Corneal Juvenile Xanthogranuloma in a 4-Month-Old Child

Ashmore, Emily D.; Wilson, Matthew W.; Morris, William R.; Hill, D. Ashley; Rodríguez‐Galindo, Carlos; Haik, Barrett G.

doi:10.1001/archopht.121.1.117

Cited by 20 publications

(7 citation statements)

References 3 publications

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“…In non-sight threatening condition, it can be treated with high-dose topical steroid, periocular steroid and systemic corticosteroid [2]. There has been reports of successfully treating limbal JXG using topical corticosteroids in a four-month-old male patient, and also using intralesional corticosteroid for congenital eyelid JXG in an 18-day-old infant [9, 10].…”

Section: Discussionmentioning

confidence: 99%

Eyelid Juvenile Xanthogranuloma: A Case Report and Literature Review

Hassan¹,

Talib²,

Adzman³

et al. 2019

Cureus

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Juvenile xanthogranuloma (JXG) is an uncommon condition affecting the eye. We herein report a rare case of eyelid swelling in paediatric age group. A three-year-old Malay boy presented with chronic painless left upper eyelid mass which did not resolve with topical steroid. Clinically, the mass was a non-tender and firm nodular swelling which located at the lateral 1/3 of the left upper lid. Total excisional biopsy of the swelling was done and histopathological findings were consistent with JXG. The systemic associations and the treatment options for ocular JXG are discussed.

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Section: Discussionmentioning

confidence: 99%

Eyelid Juvenile Xanthogranuloma: A Case Report and Literature Review

Hassan¹,

Talib²,

Adzman³

et al. 2019

Cureus

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“…Of the reported cases, 25 (92.6%) were treated surgically with excisional biopsy or total excision. Ashmore et al 3 reported resolution of corneal xanthogranuloma with topical steroid regimen of loteprednol etabonate every 2 h and fluorometholone ointment nightly in a 4-month-old infant. Longmuir et al 9 reported a 14-month-old case treated with methotrexate, whose attempt to taper the medication dose resulted in a relapse.…”

Section: Discussionmentioning

confidence: 99%

“…2 Attempts to treat corneoscleral xanthogranulomas included topical steroids, angiogenesis inhibitors, immunosuppressive therapy, radiation, and surgical excision with or without lamellar keratoplasty. [2][3][4] Although several reports advocated the surgical approach, 2,5 no clear evidence exists to support any particular treatment modality. We report herein a XD patient with limbal xanthogranuloma showing complete remission after treatment with methotrexate and azathioprine.…”

Section: Introductionmentioning

confidence: 99%

Corneoscleral xanthogranuloma treated with chemotherapy: The room for nonsurgical management

Mustafa

Daoud

2019

European Journal of Ophthalmology

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Purpose: To describe a clinical case of corneoscleral xanthogranuloma, a rare manifestation of juvenile xanthogranuloma, and xanthoma disseminatum, which responded well to chemotherapy. Methods: Interventional case report and literature search. Results: A 9-year-old female patient with a disseminated disease showed complete regression of her corneoscleral xanthogranuloma with methotrexate and azathioprine therapy. Conclusion: Since they are potentially blinding, corneoscleral xanthogranulomas are commonly surgically excised. While surgical resection has been widely advocated in the literature, immunosuppressive therapy alone may be a pertinent management line of corneoscleral xanthogranuloma, especially with systemic involvement.

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“…7,16 Irradiation without short-term recurrence was used in 2 cases, one combined with resection. 17 Surgical removal of the lesion was the only curative option in 20 of the 22 patients 15,7 requiring lamellar grafting in a number of cases. Because incomplete excision often ended in a recurrence, 2,6,18 total resection was considered the only viable treatment option to date, 1 and routine combination with a graft has been suggested.…”

Section: Discussionmentioning

confidence: 99%