A Rare Pediatric Case of Allopurinol-Induced Drug Reaction With Eosinophilia and Systemic Symptoms (DRESS) Successfully Treated With Intravenous Immunoglobulins

Author:

Rotulo Gioacchino Andrea12,Campanello Claudia1,Battaglini Marcella1,Bassi Marta1,Pastorino Carlotta3,Angeletti Andrea4,Brisca Giacomo5,Signa Sara16,Caorsi Roberta6,Ghiggeri Gian Marco4

Affiliation:

1. Department of Neuroscience, Rehabilitation, Ophthalmology, Genetics, Maternal and Child Health (GAR, CC, M Battaglini, M Bassi, SS), University of Genoa, Genoa, Italy;

2. Clinical and Research Unit of Clinical Immunology and Vaccinology (GAR), IRCCS Bambino Gesù Children Hospital, Rome, Italy;

3. Dermatology Unit (CP), Giannina Gaslini Children's Hospital, Genoa, Italy;

4. Division of Nephrology, Dialysis, and Transplantation (AA, GMG), Giannina Gaslini Children’s Hospital, Genoa, Italy;

5. Subintensive Care Unit (GB), IRCCS Istituto Giannina Gaslini, Genoa, Italy.

6. Autoinflammatory Diseases and Immunodeficiencies Center (SS, RC), IRCCS Istituto Giannina Gaslini, Genoa, Italy;

Abstract

Allopurinol-induced drug reaction syndrome with eosinophilia and systemic symptoms (A-DRESS) is a well-described condition in adults, whereas it is uncommon among children. We describe a case of A-DRESS in a 16-year-old male with steroid-dependent nephrotic syndrome. He presented a life-threatening clinical course with persisting fever, skin rash, eosinophilia, lymphadenopathy, distributive shock, and herpesvirus 6 detection. The withdrawal of allopurinol and a combination of intravenous immunoglobulins (IVIGs) and systemic corticosteroids led to the patient’s recovery without sequelae. Drug reaction with eosinophilia and systemic symptoms (DRESS) in pediatrics is rare and can present in a severe form. Early diagnosis and timely treatment are critical for prognostic purposes. This report suggests the potentially crucial role of IVIG in the treatment of patients with A-DRESS.

Publisher

Pediatric Pharmacy Advocacy Group

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