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Unusual presentation of a vascularized iris lesion in an infant
Sasikala Elizabeth Anilkumar1, Parag K Shah2, V R Saravanan3
1Department of Paediatric Ophthalmology and Strabismus, Aravind Eye Hospital and Postgraduate Institute of Ophthalmology, Coimbatore, Tamil Nadu, India.
Insights
Juvenile xanthogranuloma (JXG) can present as an unusual intraocular mass in infants, causing cataracts. Early diagnosis and surgical intervention are crucial for preserving vision and the affected eye.
Area of Science:
- Ophthalmology
- Pediatric Oncology
- Dermatology
Background:
- Juvenile xanthogranuloma (JXG) is a rare, benign, self-limiting condition primarily affecting infants and children.
- Ocular involvement is the most common extracutaneous manifestation of JXG.
- Cataract formation secondary to intraocular JXG is infrequently reported.
Observation:
- A 10-month-old girl presented with unilateral eye redness and watering.
- Ophthalmic examination revealed an iris vascular lesion and cataract in the left eye.
- Advanced imaging identified an extensive iris and ciliary body mass without calcification.
Findings:
- Histopathological analysis confirmed the mass as juvenile xanthogranuloma (JXG) with associated vascular proliferation.
- The case presented unusually with diffuse intraocular infiltration and cataract, distinct from typical JXG ocular presentations.
- Surgical management included cataract extraction and trans-scleral mass excision.
Implications:
- This case highlights the importance of considering JXG in the differential diagnosis of pediatric intraocular masses.
- Prompt recognition and a systematic approach are vital for successful sight-saving and organ-salvaging treatment in such rare presentations.
- Further research into the pathogenesis and optimal management of intraocular JXG is warranted.
Abstract:
We report an unusual presentation of a 10-month-old girl with left eye (LE) redness and watering. Evaluation showed an iris vascular lesion and lens opacity in her LE. Child underwent USG B-scan and ultrasound biomicroscopy, by which an extensive mass lesion arising from iris and ciliary body with absent calcification was revealed. Following extensive evaluation, child underwent cataract extraction and trans-scleral total excision of the mass lesion. Histopathology proved it as juvenile xanthogranuloma (JXG) with vascular proliferation. JXG is a rare benign self-limiting dermatologic disorder affecting mainly infants and small children. Ocular lesions are the most common extracutaneous manifestation. Cataract in JXG is less frequently reported. This case is reported due to its rarity and as it presented solely as an intraocular lesion with combined diffuse infiltration into ciliary body and cataract which is unusual. Early recognition and systematic approach helped in sight saving and organ salvaging.
