Juvenile Xanthogranuloma–A Rare Cause of Unilateral Orbital Swelling in an Elderly Patient

Patel, Kanak; El-Yassir, M.; Erzurum, Sergul A.

doi:10.3109/01676830.2014.997393

Cited by 5 publications

(3 citation statements)

References 7 publications

(13 reference statements)

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“…Extracutaneous JXG has been reported to affect the orbital cone in elderly patients,[ 14 ] but difficult control of progression is rare in JXG, and so are pericardial and peritoneal involvement when considering extracutaneous forms. [ 11 ]…”

Section: Discussionmentioning

confidence: 99%

Prevalence of temporomandibular disorders among medical students of Wardha district: A cross-sectional study

Civit

Godoy

Conde

et al. 2021

J Datta Meghe Inst Med Sci Univ

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A 70-year-old male presented with orbital masses affecting the muscular cone. His past medical history was notable for diabetes mellitus, ischemic cardiopathy, sleep-apnea syndrome, and multiple serous effusions. The first biopsy specimen of affected orbital tissue revealed fibrohistiocytic infiltration resembling xanthogranuloma or Erdheim–Chester disease (ECD). An ulterior biopsy of affected orbital tissue showed lymphocyte emperipolesis with immunopositivity for CD68 and S100 but negative staining for CD1a marker, strongly suggesting Rosai–Dorfman disease (RDD). Afterward, pericardium and peritoneal effusions resulted in constrictive pericarditis and retroperitoneal fibrosis, respectively. The absence of distinctive clinical features made the diagnosis especially challenging. Attempts to control the disease using corticosteroids, radiation, orbital surgery, and interferon were unsuccessful. Aggressive treatments such as chemotherapy were not considered appropriate due to the general deterioration of our patient. Although the possibility of two concurrent diseases (e.g., systemic ECD and orbital RDD) cannot be discarded, we interpreted the orbital findings as likely due to RDD, and the entire condition of our patient as an extranodal RDD with atypical clinicopathological findings and outcome.

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Section: Discussionmentioning

confidence: 99%

Prevalence of temporomandibular disorders among medical students of Wardha district: A cross-sectional study

Civit

Godoy

Conde

et al. 2021

J Datta Meghe Inst Med Sci Univ

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“…The effectiveness of radiation therapy is unclear due to the limited number of cases; even though radiation therapy often exhibited none or a small palliating stabilizing effect on XD as seen in our case, two reports experienced successful outcome in this context [15,16]. Considering systemic management, ECD treatment options included interferon-α, anakinra and Braf targeting drugs [3,17].…”

Section: Discussionmentioning

confidence: 99%

Cerebral and Cutaneous Involvements of Xanthoma Disseminatum Successfully Treated with an Interleukin-1 Receptor Antagonist: A Case Report and Minireview

Campero¹,

Campero

Guerrero

et al. 2016

Dermatology

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A young male presented with panhypopituitarism (including diabetes insipidus) and temporal lobe epilepsy. A histology specimen of cutaneous papules was diagnostic of non-Langerhans histiocytosis. The diagnosis of xanthoma granulomata was considered based on the clinical and brain MRI findings. Brain lesions significantly worsened over time despite radiotherapy until anakinra induced a complete clinical and radiological remission of all active lesions. Although a single case, the outcome of this patient with xanthoma disseminatum treated with an interleukin-1 receptor antagonist opens and strengthens new and recent physiopathogenic and treatment perspectives for the otherwise difficult-to-treat non-Langerhans cell histiocytosis. Similar results with anakinra have been observed in patients with Erdheim-Chester disease and in multicentric reticulohistiocytosis.

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“…However, lesions in the eyelid, orbit, and cornea have also been reported. 38 Typically occurring before the age of 2, it may manifest with proptosis and involvement of extraocular muscles. Iris lesions are vascular nodules that can bleed easily, causing spontaneous hyphema and glaucoma.…”

Section: Inflammatory Massesmentioning

confidence: 99%

Pediatric ocular tumors: An overview

Raj,

Sahu

2024

IJOOO

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The majority of ocular tumors in children are benign, but specific malignant neoplasms pose a risk to both life and vision. It's indeed crucial for pediatricians to be aware of ocular tumors in children, as early detection and proper management can significantly impact the child's health and vision. As a result, swift recognition and timely referral of patients to an ocular oncologist are imperative for effective clinical management. This article provides an overview of general concepts surrounding ocular tumors in the pediatric age group, offering brief insights into the clinical features and management of significant tumors affecting the eyelids, conjunctiva, intraocular structures, and orbit.

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Juvenile Xanthogranuloma–A Rare Cause of Unilateral Orbital Swelling in an Elderly Patient

Abstract: Juvenile Xanthogranuloma affecting one orbit is very rare with unilateral involvement in an elderly patient. Steroids and radiation therapy were very effective in treatment and provided impressive results.

Cited by 5 publications

References 7 publications

Prevalence of temporomandibular disorders among medical students of Wardha district: A cross-sectional study

Prevalence of temporomandibular disorders among medical students of Wardha district: A cross-sectional study

Cerebral and Cutaneous Involvements of Xanthoma Disseminatum Successfully Treated with an Interleukin-1 Receptor Antagonist: A Case Report and Minireview

Pediatric ocular tumors: An overview

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