Sir,
Eosinophilic cellulitis (EC) also known as Wells syndrome is a dermatosis characterized by recurrent urticaria or cellulite-like, erythematous, and edematous plaques. Corticosteroids are the first-line treatment option for treatment. Colchicine has an anti-inflammatory effect and it is effective in the treatment of eosinophilic cellulitis. Here, we present a case of eosinophilic cellulitis that was unresponsive to treatment with systemic prednisolone and resolved with colchicine.
An 18-year-old woman was admitted to our clinic due to a red tender lesion in her right arm for 2 months. There were no constitutional symptoms. The patient had an insect bite history. Systemic antibiotic therapy was initiated with the diagnosis of cellulitis, but there was no regression of lesion after treatment. On the dermatologic examination, an irregularly limited erythematous plaque was observed on the right arm [Figure 1]. Eosinophilia (15.5%) and immunoglobulin E (IgE) elevation (143IU/mL) were detected in laboratory tests. Peripheral blood smear was normal. Histopathologic examination of the lesion showed mixed type cell infiltration rich in eosinophils and occasional flame figures in the dermis and subcutaneous fat tissue [Figure 2]. With these findings, the patient was diagnosed with Wells syndrome. Because of the partial regression of the lesion after 2 week of 40 mg/day prednisolone and topical fusidic acid/betamethasone valerate treatment, 1.5 mg/day colchicine was added and the prednisolone was stopped. At 3 weeks after treatment, the plaque resolved but there was a grayish color change with atrophic appearance in the lesional area [Figure 3]. Colchicine treatment was stopped after 3 months. No recurrence was noted in 6 month follow-ups. In addition, there were no systemic side-effects in the patient due to the use of colchicine.
;)
Figure 1: Irregularly limited erythematous plaque on the right arm
Figure 2: Mixed type cell infiltrations rich in eosinophils and occasional flame figuresin the dermis and subcutaneous fat tissue (H and E, ×100) (marked in black arrows)
Figure 3: Pale atrophic lesion on right arm after treatment
EC usually presents with prodromal symptoms such as pain, burning, and pruritus, followed by one or more erythematous infiltrating plaques that are urticaria-like and tend to expand to the periphery. Pale atrophic lesions occurring during the healing process may be clinically confused with morphea.[12]
The etiopathogenesis of EC is not fully obvious, but some factors are blamed. It has been suggested that insect bites may be a triggering factor in the onset of disease in some patients. Other accused etiological reasons are; hematologic disorders, infections, and malignancies.[34] Insect bites were considered as a triggering factor in our case. Peripheral eosinophilia is common but not necessary for diagnosis.[34] Eosinophilia and IgE elevation were detected in our patient.
Diagnosis is based on clinicopathologic findings. Histological features include dermal edema, lymphocytic infiltrates with numerous eosinophils, eosinophilic structures, and histiocytes located around collagen fibers forming flame figures. This infiltrate can reach the subcutaneous tissue, and rarely even to the muscles.[124]
Systemic and topical corticosteroids are the first-line treatment option in EC. In addition, agents with anti-inflammatory effects, such as antihistamines, hydroxychloroquine, and dapsone, have been used successfully in the treatment of EC.[3]
Colchicine is a neutral and liposoluble tricyclic alkaloid. Colchicine inhibits tubulin polymerization by binding at the colchicine binding site of tubulin, thereby decreasing the expression of adhesion molecules on the membranes and inhibiting neutrophil chemotaxis Moreover, colchicine has also been shown to exert anti-inflammatory activity by preventing the “pore” formation which is necessary for the caspase-1 maturation and IL-1β release in the purinergic P2X receptors.[5] There are three cases in the literature where colchicine treatment is successfully used in EC treatment. In these case reports, the clinical response was attributed to the anti-inflammatory effect of colchicine treatment.[24] There is scarce data about the use and dosage of colchicine in Wells syndrome. Since there was no consensus on this issue, the dose used in Behçet's disease was started.
Our case which did not respond completely to systemic prednisolone treatment was successfully treated with colchicine. We think that colchicine is effective with its anti-inflammatory effect in the treatment of eosinophilic cellulitis. More case reports are needed to confirm whether colchicine treatment is a reproducible treatment option.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
REFERENCES
1. Bansal M, Rai T, Pandey SS. Wells syndrome Indian Dermatol Online J. 2012;3:187–9
2. Puzas ÁI, Álvarez LM, Menéndez ÁF, Romero Yuste S, Gómez OP. Wells’ syndrome successfully treated with colchicine Case Rep Dermatol. 2017;9:65–9
3. Rassler F, Lukacs J, Elsner P. Treatment of eosinophilic cellulitis (Wells syndrome) – A systematic review J Eur Acad Dermatol Venereol. 2016;30:1465–79
4. Paquet P, Laso-Dosal F, de la Brassinne M. Wells’ syndrome: Report of 2 cases Dermatology. 1992;184:139–41
5. An I, Ucmak D, Ibiloglu I, Demir V, Akdeniz S. Colchicine may be of therapeutic benefit in prurigo pigmentosa Pediatr Dermatol. 2018;35:e202–3
