□
CASE REPORT
□
Guillain-Barré Syndrome Following Bee Venom Acupuncture Hyun Jo Lee 1, In Seok Park 1, Jon-In Lee 2 and Joong-Seok Kim 1
Abstract Bee venom acupuncture has been widely used in Oriental medicine with limited evidence of effectiveness. Most of the complications due to bee venom acupuncture are local or systemic allergic reactions. However, serious medical and neurological complications have also been reported. We herein describe the treatment of a 68-year-old woman who developed progressive quadriplegia 10 days after receiving multiple honeybee venom sting acupuncture treatments. The electrophysiological findings were consistent with Guillain-Barré syndrome (GBS). The temporal relationship between the development of GBS and honeybee venom sting acupuncture is suggestive of a cause-and-effect relationship, although the precise pathophysiology and causative components in honeybee venom need to be verified. Key words: bee venom acupuncture, Guillain-Barré syndrome (Intern Med 54: 975-978, 2015) (DOI: 10.2169/internalmedicine.54.2238)
venom acupuncture.
Introduction Case Report Guillain-Barré syndrome (GBS) is an acute polyneuropathy clinically characterized by acroparesthesia and progressive weakness. In most cases, various antecedent viral or bacterial infections and immunizations are noted 2-4 weeks before the onset of GBS (1, 2). In some cases, biological toxins have been reported as a cause of GBS (3, 4). Acupuncture therapies using the venom of Apis mellifera (in Korean, Bongdok), commonly known as the honeybee, have been extensively used in Oriental medicine with limited evidence of effectiveness. This procedure can cause several adverse events such as local allergic reactions at the site of stings (e.g., pain, swelling, erythema, pruitis, and urticaria), unusual toxic reactions (e.g., anaphylaxis), or serious medical or neurological complications (e.g., serum sickness, vasculitis, nephrosis, neuritis, and encephalitis) (5). Therefore, it is important that physicians, neurologists, and associated healthcare providers are aware of such adverse events. We herein report a patient who developed GBS 10 days after receiving honeybee venom sting acupuncture. This is the first case of iatrogenic GBS following exposure to bee
A 68-year-old woman was admitted to the emergency room with progressive symmetrical muscle weakness and tingling sensation in both legs for 3 days. For approximately two years the patient has had pain in both of her knees and lower back. Two weeks prior to her admission, the patient began to receive multiple site honeybee venom sting acupuncture treatments on her knees and lower back by an apitherapist for the purpose of pain relief. She received bee venom acupuncture on a twice-per-week schedule with approximately 10 acupunctures each session. These procedures were repeated three times. The patient began to experience weakness and difficulty walking 10 days after the first bee venom acupuncture, which progressed to her upper limbs. There were no antecedent events of fever, upper respiratory infection, diarrhea, or vaccination. The patient had wellcontrolled hypertension. She also had a history of tumor resection for early colon cancer 8 years ago. She did not receive any chemotherapy because the results of surgical assessment was complete resection with no remnant tumor. No
１
Department of Neurology, College of Medicine, The Catholic University of Korea, Korea and ２Department of Rehabilitation Medicine, College of Medicine, The Catholic University of Korea, Korea Received for publication December 2, 2013; Accepted for publication September 11, 2014 Correspondence to Dr. Joong-Seok Kim, [email protected]
975
Intern Med 54: 975-978, 2015
DOI: 10.2169/internalmedicine.54.2238
Table 1. Results of Nerve Conduction Studies Performed on the Second Hospital Day Motor nerve Median (right/left)
Ulnar (right/left)
Common peroneal (right/left)
Tibial (right/left)
Sensory nerve Median (right/left)
Ulnar (right/left)
Sural (right/left)
Segment TL
Latency (ms) 9.4/7.65(< 4.0)
Amplitude (mV) 2.0/2.9 (>6)
NCV (m/s)
W-E
12.75/11.9
1.9/2.6 (>6)
56.7/48.2 (>50.5)
E-AX
14.40/13.0
1.8/2.4 (>6)
54.5/63.6 (>51.2)
F-wave
NP / NP (8)
66.7/62.8 (>51.2)
BE-AE
11.45/11.65
3.3/3.3 (>8)
54.3/51.7 (>51.6)
AE-AX
16.20/16.42
3.2/3.2 (>8)
54.7/53.2 (>52.7)
F-wave
NP / NP (1.5)
F-wave
NP / NP (6)
F-wave
NP / NP (39.3)
W-E
19.8/24.8 (>15)
67.2/65.3 (>50)
E-AX
48.6/75.1 (>12.1)
64.7/77.3 (>48.8)
F-W
7.5/7.2 (>7.9)
41.4/46.0 (>37.5)
W-E
22.4/17.5 (>13.8)
67.2/69.2 (>48.5)
E-AX
28.9/28.5 (>12.1)
82.6/76.7 (>44.1)
MC
11.8/13.2 (>6)
66.6/68.8 (>32.1)
50.7/49.8 (>40.5)
41.2/44.3 (>40.5)
TL: terminal latency, W: wrist, E: elbow, AX: axilla, BE: below elbow, AE: above elbow, PF: popliteal fossa, K: knee, A: ankle, F: finger, MC: middle calf, NP: No potential evoked
personal history of neurological disease was noted. On examination, the patient was alert and oriented with a blood pressure of 125/79 mmHg, a regular heartbeat of 77 beats/min, normal breath rate of 15/min, and a temperature of 36.2℃. A cranial nerve examination did not disclose any abnormalities, and a sensory examination was normal. Her motor power was symmetric with 4 of 5 on the bilateral upper and lower extremities. Total areflexia was noted, but no pathological reflexes were recorded. The laboratory testing, including a complete blood count, blood chemistry, thyroid function test, vasculitis markers, HIV antibody, and the rapid plasma reagin test for syphilis, was within normal limits. Cultures and serologic studies for Campylobacter jejuni, cytomegalovirus, Epstein-Barr virus, and Mycoplasma pneumoniae were found to be negative. Paraneoplastic antibodies (anti-HU, anti-Ri, anti-Yo, antiMAG, and anti-myelin) were also negative. A cerebrospinal fluid (CSF) examination revealed no white blood cells, a protein level of 78.4 mg/dL, and a glucose level of 67 mg/
dL (simultaneous blood glucose level was 106 mg/dL). CSF oligoclonal bands, serum IgG anti-GM1 antibodies, and antiGQIb antibodies were negative. Nerve conduction studies, performed on the second hospital day, revealed decreased amplitudes of the compound motor nerve action potentials and prolonged terminal latencies of bilateral median, ulnar, common peroneal, and tibial nerves. Late responses, including F waves and H reflex, were not detected. However, no abnormalities were detected in antidromic methods for studying sensory nerve action potentials (Table 1). Two days after admission, the patient’s weakness progressed to grade 3 in the distal limbs and grade 2 in the proximal limbs bilaterally. Diffuse pain and tenderness of both thighs and paraspinal muscles were also noted. The patient developed facial diplegia, dysarthria, and dysphagia. Treatment was started with intravenous immunoglobulin (400 mg/kg/day) for 5 days. Two day after treatment, the patient developed hypoxemia of 85% in ambient air and mild tachypnea (26/min). A physical examination disclosed
976
Intern Med 54: 975-978, 2015
DOI: 10.2169/internalmedicine.54.2238
Table 2. Results of Follow-up Nerve Conduction Studies Performed on the 14th Hospital Day Motor nerve Median (right/left)
Ulnar (right/left)
Common peroneal (right/left)
Tibial (right/left)
Sensory nerve Median (right/left)
Ulnar (right/left)
Segment TL
Latency (ms) 15.15/17.15(< 4.0)
Amplitude (mV) 2.1/1.1 (>6)
NCV (m/s)
W-E
18.80/21.30
1.9/0.9 (>6)
52.1/50.6 (> 50.5)
E-AX
20.35/19.90
1.8/0.9 (>6)
51.6/57.1 (>51.2)
F-wave
NP / NP (8)
46.8/41.7 (>51.2)
BE-AE
17.30/16.25
0.8/1.1 (>8)
55.6/50.0 (>51.6)
AE-AX
18.20/18.20
0.8/1.0 (>8)
50.0/46.2 (>52.7)
F-wave
NP / NP (1.5)
F-wave
NP / NP (6)
F-wave
NP / NP (15)
64.5/67.9 (>50)
E-AX
45.6/68.9 (>12.1)
64.0/55.9 (>48.8)
F-W
NP/NP (>7.9)
45.4/44.3 (>40.5)
NP/41.2 (>40.5)
NCV (m/s)
W-E
9.4/6.9 (>13.8)
79.2/75.8 (>48.5)
E-AX
28.1/21.3 (>12.1)
67.7/90.9 (>44.1)
Sural MC 10.1/11.8 (>6) 61.5/70.6 (>32.1) (right/left) TL: terminal latency, W: wrist, E: elbow, AX: axilla, BE: below elbow, AE: above elbow, PF: popliteal fossa, K: knee, A: ankle, F: finger, MC: middle calf, NP: No potential evoked
decreased breath sound over both lung fields. A chest X-ray obtained on the fifth hospital day showed mild consolidation in the right middle to lower lung area. A blood gas analysis with a 6 L/min oxygen supply delivered via mask showed hypoxemia, 68.5 mmHg; PaCO2, 40.1 mmHg; pH, 7.500; and bicarbonate, 31.0 mM/L. The patient was also diagnosed as having aspiration pneumonia and admitted to the intensive care unit. Her respiratory status was remarkably improved after endotracheal intubation and antibiotic treatment. Nine days after admission, the patient’s muscle power began to clinically improve; two months after admission, her manual muscle test grade returned to normal, and she was discharged without complications. Follow-up nerve conduction studies were performed nine days after treatment and revealed the same pattern, but more severe abnormalities on compound motor action potentials. In addition, diminished median and ulnar sensory nerve action potentials in the distal part were detected. These results suggested a demyelinating motor/sensory polyneuropathy,
which was mainly present in extremely distal parts of the limbs (Table 2).
Discussion The venom of Apis mellifera, commonly known as the honeybee, contains various compounds. Some of these venomous components have functions that act directly on the tissue, whereas others activate proteins. These include biogenic amines (e.g., acetylcholine, dopamine, histamine, norepinephrine, and serotonin), polypeptides, protein toxins (e.g., apamin, tertiapin, melittin, and kinins), and enzymes (e.g., hyaluronidase, phospholipase A2, and acid phosphatase) (6). These compounds lead to local or systemic immunologic reactions that are expressed through a variety of local or systemic allergic reactions. Neurologic complications that are also presumed to be immune-medicated reactions can occur with bee venom poisoning. These include myasthenia gravis, myeloradiculopathy, optic neuropathy,
977
Intern Med 54: 975-978, 2015
DOI: 10.2169/internalmedicine.54.2238
parkinsonism, and stroke (7-11). Few case reports of GBS and its variants associated directly with bee stings have been reported (12, 13). In these reports, the patients had GBS symptoms 2-5 days after exposure to one or multiple bee stings. The causative insects were various types of hymenoptera, such as honeybees or wasps, rather than one kind of bee. However, no clear pathophysiology has yet been clarified in regard to which bee venom components may cause the immunologic reaction that induces GBS. In our patient, GBS symptoms appeared 10 days after multiple honeybee venom sting acupunctures. The occurrence of GBS following bee venom acupuncture with no symptoms of infection, no history of vaccination, or other well-known antecedent episodes for GBS suggests a possible association between the two conditions. A potential explanation is that both a direct toxic effect of the bee venom and an indirect effect involving an immunological reaction contributed to the development of GBS, although we cannot be entirely sure of the role of bee venom acupuncture in our patient because re-challenge experiments could not be performed. Considering the temporal association of the symptoms with the history of bee venom acupuncture, as well as the absence of any other antecedent event, we considered the “demyelinating” GBS as secondary to some of these venomous components. Although the pathophysiology between infection and GBS comprises molecular mimicry (14) as the causative mechanism, it remains unproven in this case. The possibility that subclinical infection associated with the procedure of bee venom acupuncture should also be considered. In addition, it is possible that the patient’s symptoms were unrelated to the bee venom acupuncture, so other factors cannot be ruled out for the cause of this condition. However, we know of no naturally occurring illness with all of the features exhibited by our patient. Therefore, we speculate that the bee venom acupuncture
resulted in GBS in this case, although we have not unequivocally established a cause-and-effect relationship. This report alerts neurologists and physicians that patients may develop adverse events after undergoing bee venom acupuncture. Moreover, bee venom acupuncture may be a possible cause of GBS. The authors state that they have no Conflict of Interest (COI).
References 1. Dimachkie MM, Barohn RJ. Guillain-Barré syndrome and variants. Neurol Clin 31: 491-510, 2013. 2. Uncini A, Kuwabara S. Electrodiagnostic criteria for GuillainBarré syndrome: a critical revision and the need for an update. Clin Neurophysiol 123: 1487-1495, 2012. 3. Devere R. Guillain-Barré syndrome after a jellyfish sting. J Clin Neuromuscul Dis 12: 227-230, 2011. 4. Neil J, Choumet V, Le Coupanec A, d’Alayer J, Demeret S, Musset L. Guillain-Barré syndrome: first description of a snake envenomation aetiology. J Neuroimmunol 242: 72-77, 2012. 5. Reisman RE. Insect stings. N Eng J Med 331: 523-527, 1994. 6. Brown TC, Tankersley MS. The sting of the honeybee: an allergic perspective. Ann Allergy Asthma Immunol 107: 463-470, 2011. 7. Brumlik J. Myasthenia gravis associated with wasp sting. JAMA 235: 2120-2121, 1976. 8. Leopold NA, Bara-Jimenez W, Hallett M. Parkinsonism after a wasp sting. Mov Disord 14: 122-127, 1999. 9. Maltzman JS, Lee AG, Miller NR. Optic neuropathy occurring after bee and wasp sting. Ophthalmology 107: 193-195, 2000. 10. Park JH, Jung MK, Lee TK, Ahn MY, Bang CO. A case of ischemic stroke following bee venom acupuncture. J Korean Neurol Assoc 18: 356-358, 2000. 11. Van Antwerpen CL, Gospe SM Jr, Wade NA. Myeloradiculopathy associated with wasp sting. Pediatr Neurol 4: 379-380, 1988. 12. Marks HG, Augustyn P, Allen RJ. Fisher’s syndrome in children. Pediatrics 60: 726-729, 1977. 13. Bachman DS, Paulson GW, Mendell JR. Acute inflammatory polyradiculoneuropathy following Hymenoptera stings. JAMA 247: 1443-1445, 1982. 14. Ang CW, Jacobs BC, Laman JD. The Guillain-Barré syndrome: a true case of molecular mimicry. Trends Immunol 25: 61-66, 2004.
Ⓒ 2015 The Japanese Society of Internal Medicine http://www.naika.or.jp/imonline/index.html
978
CASE REPORT
□
Guillain-Barré Syndrome Following Bee Venom Acupuncture Hyun Jo Lee 1, In Seok Park 1, Jon-In Lee 2 and Joong-Seok Kim 1
Abstract Bee venom acupuncture has been widely used in Oriental medicine with limited evidence of effectiveness. Most of the complications due to bee venom acupuncture are local or systemic allergic reactions. However, serious medical and neurological complications have also been reported. We herein describe the treatment of a 68-year-old woman who developed progressive quadriplegia 10 days after receiving multiple honeybee venom sting acupuncture treatments. The electrophysiological findings were consistent with Guillain-Barré syndrome (GBS). The temporal relationship between the development of GBS and honeybee venom sting acupuncture is suggestive of a cause-and-effect relationship, although the precise pathophysiology and causative components in honeybee venom need to be verified. Key words: bee venom acupuncture, Guillain-Barré syndrome (Intern Med 54: 975-978, 2015) (DOI: 10.2169/internalmedicine.54.2238)
venom acupuncture.
Introduction Case Report Guillain-Barré syndrome (GBS) is an acute polyneuropathy clinically characterized by acroparesthesia and progressive weakness. In most cases, various antecedent viral or bacterial infections and immunizations are noted 2-4 weeks before the onset of GBS (1, 2). In some cases, biological toxins have been reported as a cause of GBS (3, 4). Acupuncture therapies using the venom of Apis mellifera (in Korean, Bongdok), commonly known as the honeybee, have been extensively used in Oriental medicine with limited evidence of effectiveness. This procedure can cause several adverse events such as local allergic reactions at the site of stings (e.g., pain, swelling, erythema, pruitis, and urticaria), unusual toxic reactions (e.g., anaphylaxis), or serious medical or neurological complications (e.g., serum sickness, vasculitis, nephrosis, neuritis, and encephalitis) (5). Therefore, it is important that physicians, neurologists, and associated healthcare providers are aware of such adverse events. We herein report a patient who developed GBS 10 days after receiving honeybee venom sting acupuncture. This is the first case of iatrogenic GBS following exposure to bee
A 68-year-old woman was admitted to the emergency room with progressive symmetrical muscle weakness and tingling sensation in both legs for 3 days. For approximately two years the patient has had pain in both of her knees and lower back. Two weeks prior to her admission, the patient began to receive multiple site honeybee venom sting acupuncture treatments on her knees and lower back by an apitherapist for the purpose of pain relief. She received bee venom acupuncture on a twice-per-week schedule with approximately 10 acupunctures each session. These procedures were repeated three times. The patient began to experience weakness and difficulty walking 10 days after the first bee venom acupuncture, which progressed to her upper limbs. There were no antecedent events of fever, upper respiratory infection, diarrhea, or vaccination. The patient had wellcontrolled hypertension. She also had a history of tumor resection for early colon cancer 8 years ago. She did not receive any chemotherapy because the results of surgical assessment was complete resection with no remnant tumor. No
１
Department of Neurology, College of Medicine, The Catholic University of Korea, Korea and ２Department of Rehabilitation Medicine, College of Medicine, The Catholic University of Korea, Korea Received for publication December 2, 2013; Accepted for publication September 11, 2014 Correspondence to Dr. Joong-Seok Kim, [email protected]
975
Intern Med 54: 975-978, 2015
DOI: 10.2169/internalmedicine.54.2238
Table 1. Results of Nerve Conduction Studies Performed on the Second Hospital Day Motor nerve Median (right/left)
Ulnar (right/left)
Common peroneal (right/left)
Tibial (right/left)
Sensory nerve Median (right/left)
Ulnar (right/left)
Sural (right/left)
Segment TL
Latency (ms) 9.4/7.65(< 4.0)
Amplitude (mV) 2.0/2.9 (>6)
NCV (m/s)
W-E
12.75/11.9
1.9/2.6 (>6)
56.7/48.2 (>50.5)
E-AX
14.40/13.0
1.8/2.4 (>6)
54.5/63.6 (>51.2)
F-wave
NP / NP (8)
66.7/62.8 (>51.2)
BE-AE
11.45/11.65
3.3/3.3 (>8)
54.3/51.7 (>51.6)
AE-AX
16.20/16.42
3.2/3.2 (>8)
54.7/53.2 (>52.7)
F-wave
NP / NP (1.5)
F-wave
NP / NP (6)
F-wave
NP / NP (39.3)
W-E
19.8/24.8 (>15)
67.2/65.3 (>50)
E-AX
48.6/75.1 (>12.1)
64.7/77.3 (>48.8)
F-W
7.5/7.2 (>7.9)
41.4/46.0 (>37.5)
W-E
22.4/17.5 (>13.8)
67.2/69.2 (>48.5)
E-AX
28.9/28.5 (>12.1)
82.6/76.7 (>44.1)
MC
11.8/13.2 (>6)
66.6/68.8 (>32.1)
50.7/49.8 (>40.5)
41.2/44.3 (>40.5)
TL: terminal latency, W: wrist, E: elbow, AX: axilla, BE: below elbow, AE: above elbow, PF: popliteal fossa, K: knee, A: ankle, F: finger, MC: middle calf, NP: No potential evoked
personal history of neurological disease was noted. On examination, the patient was alert and oriented with a blood pressure of 125/79 mmHg, a regular heartbeat of 77 beats/min, normal breath rate of 15/min, and a temperature of 36.2℃. A cranial nerve examination did not disclose any abnormalities, and a sensory examination was normal. Her motor power was symmetric with 4 of 5 on the bilateral upper and lower extremities. Total areflexia was noted, but no pathological reflexes were recorded. The laboratory testing, including a complete blood count, blood chemistry, thyroid function test, vasculitis markers, HIV antibody, and the rapid plasma reagin test for syphilis, was within normal limits. Cultures and serologic studies for Campylobacter jejuni, cytomegalovirus, Epstein-Barr virus, and Mycoplasma pneumoniae were found to be negative. Paraneoplastic antibodies (anti-HU, anti-Ri, anti-Yo, antiMAG, and anti-myelin) were also negative. A cerebrospinal fluid (CSF) examination revealed no white blood cells, a protein level of 78.4 mg/dL, and a glucose level of 67 mg/
dL (simultaneous blood glucose level was 106 mg/dL). CSF oligoclonal bands, serum IgG anti-GM1 antibodies, and antiGQIb antibodies were negative. Nerve conduction studies, performed on the second hospital day, revealed decreased amplitudes of the compound motor nerve action potentials and prolonged terminal latencies of bilateral median, ulnar, common peroneal, and tibial nerves. Late responses, including F waves and H reflex, were not detected. However, no abnormalities were detected in antidromic methods for studying sensory nerve action potentials (Table 1). Two days after admission, the patient’s weakness progressed to grade 3 in the distal limbs and grade 2 in the proximal limbs bilaterally. Diffuse pain and tenderness of both thighs and paraspinal muscles were also noted. The patient developed facial diplegia, dysarthria, and dysphagia. Treatment was started with intravenous immunoglobulin (400 mg/kg/day) for 5 days. Two day after treatment, the patient developed hypoxemia of 85% in ambient air and mild tachypnea (26/min). A physical examination disclosed
976
Intern Med 54: 975-978, 2015
DOI: 10.2169/internalmedicine.54.2238
Table 2. Results of Follow-up Nerve Conduction Studies Performed on the 14th Hospital Day Motor nerve Median (right/left)
Ulnar (right/left)
Common peroneal (right/left)
Tibial (right/left)
Sensory nerve Median (right/left)
Ulnar (right/left)
Segment TL
Latency (ms) 15.15/17.15(< 4.0)
Amplitude (mV) 2.1/1.1 (>6)
NCV (m/s)
W-E
18.80/21.30
1.9/0.9 (>6)
52.1/50.6 (> 50.5)
E-AX
20.35/19.90
1.8/0.9 (>6)
51.6/57.1 (>51.2)
F-wave
NP / NP (8)
46.8/41.7 (>51.2)
BE-AE
17.30/16.25
0.8/1.1 (>8)
55.6/50.0 (>51.6)
AE-AX
18.20/18.20
0.8/1.0 (>8)
50.0/46.2 (>52.7)
F-wave
NP / NP (1.5)
F-wave
NP / NP (6)
F-wave
NP / NP (15)
64.5/67.9 (>50)
E-AX
45.6/68.9 (>12.1)
64.0/55.9 (>48.8)
F-W
NP/NP (>7.9)
45.4/44.3 (>40.5)
NP/41.2 (>40.5)
NCV (m/s)
W-E
9.4/6.9 (>13.8)
79.2/75.8 (>48.5)
E-AX
28.1/21.3 (>12.1)
67.7/90.9 (>44.1)
Sural MC 10.1/11.8 (>6) 61.5/70.6 (>32.1) (right/left) TL: terminal latency, W: wrist, E: elbow, AX: axilla, BE: below elbow, AE: above elbow, PF: popliteal fossa, K: knee, A: ankle, F: finger, MC: middle calf, NP: No potential evoked
decreased breath sound over both lung fields. A chest X-ray obtained on the fifth hospital day showed mild consolidation in the right middle to lower lung area. A blood gas analysis with a 6 L/min oxygen supply delivered via mask showed hypoxemia, 68.5 mmHg; PaCO2, 40.1 mmHg; pH, 7.500; and bicarbonate, 31.0 mM/L. The patient was also diagnosed as having aspiration pneumonia and admitted to the intensive care unit. Her respiratory status was remarkably improved after endotracheal intubation and antibiotic treatment. Nine days after admission, the patient’s muscle power began to clinically improve; two months after admission, her manual muscle test grade returned to normal, and she was discharged without complications. Follow-up nerve conduction studies were performed nine days after treatment and revealed the same pattern, but more severe abnormalities on compound motor action potentials. In addition, diminished median and ulnar sensory nerve action potentials in the distal part were detected. These results suggested a demyelinating motor/sensory polyneuropathy,
which was mainly present in extremely distal parts of the limbs (Table 2).
Discussion The venom of Apis mellifera, commonly known as the honeybee, contains various compounds. Some of these venomous components have functions that act directly on the tissue, whereas others activate proteins. These include biogenic amines (e.g., acetylcholine, dopamine, histamine, norepinephrine, and serotonin), polypeptides, protein toxins (e.g., apamin, tertiapin, melittin, and kinins), and enzymes (e.g., hyaluronidase, phospholipase A2, and acid phosphatase) (6). These compounds lead to local or systemic immunologic reactions that are expressed through a variety of local or systemic allergic reactions. Neurologic complications that are also presumed to be immune-medicated reactions can occur with bee venom poisoning. These include myasthenia gravis, myeloradiculopathy, optic neuropathy,
977
Intern Med 54: 975-978, 2015
DOI: 10.2169/internalmedicine.54.2238
parkinsonism, and stroke (7-11). Few case reports of GBS and its variants associated directly with bee stings have been reported (12, 13). In these reports, the patients had GBS symptoms 2-5 days after exposure to one or multiple bee stings. The causative insects were various types of hymenoptera, such as honeybees or wasps, rather than one kind of bee. However, no clear pathophysiology has yet been clarified in regard to which bee venom components may cause the immunologic reaction that induces GBS. In our patient, GBS symptoms appeared 10 days after multiple honeybee venom sting acupunctures. The occurrence of GBS following bee venom acupuncture with no symptoms of infection, no history of vaccination, or other well-known antecedent episodes for GBS suggests a possible association between the two conditions. A potential explanation is that both a direct toxic effect of the bee venom and an indirect effect involving an immunological reaction contributed to the development of GBS, although we cannot be entirely sure of the role of bee venom acupuncture in our patient because re-challenge experiments could not be performed. Considering the temporal association of the symptoms with the history of bee venom acupuncture, as well as the absence of any other antecedent event, we considered the “demyelinating” GBS as secondary to some of these venomous components. Although the pathophysiology between infection and GBS comprises molecular mimicry (14) as the causative mechanism, it remains unproven in this case. The possibility that subclinical infection associated with the procedure of bee venom acupuncture should also be considered. In addition, it is possible that the patient’s symptoms were unrelated to the bee venom acupuncture, so other factors cannot be ruled out for the cause of this condition. However, we know of no naturally occurring illness with all of the features exhibited by our patient. Therefore, we speculate that the bee venom acupuncture
resulted in GBS in this case, although we have not unequivocally established a cause-and-effect relationship. This report alerts neurologists and physicians that patients may develop adverse events after undergoing bee venom acupuncture. Moreover, bee venom acupuncture may be a possible cause of GBS. The authors state that they have no Conflict of Interest (COI).
References 1. Dimachkie MM, Barohn RJ. Guillain-Barré syndrome and variants. Neurol Clin 31: 491-510, 2013. 2. Uncini A, Kuwabara S. Electrodiagnostic criteria for GuillainBarré syndrome: a critical revision and the need for an update. Clin Neurophysiol 123: 1487-1495, 2012. 3. Devere R. Guillain-Barré syndrome after a jellyfish sting. J Clin Neuromuscul Dis 12: 227-230, 2011. 4. Neil J, Choumet V, Le Coupanec A, d’Alayer J, Demeret S, Musset L. Guillain-Barré syndrome: first description of a snake envenomation aetiology. J Neuroimmunol 242: 72-77, 2012. 5. Reisman RE. Insect stings. N Eng J Med 331: 523-527, 1994. 6. Brown TC, Tankersley MS. The sting of the honeybee: an allergic perspective. Ann Allergy Asthma Immunol 107: 463-470, 2011. 7. Brumlik J. Myasthenia gravis associated with wasp sting. JAMA 235: 2120-2121, 1976. 8. Leopold NA, Bara-Jimenez W, Hallett M. Parkinsonism after a wasp sting. Mov Disord 14: 122-127, 1999. 9. Maltzman JS, Lee AG, Miller NR. Optic neuropathy occurring after bee and wasp sting. Ophthalmology 107: 193-195, 2000. 10. Park JH, Jung MK, Lee TK, Ahn MY, Bang CO. A case of ischemic stroke following bee venom acupuncture. J Korean Neurol Assoc 18: 356-358, 2000. 11. Van Antwerpen CL, Gospe SM Jr, Wade NA. Myeloradiculopathy associated with wasp sting. Pediatr Neurol 4: 379-380, 1988. 12. Marks HG, Augustyn P, Allen RJ. Fisher’s syndrome in children. Pediatrics 60: 726-729, 1977. 13. Bachman DS, Paulson GW, Mendell JR. Acute inflammatory polyradiculoneuropathy following Hymenoptera stings. JAMA 247: 1443-1445, 1982. 14. Ang CW, Jacobs BC, Laman JD. The Guillain-Barré syndrome: a true case of molecular mimicry. Trends Immunol 25: 61-66, 2004.
Ⓒ 2015 The Japanese Society of Internal Medicine http://www.naika.or.jp/imonline/index.html
978
