A 48-year-old female patient was treated with hydroxychloroquine sulfate and methotrexate for rheumatoid arthritis. After two weeks, tender erythema and papules appeared on the trunk and limbs, accompanied by fever. Skin biopsy was suggestive of neutrophilic infiltration in the superficial mesodermal layer of the dermis. After hydroxychloroquine sulfate withdrawal and 18 days of methylprednisolone sodium succinate and halometasone cream treatment, she improved and was diagnosed with acute febrile neutrophilic dermatosis. By correlation analysis, the causal relationship between acute febrile neutrophilic dermatosis and hydroxychloroquine sulfate was evaluated as “most likely”, which had not been reported in China. The pathogenesis of hydroxychloroquine sulfate-induced acute febrile neutrophilic dermatosis may be related to elevated granulocyte-colony stimulating factor levels, abnormal cytokine regulation, and genetic susceptibility. Discontinuation of the suspected drug and application of glucocorticoids are currently the most effective treatment.

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