Introduction: Idiopathic hypereosinophilic syndrome is defined by the combination of persistent eosinophilia and evidence of tissue damage. It is a diagnosis of exclusion.
Case: We report the case of a 53-year-old man with idiopathic hypereosinophilic syndrome associated with an orbital inflammatory pseudotumor. Therapy with corticosteroids rapidly decreased the number of circulating eosinophiles; the orbital inflammatory pseudotumor regressed at the same time.
Discussion: This is the first report of idiopathic hypereosinophilic syndrome associated with an orbital inflammatory pseudotumor.
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