Eosinophilic Cellulitis After Honeybee Sting Hsing-Lin Lin,1,2 Jiun-Nong Lin,3 Chao-Wen Chen,1,2 Liang-Chi Kuo,1,2 Wei-Che Lee1,2*

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CASE REPORT

Stings by honeybees are not uncommon and most cases cause pain but no significant medical problems. Some patients, however, have lethal complications such as acute anaphylactic shock. Cellulitis caused by honeybee sting is very rare and can be a late complication in some patients. We report a 45-year-old female patient who was stung by a honeybee, and whose right forearm showed progressive swelling with bullous for- mation after the sting. She was sent to our emergency department with the diagnosis of right hand cellulitis. After treatment with antibiotics for 5 days, the lesions showed no response. Then, systemic steroid was used and the lesion gradually resolved. Diagnosis of Wells’ syndrome was made according to clinical appearance, course and characteristic histopathological findings. [J Formos Med Assoc 2009;108(12):964–966]

Key Words: bees, cellulitis, eosinophilia, insect bites and stings, Wells’ syndrome

Wells’ syndrome is a rare disease of unknown eti- Case Report ology. This syndrome was described initially by Wells in 1971 as a recurrent granulomatous der- An otherwise healthy 45-year-old woman was matitis with eosinophilia,1 and it was later re- stung by a honeybee on her right dorsal palm named as eosinophilic cellulitis. This cellulitis is while working outside. At first, she only felt se- difficult to differentiate from the bacterial form vere pain. She removed the stinger by herself. and might be treated at first visit with antibiotics, After the sting, she started to feel severe and per- without response. A combination of bacteria can sistent pain over the wound. Then, her right hand decrease the suspicion of the disease. Diagnosis of started to swell progressively. The lesions on the Wells’ syndrome is made by the clinical appear- right hand were painful and intensely itchy, and ance, course and characteristic histopathological they progressed within hours to erythema, ulcer- findings. The relationship between arthropod ation and swelling of the forearm. Two days later, and eosinophilic cellulitis is not very clear. the right forearm began to blister and become We report the case of a patient stung by a erythematous (Figure 1). She was afebrile and had honeybee, which resulted in cellulitis. Histology no systemic symptoms. Laboratory studies were revealed prominent diffuse infiltration of eosino- significant for an elevated white blood cell count phils into the subcutaneous fat. Wells’ syndrome of 12,100 cells/L, with peripheral eosinophilia was diagnosed and the patient received systemic of 43%. The patient was admitted with pre- steroid. The two different diseases must be differ- sumed bacterial cellulitis and was treated with entiated because of the different treatments that intravenous oxacillin for 5 days. After the patient are required. failed to respond to systemically administered

©2009 Elsevier & Formosan Medical Association ...... 1Division of Trauma, Department of Surgery and 2Emergency Medicine, Kaohsiung Medical University Hospital, Kaohsiung Medical University, and 3Division of Infectious Diseases, Department of Internal Medicine, E-Da Hospital/I- Shou University, Kaohsiung, Taiwan.

Received: December 13, 2007 *Correspondence to: Dr Wei-Che Lee, Division of Trauma, Department of Surgery, Revised: April 8, 2008 Kaohsiung Medical University Hospital, 100 Tzyou 1st Road, Kaohsiung 807, Taiwan. Accepted: April 16, 2008 E-mail: [email protected]

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or tender burning sensation, followed by cellulitis- like eruption, well-demarcated nodules or plaques, and the development of vesicles or bullae. The lesions progress rapidly over 2–3 days and re- solve gradually over 2–8 weeks. There are several precipitating factors, including arthropod bites,2 cutaneous viral infections,3 and cutaneous para- sitic infestations.4–6 Some reports also have men- Figure 1. Black ulceration (arrow) as a result of bee sting tioned leukemia,7 myeloproliferative disorders,8 was found on the right dorsal hand, with acute onset of atopic dermatitis,9 fungal infections and drug edematous erythematous plaques and bullae over the 10 right forearm. hypersensitivity reactions. Honeybee stings rarely cause cellulitis. Wells’ cellulitis is found rarely and it can be caused by hyperactivity of the immune system. The manifestations of eosinophilic cellulitis are difficult to differentiate from bacterial cellulitis, and it is easily misdiagnosed. In previous studies, some cases have started with antibiotic treatment, as with our patient. The diagnosis of eosinophilic cellulitis was established after a skin biopsy had been performed. However, before the pathologi- 200 mm cal report, failure of antibiotic treatment, with peripheral blood eosinophilia elevation, remind Figure 2. Hematoxylin and eosin staining of the biopsy the physician to be alert to the diagnosis and from the right forearm showing dermal flame figure char- start treatment with systemic steroid. Patients acterized by a central region of degenerated collagen with bacterial cellulitis can also have fever, and coated by eosinophilic granular material and bordered by a rim of epithelioid histiocytes. the laboratory data can reveal white blood cell shifting to the left and bacteremia. Diagnosis antibiotics, a diagnosis of bullous eosinophilic of Wells’ syndrome is based on typical clinical cellulitis was made by the histological finding of presentations such as single or multiple erythe- prominent diffuse infiltration of eosinophils into matous and edematous urticarial plaques that the subcutaneous tissue (Figure 2). The patient resemble cellulitis, which persist for weeks or received methylprednisolone at 40 mg/day, with months, and heal without scar and the character- subsequent tapering to 20 mg/day for 10 days. istic histological picture of flame figures. Other Complete remission of skin eruption was ob- diseases such as vesiculobullous eczematous der- tained within 3 weeks. She was discharged and matitis, bullous pemphigoid, and other inflam- followed-up at our outpatient department. matory conditions should be differentiated from eosinophilic cellulitis according to pathological findings. Discussion The rarity and clinical manifestations of Wells’ cellulitis render it difficult to differentiate from Wells’ syndrome is an uncommon condition bacterial cellulitis. However, laboratory data can of unknown etiology. The diagnosis of Wells’ help the emergency physician and clinician to syndrome is made according to the clinical ap- make the differential diagnosis between the two pearance, course and characteristic histopatho- diseases before the pathological report, and start logical findings. Lesions can begin with itching appropriate treatment more promptly.

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