[abstract] MANAGING OPHTHALMIC NECROBIOTIC XANTHOGRANULOMA: A MULTIMODALITY APPROACH COMBINING ANTI-HISTOCYTIC CHEMOTHERAPY (CLADRIBINE), SURGICAL DEBRIDEMENT, ANTIMICROBIAL AND HYPERBARIC OXYGEN THERAPY, AND LIMITED SURGICAL GRAFTING

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[abstract] MANAGING OPHTHALMIC NECROBIOTIC XANTHOGRANULOMA: A MULTIMODALITY APPROACH COMBINING ANTI-HISTOCYTIC CHEMOTHERAPY (CLADRIBINE), SURGICAL DEBRIDEMENT, ANTIMICROBIAL AND HYPERBARIC OXYGEN THERAPY, AND LIMITED SURGICAL GRAFTING

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Title: [abstract] MANAGING OPHTHALMIC NECROBIOTIC XANTHOGRANULOMA: A MULTIMODALITY APPROACH COMBINING ANTI-HISTOCYTIC CHEMOTHERAPY (CLADRIBINE), SURGICAL DEBRIDEMENT, ANTIMICROBIAL AND HYPERBARIC OXYGEN THERAPY, AND LIMITED SURGICAL GRAFTING
Author: Claus, P; Patel, S; Gibson, L; Dronca, R; Colgan, J
Abstract: BACKGROUND: Necrobiotic Xanthogranuloma (NXG) is a rare progressive and destructive histocytic disease often associated with paraproteinemia of hematologic and lymphoproliferative disorders. It typically presents with slowly progressive and destructive cutaneous lesions with a high propensity for periorbital tissues. Treatments that have been reported include surgical excision, local corticosteroid injection, plasmapheresis, chemotherapy or radiotherapy in various combinations all with variable responses. Surgical excision and injections are generally avoided due to the high rate of non-healing extension and local recurrence. Some patients experience rapid ulceration with severe local destruction that necessitates aggressive surgical debridement and supportive healing measures. We report the successful treatment of a patient with threatened loss of total vision and cranial bone exposure. MATERIALS AND METHODS: Our patient presented with multiple myeloma and advanced periorbital tissue involvement of NXG with extension into the sclera, complicated by severe exposure keratopathy, infection of the cornea, anterior chamber and periorbital tissues of both eyes with mixed bacteria that included pseudomonas aeruginosa and fungal elements. Initial debridement required enucleation of one eye due to endophthalmitis after corneal perforation. The fellow eye and periorbital tissues responded to aggressive multi-modality therapy which included targeted anti-histocytic chemotherapy (Cladribine), 32 HBOT sessions (100% FiO2, 2.0 ATA x 90 minutes), systemic periocular and intraocular antibiotic and antifungal therapy, and conjunctival flap procedure. RESULTS: This synergistic approach resulted in control of the previously advancing destructive process and the rapid production of a healthy granular wound bed that healed by secondary intention. Hand motion vision was preserved in the surviving eye with potential for improved vision with a keratoprosthesis in the future. CONCLUSIONS: After failing conservative management, this complicated case of NXG was successfully treated with multiple synergistic therapies and aggressive surgical debridement. Excellent patient outcome was observed with partial vision preservation, rapid wound healing, decreased discomfort and favorable cosmetic appearance.
Description: Abstract of the Undersea and Hyperbaric Medical Society, Inc. Annual Scientific Meeting, Las Vegas, Nevada, USA. (http://www.uhms.org)
URI: http://archive.rubicon-foundation.org/9153
Date: 2009

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  • UHMS Meeting Abstracts
    This is a collection of the published abstracts from the Undersea and Hyperbaric Medical Society (UHMS) annual meetings.

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