Haematological and/or biochemical abnormalities were detected in seven patients and seven patients had other systemic diseases which were considered to have an immune basis. One patient subsequently developed non-Hodgkin’s lymphoma. Conclusion—The investigation and management of orbital Xanthogranulomas requires a multidisciplinary approach even though the diagnosis may be suspected clinically. Imaging delineates the extent of disease and involvement of local structures and may influence the diVerential diagnosis. The juvenile form may be more locally aggressive, causing bone destruction with consequent intracranial extension. (Br J Ophthalmol 2000;84:251‐258)
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Orbital Xanthogranuloma in adults.
The British journal of ophthalmology, 1991Co-Authors: Geoffrey E Rose, Bhupendra C. K. Patel, A. Garner, John E. WrightAbstract:The onset of periorbital Xanthogranuloma in adults is rare and may be accompanied by haematological abnormalities and malignancy. The appearance of the eyelid lesions is virtually diagnostic, producing readily recognisable diffuse, yellow plaques, and affected patients should be investigated and reviewed regularly for systemic disease. Three cases are described, in which periorbital cutaneous plaques were associated with abnormal tissues in the superior part of the orbit; these abnormal tissues caused displacement or restricted movement of the globe or upper eyelid. The possibility that two cases represent a necrobiotic type of Xanthogranuloma is presented. Nine years after the onset of Xanthogranuloma one patient developed non-Hodgkin9s lymphoma. A multiple-drug regimen of systemic chemotherapy, given for lymphoma, caused a marked clinical reduction in the periorbital Xanthogranuloma.
Arthur E. Marlin - One of the best experts on this subject based on the ideXlab platform.
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Giant bilateral Xanthogranulomas in a child: case report.
Neurosurgery, 1992Co-Authors: Sarah J. Gaskill, Victor Saldivar, Joel Rutman, Arthur E. MarlinAbstract:Symptomatic Xanthogranulomas are rare lesions that most commonly occur in adults. A case of giant bilateral Xanthogranulomas in a 6-year-old boy, who remains without tumor recurrence 9 years after resection, is presented. The operative management of these unusually large lesions is discussed. The pathogenesis of Xanthogranulomas is reviewed as it relates to the presentation of these lesions in the pediatric population.
