Systemic mastocytosis

We describe two patients with claw hand as a result of a bee sting. It is likely that this was caused by the apamin in the sting which has an effect on the upper limb, at the spinal cord and on the peripheral nerves. It is important to recognise that the claw hand is not owing to compartment syndrome. Both patients were treated conservatively with full resolution within 48 hours, without any lasting effects.

Bee stings cause local effects such as swelling, burning and redness hut have not been reported to cause claw hand. We describe two cases in which claw hand developed. In both the condition resolved after conservative treatment.

Case reports

Case one. A 20-year-old woman was stung by a bee on her left index finger while gardening. Within an hour she started to develop clawing of her left hand (Fig. 1) associated with paraesthesiae of the entire hand, which gradually worsened, There were no signs of anaphylactic shock or allergy. She had undergone bilateral carpal tunnel decompression four years before.

On examination there was evidence of the bee sting on the index finger just below the nail. She had painful clawing of the fingers which were fully flexed at the proximal and distal interphalangeal joints and hyperextended at the metacarpophalangeal joints; the thumb was adducted and externally rotated. It was not possible to extend the fingers either passively or actively because of the spasm. The sensation was diminished over all the fingers. There was minimal swelling over the flexor compartment of the forearm which was not tense. At this stage we were unsure as to treatment. We contacted the Poisons Centre and a specialist hand unit, but neither had heard of this particular manifestation of a bee sting and was unable to give any constructive advice.

The clinical findings were such that we did not consider this to be a compartment syndrome and therefore continued to observe the patient treating her with oral anti-inflammatory medication, ann-histamines and prednisolone. Her symptoms improved over 48 hours and the clawing resolved. She was able to extend and flex her fingers actively on discharge. When seen 15 months later she had made a full recovery.

Case two. A 65-year-old woman was stung by a bee in the garden. She developed clawing of the right hand within two hours. When examined the clawing of the fingers was evident and sensation was diminished over all of them. In view of our experience with the first case, we treated this patient also with anti-inflammatory medication, anti-histammes and oral prednisolone. She made a complete recovery and was discharged after 4S hours. When seen nine months later she had normal function m the hand.

Discussion

Bee stings cause local effects such as burning pain, stinging pain, swelling, and redness and may be associated with panic, hysteria, restlessness and anxiety. Severe systemic effects such as swelling of the tongue and throat, difficulty in breathing and shock occur in less than 1% of cases. The symptoms are increased by heat.14

Bee stings have not been reported in the literature to cause claw hand. Bee venom is composed of melittin, iminime, phosphohpase A, phosphohpase B, apamin and acid phosphatase. Melittin, phosphohpase A and phospholipase B cause cell lysis; acid phosphatase and iminime may cause allergic reactions.2,3 Apamin is an extremely potent neurotoxin which acts by blocking Ca^sup ++^-dependent K+ channels in the brain and spinal cord. It is a potent convulsant and also induces motor hyperactivity.2,5 Allergic reactions occur by way of a type-I hypersensitivity reaction mediated by IgH with release of chemical mediators of inflammation.2 Individuals with a high level of tryptase and mastocytosis are susceptible to severe allergic reactions from Hymenoptera venom.1

Ross6 and Goldstein, Rucker and Woltman7 described peripheral neuritis after multiple bee stings and ascribed this to an allergic reaction to the venom. Maltzman et al3 and Chen and Richardson5 reported optic neuropathy after bee stings to the eyelids. Apamin has been implicated in the development of optic neuritis and atrophy.2,3 Other unusual effects such as atrial fibrillation, cerebral infarction, acute myocardial infarction, Fishers syndrome and acute inflammatory polyradiculoneuropathy (the Guillain-Barre syndrome) have been seen.8-12 The mechanism of the claw hand is probably owing to a combination of the central action of apamin on the spinal cord and a peripheral action in the form of median and ulnar neuritis resulting from an allergic reaction to the bee venom.2,5-7 The central effects could explain the spasm of the long flexors in the forearm.

In claw hand from a bee sting the fingers are fully flexed at the proximal and distal interphalangeal joints with hyperextension of the metacarpophalangeal joints. It is not possible to extend the fingers passively because of the intensity of spasm. In a compartment syndrome, in which the fingers are held in flexion at the metacarpophalangeal and proximal interphalangeal joint, it is usually possible to extend the fingers passively to a degree although accompanied by pain in the relevant muscle compartment. Altered sensation over all the fingers after a bee sting is likely to be due to the effect of the toxin on the peripheral nerves, whereas in compartment syndrome, loss of sensation occurs when the increased pressure compresses the peripheral nerve. In the case of a bee sting both the capillary circulation and the radial pulse are intact, but in the later stages of the compartment syndrome, both are comprised. There may be minor swelling over the flexor compartment of the forearm but it is not tense or tender in contrast to the compartment syndrome. If in doubt, the pressure in the compartment of the forearm must be measured.13,14

No benefits in any form have been received or will be received from a commercial party related directly or indirectly to the subject of this article.

References

1. Marshall H. What makes a bee sting deadly. Trends Immunol 2001;22:183

2. Douglas GS, Raymond JR. Corneal bee sting with retained stinger. J Emerg Med 2001;20:125-8.

3. Maltzman JS, Lee AG, Miller NR. Optic neuropathy occurring after bee and wasp sting. Ophthalmology2000;107:193-5.

4. Ewan PC. ABC of allergies venom allergy. BMJ 1998:1365-8.

5. Chen CJ, Richardson CD. Bee sting induced ocular changes. Ann Ophthalmol 1986; 18:285-6.

6. Ross AT. Peripheral neuritis: allergy to honeybee stings. J Allergy 1939;10:382-4.

7. Goldstein NP, Rucker CW, Woltman HW. Neuritis occurring after insect stings. JAMA 1960;173:1727-30

8. Law DA, Beto RJ, Dulaney J, el al. Atrial flutter and fibrillation following bee stings. Am J Cardiol 1997;80:1255.

9. Bachman DS, Paulson GW, Mendell JR. Acute inflammatory polyradiculoneuropathy following hymenoptera stings. JAMA 1982:247:1443-5.

10. Marks HG, Augustyn P, Allen RJ. Fisher's syndrome in children. Pediatrics 1977;60: 726-9.

11. Starr JC, Brasher GW. Wasp sting anaphylaxis with cerebral infarction. Ann Allergy 1977:39:431-3.

12. Jones E, Joy M. Acute myocardial infarction after a wasp sting. Br Heart Jour 1988; 59:506-8

13. McQueen MM, Court-Brown CM. Compartmental monitoring in tibial fractures: the pressure threshold for decompression. J Bone Joint Surg [Br] 1996;78-B: 99-104.

14. McQueen MM, Gaston P, Court-Brown CM. Acute compartment syndrome: who is at risk? J Bone Joint Surg [Br]2000;82-B:200-3.

R. Saravanan, R. King, J. White

From Oldchurch Hospital, Romford, England

R. Saravanan, FRCS, Orthopaedic Specialist Registrar

R. King, FRCS, Orthopaedic Consultant

J. While, FRCS, Orthopaedic Consultant

Oldchurch Hospital NHS

Trust, Romford RM7 0BE,

UK.

Correspondence should be sent to Mr R. Saravanan at 57 Roding Lane North, Woodford Green, Essex IG8 8NR, UK.

©2004 British Editorial Society of Bone and Joint Surgery

doi:10.1302/0301 620X.86B3. 14311 $2.00

J Bone Joint Surg [Br] 2004;86-B:404-5.

Received 10 March 2003;

Accepted after revision 9 July 2003

Copyright British Editorial Society of Bone & Joint Surgery Apr 2004
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