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Cataract surgery in juvenile xanthogranuloma: case report and a brief review of literature.

Muralidhar R, Jain A, Vijayalakshmi P, Suparna G, Santhi R, Shetty S - Indian J Ophthalmol (2013)

Bottom Line: There is limited literature on the management of cataracts in juvenile xanthogranuloma (JXG).A 2-month-old girl presented to us with hyphema, secondary glaucoma OU and skin nodules suggestive of JXG.She developed bilateral cataracts during her follow-up and was treated successfully with cataract surgery and aphakic rehabilitation.

View Article: PubMed Central - PubMed

Affiliation: Department of Pediatric Ophthalmology, Aravind Eye Hospital, Madurai, Tamil Nadu, India.

ABSTRACT
There is limited literature on the management of cataracts in juvenile xanthogranuloma (JXG). A 2-month-old girl presented to us with hyphema, secondary glaucoma OU and skin nodules suggestive of JXG. She developed bilateral cataracts during her follow-up and was treated successfully with cataract surgery and aphakic rehabilitation.

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From top left, corneal edema right eye; top right: Hyphema after resolution of corneal edema left eye; bottom left: Development of cataract; bottom right: After cataract extraction
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Figure 1: From top left, corneal edema right eye; top right: Hyphema after resolution of corneal edema left eye; bottom left: Development of cataract; bottom right: After cataract extraction

Mentions: A 2-month-old girl was brought to us with haziness of both eyes for the past 20 days. She was a full-term baby delivered by cesarean section with a birth weight of 2.75 kg. Her antenatal period was uneventful. The parents denied any history of trauma and the child was otherwise healthy. On examination (under general anesthesia), she was noted to have conjunctival hyperemia, increased corneal diameter (13.5 mm OD, 11 mm OS), corneal haze with small blood clots in the anterior chamber of both eyes. No iris nodules were evident [Fig. 1]. Intraocular pressure (IOP) was noted to be 20 mm Hg OD and 8 mm Hg OS by Perkin's tonometer. A B-scan ultrasound of the posterior segment was normal OU. A few yellowish-orange skin nodules were noted on the forehead and neck [Fig. 2], that progressively increased on follow-up. Remainder of her systemic and neurological examination was normal. Her hemogram and coagulation profile were within the normal limits. Excision biopsy of a forehead skin lesion was performed under general anesthesia and histopathological examination of the specimen showed non-langerhans histiocytic proliferation [Fig. 3]. Immunohistochemistry was negative for CD1a and cytokeratin; positive for CD68. With the above clinical picture and laboratory finding, a diagnosis of JXG was made.


Cataract surgery in juvenile xanthogranuloma: case report and a brief review of literature.

Muralidhar R, Jain A, Vijayalakshmi P, Suparna G, Santhi R, Shetty S - Indian J Ophthalmol (2013)

From top left, corneal edema right eye; top right: Hyphema after resolution of corneal edema left eye; bottom left: Development of cataract; bottom right: After cataract extraction
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC3959089&req=5

Figure 1: From top left, corneal edema right eye; top right: Hyphema after resolution of corneal edema left eye; bottom left: Development of cataract; bottom right: After cataract extraction
Mentions: A 2-month-old girl was brought to us with haziness of both eyes for the past 20 days. She was a full-term baby delivered by cesarean section with a birth weight of 2.75 kg. Her antenatal period was uneventful. The parents denied any history of trauma and the child was otherwise healthy. On examination (under general anesthesia), she was noted to have conjunctival hyperemia, increased corneal diameter (13.5 mm OD, 11 mm OS), corneal haze with small blood clots in the anterior chamber of both eyes. No iris nodules were evident [Fig. 1]. Intraocular pressure (IOP) was noted to be 20 mm Hg OD and 8 mm Hg OS by Perkin's tonometer. A B-scan ultrasound of the posterior segment was normal OU. A few yellowish-orange skin nodules were noted on the forehead and neck [Fig. 2], that progressively increased on follow-up. Remainder of her systemic and neurological examination was normal. Her hemogram and coagulation profile were within the normal limits. Excision biopsy of a forehead skin lesion was performed under general anesthesia and histopathological examination of the specimen showed non-langerhans histiocytic proliferation [Fig. 3]. Immunohistochemistry was negative for CD1a and cytokeratin; positive for CD68. With the above clinical picture and laboratory finding, a diagnosis of JXG was made.

Bottom Line: There is limited literature on the management of cataracts in juvenile xanthogranuloma (JXG).A 2-month-old girl presented to us with hyphema, secondary glaucoma OU and skin nodules suggestive of JXG.She developed bilateral cataracts during her follow-up and was treated successfully with cataract surgery and aphakic rehabilitation.

View Article: PubMed Central - PubMed

Affiliation: Department of Pediatric Ophthalmology, Aravind Eye Hospital, Madurai, Tamil Nadu, India.

ABSTRACT
There is limited literature on the management of cataracts in juvenile xanthogranuloma (JXG). A 2-month-old girl presented to us with hyphema, secondary glaucoma OU and skin nodules suggestive of JXG. She developed bilateral cataracts during her follow-up and was treated successfully with cataract surgery and aphakic rehabilitation.

Show MeSH
Related in: MedlinePlus