Recurrent episodes of isolated periorbital angioedema associated with separate courses of ibuprofen: A diagnostic dilemma

Abstract

Drug hypersensitivity reactions (DHRs) to nonsteroidal anti-inflammatory drugs (NSAIDs) presenting as isolated periorbital angioedema (PE), especially that caused by Ibuprofen have not been extensively reported in the persons of Indian ethnicity. A 58-year-old Indian male was prescribed tablet Ibuprofen for lower backache. Shortly after intake of the tablet, the patient developed severe itching, redness, and watering in both eyes followed by bilateral progressive PE, without involvement of skin elsewhere, or the respiratory system, and in the absence of features suggestive of severe anaphylaxis. The episode was successfully managed with parenteral hydrocortisone and pheniramine. The patient disclosed two similar episodes previously on intake of ibuprofen which were also treated conservatively. Based on a score of 10 on the Naranjo Adverse Drug Reaction Probability Scale and oral Ibuprofen challenge test (OCT) that resulted in recurrence of a similar reaction, a definitive diagnosis of drug reaction to Ibuprofen was concluded. Raised serum immunoglobulin-E levels and eosinophilia on peripheral blood smear were other significant findings. Skin-prick test (SPT) with commonly used NSAIDs was inconclusive. A negative Aspirin/5-Acetyl Salicylic-Acid (ASA) OCT strongly supported the possibility of single-NSAID induced urticaria/angioedema or anaphylaxis reaction to Ibuprofen. This case report highlights the challenges in identifying and characterizing an NSAID-DHRs, especially in the background of varied clinical presentations and discordant test results, as well as due to the lack of well-established standards for SPT, absence of validated in vitro tests, nonavailability of test reagents, and risks associated with OCT.