Purpose: This systematic review aims to consolidate evidence on ophthalmological manifestations of angiolymphoid hyperplasia with eosinophilia, focusing on its presentation, diagnosis, and management.
Methods: Preferred reporting items for systematic reviews and meta-analyses-adherent systematic review was conducted across 3 databases from inception until September 2024. Case reports and series detailing angiolymphoid hyperplasia with eosinophilia in ocular structures were included. Data on demographics, clinical presentation, diagnostic methods, and treatment outcomes were extracted and analyzed.
Results: A total of 86 patients from 52 case reports/series were included. The median age was 41 years (IQR: 22-54), with a slight male predominance (51.16%, n = 44/86). The majority of patients (94.18%, n = 81/86) presented with unilateral eye involvement. The orbit was the most frequently involved location (45.35%, n = 39/86), followed by the upper eyelid (27.91%, n = 24/86). Clinical symptoms included proptosis (36.04%, n = 31/86), ptosis (26.74%, n = 23/86), and diplopia (11.67%, n = 10/86). Surgical excision was performed in 54.65% (n = 47/86) of cases, with biopsy done for diagnostic purposes in all patients. Recurrence occurred in 13.95% (n = 12/86), with risk factors including incomplete excision and prior angiolymphoid hyperplasia with eosinophilia in the same or opposite eye. Steroid therapy was used in 18.6% (n = 16/86) but showed limited success. Newer treatment strategies with propranolol showed efficacy in recurrent cases, with no recurrence reported in patients receiving this therapy.
Conclusion: Angiolymphoid hyperplasia with eosinophilia in ocular structures presents diagnostic and therapeutic challenges. Surgical excision remains the mainstay of treatment, though recurrence is common. Therapeutic strategies with propranolol may offer alternative management options in recurrent cases.
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