INTRODUCTION

Bee sting can be categorized as follows: localized reaction with pain and edema, immunological reaction that can lead to anaphylactic shock, and systemic toxic reaction caused by release of toxin (hemolysis, coagulopathy, rhabdomyolysis, severe renal insufficiency, and hepatotoxicity). In the most severe cases, the symptoms of arterial thrombotic occlusion, which are caused by hypersensitivity reaction, are uncommon. When bee venom is sensitized, immunoglobulin E antibody is produced. Re-sensitization of bee venom causes mast cell degranulation and release of vasoactive mediators. Biological reaction to this process varies widely in location and severity; such as small local reaction, large local reaction, anaphylactic reaction, toxic reaction, and unusual reaction. If these symptoms are caused by an immunological reaction, anaphylactic shock and systemic toxic reaction may occur [1–4]. These manipulations in symptoms occur according to amount of bee venom, location of sting, species of bee, and immunological reaction caused by venom[1,2,5]. Various rare cases have been reported from outside of Korea, including acute renal failure, hepatitis, rhabdomyolysis, stroke, and myocardial infraction [1,3–5].

We reported on a patient with arterial occlusion and severe ischemia in the lower limb a few hours after multiple bee stings.

CASE

A 58-year-old man, with no cardiovascular risks, and no known or documented allergic history, was admitted to the hospital after he was envenomed by a bee sting. While climbing a mountain, the patient was stung 5 times on frontal, left inferior orbital, right elbow, and left knee. The patient complained of blurred vision and pruriginous cutaneous reactions which occurred at the sting sites, and was treated with antihistamines in the Emergency Room. After 1 hour, the patient felt pallor, pain, and coldness in the left first toe. Whole body computed tomography (CT) scan was performed and thrombus was observed in abdominal aorta, internal iliac artery, and lower extremity arteries (anterior and posterial tibial artery) (Fig. 1). The patient was transferred to our hospital to start treatment for thrombus.

The patient showed an acute ill-looking appearance with alert mentality. Blood pressure was 114/71mmHg, heart rate was 74 beats/min, body temperature was 36.5°C, respiratory rate was 33/min, and no abnormality was detected in chest and abdomen. Signs of bee sting were observed on the face, scalp, and thigh. The patient did not have left dorsalis pedis pulsation and the left 1st toe turned blue and cold.

Hemoglobin, which was normal at time of admission, decreased continuously to 9.9 g/dL. Platelet, prothrombin time, and activated partial thromboplastin time test were, 92,000/μL, 16.7 sec, and >120 sec, respectively, which suggested disseminated intravascular coagulation (DIC) (Tables 1, 2). Laboratory tests showed an increase of creatine kinasemyocardial isoenzyme to 9.93 ng/mL and creatine phosphokinase 1,034 IU/L (Table 1). Electrocardiography and chest X-ray were normal. Cardiac Doppler echo showed normal ejection flection without wall motion disorder and coronary angiography also showed normal flow. However, lower extremity angiography showed thrombus in the internal iliac artery, deep femoral artery and anterior and posterial tibial artery (Fig. 1).

On the first day of admission, tissue plasminogen activator of localized cathetering urokinase was administered, and aspirin 100mg/day, clopidogrel 75mg/day, and thrombolytics were prescribed. A platelet antiaggregant treatment should be considered in association with an anticoagulation treatment. On the second day, dorsalis pedis artery pulsation became palpable and the patient showed normal blood lab and return of left toe color and warmth. After 5 days of treatment, CT angiography showed normal blood flow in artery (Fig. 2). In the absence of any consensus, it was decided to maintain an efficient platelet antiaggregation treatment (clopidogrel 75 mg/day) for 1 year and the patient underwent regular follow-up.

DISCUSSION

Ischemia and thrombus caused by bee sting can range in severity from peripheral vascular disease like this case to acute myocardiac infarction, which can result in death. The pathway of arterial thrombosis and occlusion caused by bee sting is not well known [2,3].

The sequence of events suggests that anaphylaxis reaction was the triggering factor. The patient's immediate symptoms after bee sting are attributed to anaphylaxis. Bee venom is known to contain phospholipase A2 (PLA 2), which is composed of the most lethal peptide and chief allergen. In one study, high levels of PLA2 in the urine and serum were reported in a patient who presented with a typical immediate toxic reaction and a coagulation abnormality. In human plasma, several parameters of coagulation, including prothrombin time, activated partial thromboplastin time, and antithrombin III were affected according to the increase in PLA2 concentration. This peptide catalyzes the hydrolysis of 2-acylbonds in natural lipids, including the structural membrane phospholipids of cells, mitochondria, and other cellular constituents, thereby inhibiting cellular functions. Bee sting causes smooth muscle contraction, lowering blood pressure, increasing capillary permeability and destruction of mast cells, which is known to be related to the coagulation pathway. PLA 2 provokes coagulation abnormality and effects coagulation cascade in all stages. Enzymatic activity chain forms thromboxane A2, which accelerates platelet aggregation leading to coagulation. Thromboxane, luekotriene, and thrombogenic substances from bee venom can secrete vasoactive amine, causing vasospasm and diffuse thrombosis [2,6–10]. Another possibility of thrombus formation detached from thrombi on abdominal aorta along with hemodynamic change as sympathetic nerve stimulation derived from a bee sting could be considered as well [7,8].

Acute arterial occlusion and ischemia after bee sting is rare. There are no guidelines for surgery and medication, and no articles on treatment have been reported in Korea.

The current case was the first in which emergency percutaneous transluminal angioplasty (PTA) was attempted in a patient with thrombus caused by bee sting. CT angiography was performed to determine the location and severity of the thrombus because of a large quantity of thrombus blocking the internal iliac artery and thrombus circulating in lower extremity arterial aorta. PTA was used to catheterize a channel in the artery, but was not 100% successful; then left posterior tibial artery ballooning was performed and fibrolytic agent was injected (urokinase) 200,000 IU bolus (1/3 of the recommended dosage). After injection, continuous infusion of 2,300,000 IU mixed fluid was administered to prevent DIC progression from decrease of medication and targeting thrombolysis. Then antiplatelet drug, aspirin 100mg/day and clopidogrel 75mg/day was prescribed and after a few days normal blood flow in artery was observed on CT angiography.

There is no known treatment for arterial occlusion and ischemia in patients with DIC progression. We believe that in this case, deposition of immune complexes in the basement membrane of small blood vessels and activation of the complement system may also have contributed to the pathogenesis of the disease and an anaphylactic reaction to bee venom mediated the development of DIC. The severity of clinical and laboratory manifestations varies among patients, even when the disorder is produced by the same agent. The reasons and treatment for this variability are unclear [8,9].

Inappropriate thrombolysis treatment could provoke aggravation to systemic disease. We focused on regional restricted thrombolysis targeting occlusion of the left posterior tibial artery and accurate anti-platelet agent dosage maintenance. Use of these prudent approaches led to achievement of a good result.

Notes

Conflicts of interest

The authors report no potential conflict of interest relevant to this article.

Fig. 1

Computed tomography on the first day after bee sting. (A) Thrombus in the left internal iliac artery (black arrow) and right deep femoral artery (white arrow). (B) Image showed severe stenosis of the left anterior and posterior tibial artery.

Fig. 2

Extremity arteriography left: before and after percutaneous transluminal angioplasty (PTA). (A) (pre PTA) Percutaneous transluminal angiographic imaging showed 70% to 80% narrowing of the left posterior tibial artery. (B) PTA to catheterize a channel in the artery. (C) (post PTA) After performance of ballooning and injection of fibrolytic agent, arterial flow was restored.

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Figure & Data

References

Citations

Citations to this article as recorded by  

• A Rare Case of Acute Lower Limb Ischemia following Bee Sting
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