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Treatment of refractory uveitic macular edema with dexamethasone intravitreal implants in a pediatric patient with bilateral granulomatous idiopathic panuveitis: a case report.

Bourgault S, Aroichane M, Wittenberg LA, Lavallée A, Ma PE - J Ophthalmic Inflamm Infect (2013)

Bottom Line: Three months later, his vision had improved from 20/200 in both eyes to 20/30 in the right eye and 20/40 in the left eye.Optical coherence tomography showed complete resolution of the cystoid macular edema and subretinal fluid in both eyes.This is a rare report of the use of bilateral dexamethasone intravitreal implant in a pediatric patient.

View Article: PubMed Central - HTML - PubMed

Affiliation: Department of Ophthalmology and Visual Sciences, University of British Columbia, Vancouver, British Columbia V5Z 3N9, Canada. serge.bourgault.1@ulaval.ca.

ABSTRACT

Background: Macular edema is a common complication of uveitis and represents a therapeutic challenge, especially in children. Recently, intravitreal dexamethasone implants have been shown to decrease intraocular inflammation and to control uveitic macular edema in patients with non-infectious intermediate or posterior uveitis.

Findings: An 11-year-old boy with bilateral granulomatous idiopathic panuveitis and orbital inflammation experienced macular edema refractory to topical steroids and subcutaneous methotrexate. He was treated with off-label bilateral injections of dexamethasone intravitreal implant. Three months later, his vision had improved from 20/200 in both eyes to 20/30 in the right eye and 20/40 in the left eye. Optical coherence tomography showed complete resolution of the cystoid macular edema and subretinal fluid in both eyes.

Conclusions: This is a rare report of the use of bilateral dexamethasone intravitreal implant in a pediatric patient. The implants achieved complete resolution of the uveitic macular edema with no adverse events 3 months post-implantation.

No MeSH data available.


Related in: MedlinePlus

Color fundus photographs and fluorescein angiogram at presentation. (top right and top left) Both eyes showed optic nerve edema, dilated veins, and macular edema. (bottom right and bottom left) Late-phase fluorescein angiogram showed leakage of the optic nerve head that was more evident in the left eye, as well as patchy hyperfluorescence of the macula and petaloid leakage at the fovea in both eyes.
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Figure 1: Color fundus photographs and fluorescein angiogram at presentation. (top right and top left) Both eyes showed optic nerve edema, dilated veins, and macular edema. (bottom right and bottom left) Late-phase fluorescein angiogram showed leakage of the optic nerve head that was more evident in the left eye, as well as patchy hyperfluorescence of the macula and petaloid leakage at the fovea in both eyes.

Mentions: An 11-year-old boy of Nigerian descent was followed for bilateral granulomatous idiopathic panuveitis and orbital inflammation for 14 months (Figure 1). His past medical history was significant for hemoglobin SC disease and G6PD deficiency. At the onset of the disease, a rheumatologic workup with anti-nuclear antibody, rheumatoid factor, anti-neutrophil cytoplasmic antibodies, and HLA-B27 testing was negative. Serology for Lyme disease, toxoplasmosis, toxacariasis, cat scratch disease, human immunodeficiency virus, leptospirosis, coccidioidomycosis, and histoplasmosis was negative. Syphilis was also ruled out by a negative RPR and TP-PA. His serum calcium level was slightly elevated, but the angiotensin converting enzyme level and chest X-ray were normal. A biopsy of an enlarged right lacrimal gland biopsy was performed. Pathologic analysis was inconclusive and showed a non-specific non-granulomatous lymphocytic inflammation. A Mantoux test result showed 4 mm of induration and was considered positive as the patient came from an endemic area and was at that time treated with high-dose intravenous corticosteroids for his ocular inflammation. The patient was treated with anti-tuberculous triple-drug therapy for 2 months and then with rifampin for 4 months. Triple-drug therapy was discontinued as the patient was diagnosed with latent tuberculosis by infectious disease specialists. Indeed, he showed a positive response to corticosteroids, and aqueous and vitreous fluid cultures were negative for mycobacteria.


Treatment of refractory uveitic macular edema with dexamethasone intravitreal implants in a pediatric patient with bilateral granulomatous idiopathic panuveitis: a case report.

Bourgault S, Aroichane M, Wittenberg LA, Lavallée A, Ma PE - J Ophthalmic Inflamm Infect (2013)

Color fundus photographs and fluorescein angiogram at presentation. (top right and top left) Both eyes showed optic nerve edema, dilated veins, and macular edema. (bottom right and bottom left) Late-phase fluorescein angiogram showed leakage of the optic nerve head that was more evident in the left eye, as well as patchy hyperfluorescence of the macula and petaloid leakage at the fovea in both eyes.
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4016568&req=5

Figure 1: Color fundus photographs and fluorescein angiogram at presentation. (top right and top left) Both eyes showed optic nerve edema, dilated veins, and macular edema. (bottom right and bottom left) Late-phase fluorescein angiogram showed leakage of the optic nerve head that was more evident in the left eye, as well as patchy hyperfluorescence of the macula and petaloid leakage at the fovea in both eyes.
Mentions: An 11-year-old boy of Nigerian descent was followed for bilateral granulomatous idiopathic panuveitis and orbital inflammation for 14 months (Figure 1). His past medical history was significant for hemoglobin SC disease and G6PD deficiency. At the onset of the disease, a rheumatologic workup with anti-nuclear antibody, rheumatoid factor, anti-neutrophil cytoplasmic antibodies, and HLA-B27 testing was negative. Serology for Lyme disease, toxoplasmosis, toxacariasis, cat scratch disease, human immunodeficiency virus, leptospirosis, coccidioidomycosis, and histoplasmosis was negative. Syphilis was also ruled out by a negative RPR and TP-PA. His serum calcium level was slightly elevated, but the angiotensin converting enzyme level and chest X-ray were normal. A biopsy of an enlarged right lacrimal gland biopsy was performed. Pathologic analysis was inconclusive and showed a non-specific non-granulomatous lymphocytic inflammation. A Mantoux test result showed 4 mm of induration and was considered positive as the patient came from an endemic area and was at that time treated with high-dose intravenous corticosteroids for his ocular inflammation. The patient was treated with anti-tuberculous triple-drug therapy for 2 months and then with rifampin for 4 months. Triple-drug therapy was discontinued as the patient was diagnosed with latent tuberculosis by infectious disease specialists. Indeed, he showed a positive response to corticosteroids, and aqueous and vitreous fluid cultures were negative for mycobacteria.

Bottom Line: Three months later, his vision had improved from 20/200 in both eyes to 20/30 in the right eye and 20/40 in the left eye.Optical coherence tomography showed complete resolution of the cystoid macular edema and subretinal fluid in both eyes.This is a rare report of the use of bilateral dexamethasone intravitreal implant in a pediatric patient.

View Article: PubMed Central - HTML - PubMed

Affiliation: Department of Ophthalmology and Visual Sciences, University of British Columbia, Vancouver, British Columbia V5Z 3N9, Canada. serge.bourgault.1@ulaval.ca.

ABSTRACT

Background: Macular edema is a common complication of uveitis and represents a therapeutic challenge, especially in children. Recently, intravitreal dexamethasone implants have been shown to decrease intraocular inflammation and to control uveitic macular edema in patients with non-infectious intermediate or posterior uveitis.

Findings: An 11-year-old boy with bilateral granulomatous idiopathic panuveitis and orbital inflammation experienced macular edema refractory to topical steroids and subcutaneous methotrexate. He was treated with off-label bilateral injections of dexamethasone intravitreal implant. Three months later, his vision had improved from 20/200 in both eyes to 20/30 in the right eye and 20/40 in the left eye. Optical coherence tomography showed complete resolution of the cystoid macular edema and subretinal fluid in both eyes.

Conclusions: This is a rare report of the use of bilateral dexamethasone intravitreal implant in a pediatric patient. The implants achieved complete resolution of the uveitic macular edema with no adverse events 3 months post-implantation.

No MeSH data available.


Related in: MedlinePlus