Eccrine Syringofibroadenoma Associated with Bowen's Disease: A Case Report and Review of the Literature

Abstract

Eccrine syringofibroadenoma (ESFA) is a rare, benign adnexal neoplasm which usually manifests as a solitary nodule on the extremities of elderly patients. Few case reports have described an association between ESFA and carcinomas including squamous cell carcinoma, porocarcinoma, and basal cell carcinoma. A 66-year-old male presented with a pruritic, erythematous brownish solitary plaque with crusted and verrucous surface on the extensor side of the right thigh. The lesion developed 6 to 7 years ago, and had been growing slowly. Biopsy revealed anastomosing epithelial strands which were composed of 2 areas: the upper area consisting of dysplastic cells with prominent nucleoli and abundant mitoses, and the lower area consisting of oval and round cells, and occasionally small luminal ducts. Dysplastic cells comprised almost the entire epidermis but did not invade into the dermis. Benign syringofibroadenomatous lesion surrounded the dysplastic cells in the lowermost portion of the epidermis. Although it is still unclear whether ESFA undergoes malignant transformation or it is a reactive change to carcinoma, complete excision should be performed to prevent malignant transformation with unknown risk. (Ann Dermatol 32(1) 57∼63, 2020

Erythematous Plaque with Crusting on the Lower Leg: A Quiz

“…ESFA is classified into 5 subtypes: ( i ) solitary ESFA, ( ii ) multiple ESFA without associated cutaneous findings (eccrine syringofibroadenomatosis), ( iii ) multiple ESFA associated with hidrotic ectodermal dysplasia, ( iv ) non-familial unilateral linear ESFA, and ( v ) reactive ESFA. Reactive ESFA is a type of eccrine duct remodelling or repair associated with various inflammatory dermatoses and skin tumours ( 2 ). Several case reports have described reactive ESFA associated with carcinomas, including BD, squamous cell carcinoma, porocarcinoma, and basal cell carcinoma ( 2 , 3 ).…”

“…Reactive ESFA is a type of eccrine duct remodelling or repair associated with various inflammatory dermatoses and skin tumours ( 2 ). Several case reports have described reactive ESFA associated with carcinomas, including BD, squamous cell carcinoma, porocarcinoma, and basal cell carcinoma ( 2 , 3 ). While most non-reactive ESFA cases are identified as benign skin adnexal tumours, its malignant transformation into squamous cell carcinoma or porocarcinoma has been documented previously ( 4 ).…”

Eccrine syringofibroadenoma following venous insufficieny anddiabetic foot ulers: a case report

“…4 There are several neoplasms related to the occurrence of reactive ESFA containing Bowen disease, squamous cell carcinoma (SCC), basal cell carcinoma (BCC), porocarcinoma, and trichoepithelioma. 5 In addition, an association of some inflammatory dermatoses with ESFA has been delineated including eczema, bullous pemphigoid, epidermolysis bullosa, erosive lichen planus, elephantiasis, primary cutaneous amyloidosis, peristomal dermopathy, and chronic ulceration. Reactive ESFA in association with ulceration has also been reported with different etiologies such as diabetes mellitus, traumatic injury, venous insufficiency, burn scar, and infections such as leprosy.…”

“…Reactive ESFA in association with ulceration has also been reported with different etiologies such as diabetes mellitus, traumatic injury, venous insufficiency, burn scar, and infections such as leprosy. [2][3][4][5][6] In the review of the literature, approximately 40 cases of reactive ESFA have been described containing eight patients with a chronic leg ulcer. 4 In accordance with our case, Sirikham et al Reactive ESFA, especially its multiple forms, could cause both functional impairment and cosmetic impact on the patients' quality of life.…”

“…2 However, due to the possibility of malignant transformation of ESFA to eccrine syringofibrocarcinoma and also interrelation with SCC, surgical excision should be considered as a mainstay of therapy, especially in a solitary lesion. 5 Other therapeutic options are ablative modalities such as CO 2 laser, and also radiotherapy in the sites hard to access. 3 All the physicians and dermatologists should be considered the diagnosis of reactive ESFA as one of the differential diagnoses in slow-growing lesions next to chronic ulcers.…”