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Dl-3-n-butylphthalide (DL-NBP) has good neuroprotective function and is safe for use in patients with acute ischaemic stroke. DL-NBP induced anaphylactic shock is rarely reported. Here we describe the case of a 75-year-old woman who received an injection of DL-NBP (25 mg/100 mL intravenously guttae, twice daily) for acute ischaemic stroke. Approximately 5 min after the DL-NBP injection was administered, the patient developed a decrease in blood pressure and an increase in heart rate along with skin pruritus, mottlement of the lower limbs, discomfort, and the desire to defecate, following which DL-NBP was discontinued immediately. The patient recovered with antiallergic therapy and could tolerate further treatment. We emphasise that the increased use of DL-NBP in recent year raises the importance of attention to potential allergies in clinical use, especially in patients with a history of allergies to multiple drugs.