Cerebral infarction after anaphylactic shock due to cold-induced urticaria

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Cold-induced urticaria can lead to anaphylaxis, biphasic reactions and stroke. Prompt diagnosis is necessary because thrombolytic therapy may help resolve the condition. Cold-induced urticaria is characterized by pruritic wheals and/or angioedema due to the release of mast cell degranulation and inflammatory mediators. Cold-induced urticaria rarely causes anaphylaxis1; to our knowledge, no reports exist of thrombotic cerebral infarction from anaphylaxis. We report a case of cerebral infarction after anaphylactic shock due to cold-induced urticaria. A 60-year-old woman noticed urticaria all over her body after about 15 min of swimming in a cold river. She also fainted and had diarrhoea; thus, she called an ambulance. She was transported to our hospital by an emergency medical service helicopter. She had no history of chronic diseases or food/drug allergies. However, according to her anamnesis, holding a cold plastic bottle often caused redness and swelling in the said arm. In the helicopter, her vital signs were as follows: blood pressure, 58/40 mmHg; pulse rate, 75 beats/min; oxygen saturation, 96%; body temperature, 35.6°C; respiratory rate, 20 breaths/min; and blood glucose, 168 mg/dl. No neurological abnormalities were observed. Since she met the anaphylactic shock criteria, an intramuscular injection of 0.3 mg adrenaline was administered before take-off, followed by an infusion of extracellular fluid and intravenous antihistamine. On arrival at the hospital, her blood pressure had improved to 104/57 mmHg, and her urticaria was overall reduced. Her electrocardiogram showed normal sinus rhythm. Her blood test results showed an increased haematocrit level (50.3%), red blood cell count (558 × 106/μl), haemoglobin level (16.5 g/dl) and D-dimer (2.9 μg/ml). No pathological findings were observed on brain computed tomography. One hour post-arrival at the hospital, her consciousness level dropped to a Glasgow Coma Scale score of E3V3M6, her urticaria got worse, and blood pressure became unmeasurable; hence, an intramuscular injection of 0.3 mg adrenaline was administered again for biphasic anaphylactic shock. However, the disturbance of consciousness persisted; she developed left upper and lower limb paralysis. Manual muscle strength tests of the left upper and lower limbs showed scores of 4. Her National Institutes of Health Stroke Scale (NIHSS) score was 7. Brain magnetic resonance imaging and magnetic resonance angiography (MRA) were performed. Diffusion-weighted imaging showed a hyperintense area in the right parietal regions; MRA showed right middle cerebral artery occlusion (Figure 1a). There was no cerebral artery dissection. After thrombolytic treatment with alteplase (0.6 mg/kg) was initiated, her consciousness improved. After endovascular thrombectomy, the occlusion site was recanalized, and her NIHSS score improved to 0 (Figure 1b). She was discharged home after 8 days of hospitalization, fully recovered. (a) Brain magnetic resonance imaging and magnetic resonance angiography before thrombolytic and endovascular therapy. (b) Cerebral angiography after thrombolytic therapy and recanalization. The patient experienced anaphylactic shock characterized by hypotension and diarrhoea, in addition to erythema, after exposure to a cold stimulus. The incidence of cold-induced urticaria is 0.05%; it is common in cold regions.1 In rare cases, it can cause anaphylaxis and biphasic reactions, as in this case. Systemic reactions are rarely caused after exposure to a cold object; rather, they mostly follow aquatic activities.2 MRA showed left middle cerebral artery occlusion, which improved with thrombolytic therapy. While ischaemia due to an allergic reaction is rare, myocardial infarction in anaphylactic patients is known as Kounis syndrome.3 Although previous reports have described cases of cerebral infarction without stenosis,4 our patient had cerebral infarction with obstruction. Early administration of alteplase led to complete improvement of her neurological function. Anaphylactic shock causes a more pronounced reduction in cerebral blood flow than in blood pressure;5 intravascular dehydration and histamine-induced hypercoagulability may also occur. The symptoms of anaphylaxis caused by cold stimulus in patients who also develop stroke symptoms may completely improve with early thrombolytic and endovascular therapy, as in this case. Therefore, early diagnosis and treatment of both the allergic reaction and stroke should be considered. Written informed consent for publication was obtained from the patient. Ethics approval was not needed for the single case report. The authors received no financial support for the research, authorship and/or publication of this article. Conflict of interest: None declared. Shinya Takeuchi (Conceptualization [lead], Writing—original draft [lead], Writing—review & editing [supporting]), Masato Miyauchi (Conceptualization [supporting], Project administration [supporting], Supervision [lead], Writing—review & editing [equal]), Tomohito Kadota (Supervision [supporting], Writing—review & editing [supporting]), Maki Fukuda (Supervision [supporting], Visualization [supporting], Writing—review & editing [supporting]), and Kingo Nishiyama (Project administration [lead], Supervision [supporting], Writing—review & editing [supporting]). Data sharing is not applicable to this article as no datasets were generated or analysed during the current study.

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