A case report of recurrent adult-onset xanthogranuloma: is the radiotherapy a treatment option?

Chicas-Sett, Rodolfo; Llanas, O. Pons; Álvarez, F. Celada; Usmayo, A. Pacheco; Roldán, Susana; Collado, E.; Madrid, E; Aleman, Adolfo; Calatayud, Joaquín; Mico, Apostolov

doi:10.1007/s13691-015-0232-8

Cited by 3 publications

(2 citation statements)

References 12 publications

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“…Both enlargement and heterogenous enhancement of the frontal nerve on MRI were reported in this case [11]. There has been one documented case of imaging-confirmed nerve enlargement in a patient with sarcoidosis with involvement of the right tibial nerve [12]. To the authors' knowledge, this is the first documented case of frontal nerve enlargement secondary to IGOI, which was consistent with the clinical findings of dysaesthesia.…”

Section: Case Reports In Ophthalmologysupporting

confidence: 82%

Idiopathic Granulomatous Orbital Inflammation with Right Frontal Nerve Involvement: A Case Report

Pietris,

Tong,

Selva

2023

Case Rep Ophthalmol

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Introduction: Idiopathic granulomatous orbital inflammation is a rare subtype of orbital inflammatory disease, and frontal nerve involvement has not been previously described. Case Presentation: This is a case of a 75-year-old female who presented with a 4-week history of a right orbital mass, scalp dysaesthesia, and ptosis. Magnetic resonance imaging of the orbits confirmed a right supraorbital mass originating from the lacrimal gland with frontal nerve enlargement. Biopsy showed non-caseating granulomatous inflammation. Conclusion: The patient was treated with an intralesional triamcinolone injection and oral prednisolone and will continue to be monitored for development of systemic sarcoidosis.

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Section: Case Reports In Ophthalmologysupporting

confidence: 82%

Idiopathic Granulomatous Orbital Inflammation with Right Frontal Nerve Involvement: A Case Report

Pietris,

Tong,

Selva

2023

Case Rep Ophthalmol

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“…A case report exists on the use of external beam radiotherapy for controlling local symptoms in the periocular region of patients with AOX. 13 This treatment could be effective in specific circumstances. Additionally, rituximab has demonstrated efficacy for AOX patients with Systemic IgG4-Related Disease.…”

Section: Discussionmentioning

confidence: 99%

Bilateral Adult-Onset Orbital Xanthogranuloma: A Case Report

Chen,

Gao,

Tian

2023

CCID

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Adult-onset xanthogranuloma (AOX) is one of the four rare syndromes collectively referred to as adult xanthogranulomatous disease (AXD). It primarily occurs in the orbit and ocular adnexa and displays distinctive histopathological features, characterized by the infiltration of non-Langerhans-derived foam-like histiocytes and Touton giant cells. The presence of diffuse yellow plaques on the eyelids serves as a highly indicative feature. In this report, we present a compelling case of bilateral periorbital AOX. Initially, the patient received a diagnosis of necrotizing xanthogranuloma (NBX) and underwent treatment with dapsone, which yielded a poor response. Subsequently, through repeated biopsy, immunoprotein electrophoresis, and high-throughput sequencing, the diagnosis was revised to AOX. Subsequently, the patient’s treatment was modified to include oral hormone therapy, and no further progression of the periorbital plaque was observed. Notably, the patient’s sister was diagnosed with xanthelasma palpebrarum (XP), suggesting a potential genetic association between AOX and XP. Unfortunately, the sister declined further histologic examination and genetic sequencing of her skin lesions, impeding the acquisition of additional evidence regarding the genetic link between these two disorders. Despite the divergent pathological features, pathogenesis, and clinical presentation of AOX and xanthelasma palbrarum, clinicians should remain cognizant of the plausible genetic correlation between these two conditions and pursue further investigations when feasible.

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Therapy of 3 patients with Erdhiem-Chester disease with cladribin or cladribin in combination with cyclophosphamide. Case report and review of the therapy

Král¹,

Řehák²,

Krejčí³

et al. 2021

Vnitř Lék

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A case report of recurrent adult-onset xanthogranuloma: is the radiotherapy a treatment option?

Cited by 3 publications

References 12 publications

Idiopathic Granulomatous Orbital Inflammation with Right Frontal Nerve Involvement: A Case Report

Idiopathic Granulomatous Orbital Inflammation with Right Frontal Nerve Involvement: A Case Report

Bilateral Adult-Onset Orbital Xanthogranuloma: A Case Report

Therapy of 3 patients with Erdhiem-Chester disease with cladribin or cladribin in combination with cyclophosphamide. Case report and review of the therapy

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