Rotulo Gioacchino Andrea, Campanello Claudia, Battaglini Marcella, Bassi Marta, Pastorino Carlotta, Angeletti Andrea, Brisca Giacomo, Signa Sara, Caorsi Roberta, Ghiggeri Gian Marco
Department of Neuroscience, Rehabilitation, Ophthalmology, Genetics, Maternal and Child Health (GAR, CC, M Battaglini, M Bassi, SS), University of Genoa, Genoa, Italy.
Clinical and Research Unit of Clinical Immunology and Vaccinology (GAR), IRCCS Bambino Gesù Children Hospital, Rome, Italy.
J Pediatr Pharmacol Ther. 2024 Apr;29(2):195-199. doi: 10.5863/1551-6776-29.2.195. Epub 2024 Apr 8.
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.
别嘌醇诱发的伴有嗜酸性粒细胞增多和全身症状的药物反应综合征(A-DRESS)在成人中是一种已被充分描述的病症,而在儿童中并不常见。我们报告一例16岁患有类固醇依赖型肾病综合征的男性发生A-DRESS的病例。他呈现出危及生命的临床病程,伴有持续发热、皮疹、嗜酸性粒细胞增多、淋巴结病、分布性休克以及检测到疱疹病毒6型。停用别嘌醇并联合静脉注射免疫球蛋白(IVIG)和全身性皮质类固醇治疗后,患者康复且无后遗症。小儿药物反应伴嗜酸性粒细胞增多和全身症状(DRESS)很罕见,且可能以严重形式出现。早期诊断和及时治疗对预后至关重要。本报告提示IVIG在A-DRESS患者治疗中可能起关键作用。