Case report: Sulfasalazine-induced hypersensitivity

Abstract

Drug-induced hypersensitivity syndrome (DiHS)/drug reaction with eosinophilia and systemic symptoms (DRESS) is a systemic inflammatory condition that is characterized by multisystemic involvement (liver, blood, and skin), heterogeneous manifestations (fever, rash, lymphadenopathy, and eosinophilia), and an unpredictable course; cases of DiHS/DRESS caused by sulfasalazine are rare in children compared to adults. We report a case of a 12-year-old girl with juvenile idiopathic arthritis (JIA) and sulfasalazine-related hypersensitivity who developed fever, rash, blood abnormalities, and hepatitis complicated with hypocoagulation. The treatment with intravenous and then oral glucocorticosteroids was effective. We also reviewed 15 cases (67% male patients) of childhood-onset sulfasalazine-related DiHS/DRESS from the MEDLINE/PubMed and Scopus online databases. All reviewed cases had a fever, lymphadenopathy, and liver involvement. Eosinophilia was reported in 60% of patients. All patients were treated with systemic corticosteroids, and one patient required emergency liver transplantation. Two patients (13%) died. A total of 40.0% of patients satisfied RegiSCAR definite criteria, 53.3% were probable, and 80.0% satisfied Bocquet's criteria. Only 13.3% satisfied typical and 20.0% atypical DIHS criteria from the Japanese group. Pediatric rheumatologists should be aware of DiHS/DRESS due to its similarities to other systemic inflammatory syndromes (especially systemic JIA, macrophage activation syndrome, and secondary hemophagocytic lymphohistiocytosis). Further studies of DiHS/DRESS syndrome in children are needed to improve its recognition and differential diagnostic and therapeutic options.

Drug-induced hypersensitivity syndrome related to piperacillin-tazobactam: a case report and review of the literature

“…Only five out of 18 patients had normal liver function and the rest had elevated liver enzymes (ALT/AST), with more than half of the patients having liver enzymes more than twice the upper limit of normal values. Kuchinskaya et al ( 20 ) retrospectively analyzed the clinical characteristics of 16 patients with DiHS, and the results showed that all the patients had liver impairment, and no other organ impairment was seen in any patient, which is generally consistent with our findings.…”

Efficacy and safety of Chinese patent medicine combined with 5-aminosalicylic acid for patients with ulcerative colitis: A network meta-analysis of randomized controlled trials

“…Although 5-ASA medications have proven clinical efficacy, some of them can cause serious side effects. For instance, sulfasalazine has been associated with drug eruptions, drug-induced hypersensitivity syndrome, and acute pancreatitis [ [4] , [5] , [6] ]. Salicylic acid preparation and immunosuppressive therapy won't cause any effective effects on some patients [ 7 ].…”