These authors detail the treatment of a 56-year-old male with unusual non-healing ulcerations at previous amputation sites.
A 56 year-old male initially presented to our clinic with an unusual ulceration under the plantar aspect of his right first metatarsal head stump where a hallux amputation had been performed. In fact, both first great toes had been amputated greater than five years ago secondary to osteomyelitis from ulcerations attributed to diabetic neuropathy. Since the amputations had been performed, the patient admits that he had ulcerations over each area. The former ulcerations had subsequently healed and not returned for a few years.
The current lesion on the right first metatarsal head stump had been present for approximately three months after the patient had driven on an extended trip without his diabetic shoe gear. Despite using current offloading modalities with his custom-molded shoes, the ulcer had not healed and he stated that it looked different (see Figure 1) than his previous ulcers.
After debridement of the slightly fibrillated, necrotic and hyperkeratotic lesion, the ulcer measured 1.0 x 1.5cm with a granular, bleeding base. Due to the abnormal characteristics of the lesion, we obtained an initial punch biopsy during debridement and the results demonstrated pre-malignant tissue. We subsequently performed complete wide excision of the lesion in the operating room with determination of clear fresh frozen margins (see Figures 2,3 and 4). A pathological analysis revealed verrucous carcinoma with immunostaining suggesting tumor induction of human papillomavirus. After performing primary closure of the wound, we emphasized non-weightbearing by having the patient wear a fracture walking boot.
Six months after the onset of ulceration in the left foot, we noticed a lesion on the contralateral first metatarsal head during a routine appointment after the patient had stubbed the area on a bedpost. We performed conservative care for the initial blister, which was filled with serosanguineous fluid. Upon follow-up appointments, we addressed an eschar at the site of trauma with local wound care and offloading with a wound shoe. Initial debridement of the eschar revealed fibrotic, hemorrhagic tissue. Despite conservative measures, the lesion was increasing in size, darkening in color and changing in texture (see Figure 5). With the patient’s prevalent history of pedal carcinoma, we did a total excision of the lesion and this turned out to be verrucous carcinoma as well.
In 1925, Buschke and Löwenstein described a slowly progressing neoplasm invading the anogenital area. However, it was not until 1948 when Ackerman coined the term verrucous carcinoma to describe a similar growth within the oral cavity.1,2 Researchers have subsequently isolated the low-grade tumor to other stratified squamous locations in the plantar foot, scalp, trunk and extremities.3 The pedal presentation, which this case report focuses upon, is commonly known as epithelioma cuniculatum, cuniatum plantaris or carcinoma cuniculatum.4 The plantar foot presentation of verrucous carcinoma is named primarily due to its physical and microscopic findings.
Verrucous carcinoma is a derivative of squamous cell carcinoma, the most common malignancy found in humans.3 Squamous cell carcinoma typically evolves from precursors of actinic keratosis or squamous cell carcinoma in situ affecting keratinocytes.3 Although it is connected to squamous cell carcinoma, it is hypothesized that the plantar verrucous carcinoma variants have links to verrucous vulgaris as well due to the detection of human papillomavirus (HPV) types 6, 11, 16, and 18 in specimens.3,5,6,7 Also, histologically epithelioma cuniculatum resembles keratoacanthomas, verrucous vulgaris and epitheliomas.5
Comparably with squamous cell carcinoma, verrucous carcinoma presents with slowly progressive exophytic growths and endophytic crypts or “rabbit burrows.”8,9 Specifically, epithelioma cuniculatum is common among males over the age of 50 and individuals with mental disorders.6,10 Researchers have seen some variants over areas of trauma including burns and scars as Marjolin’s ulcerations.11,12 Other inflammatory and neoplastic reactions can preclude verrucous carcinoma. These include chronic ulcerations, dermatoses, nail bed trauma, venous stasis, pemphigoid, lichen planus and nevi transformations.11,13
Most patients have previously tried and failed topical therapies, thinking that they are treating another pathology. Differential diagnoses include dermatofibromas, seborrheic keratoses, tophaceous gout, pyogenic granulomas, eccrine poromas and amelanotic melanomas. Histologically, differential diagnoses are commonly between keratoacanthoma and verrucous vulgaris.4,13,14
If neglected and in the absence of aggressive intervention, epithelioma cuniculatum can become regionally invasive into deeper soft tissue and osseous structures. There is also a rare chance of metastasis. More frequently, superficial bacterial infections can erupt, causing drainage and malodor within affected areas.4,5,8 Researchers have shown that magnetic resonance imaging (MRI) is the overall preferred pre-operative examination to evaluate lesions of a cryptic and invasive nature but computed tomography (CT) is superior for evaluating cortical disruption.9,10
Different forms of treatment options are available for epithelioma cuniculatum. However, the decision process in management may depend upon the invasiveness of the lesion. The recommended treatment of choice in the literature is wide local excision to incorporate affected adjacent structures and decrease the chance of reoccurrence.3,6,8 Partial or complete amputations are also definitive in cases of significantly invasive lesions, poor vascularity, concomitant infections, large soft tissue defects or reoccurrence.8 Electrocautery, cryotherapy, laser treatment and topical chemotherapy have had higher reoccurrence rates and are known more as adjunctive therapy to surgical treatment.4,6,8,15
In our case, wide excision after fresh frozen sectioning was a definitive treatment for this patient. In follow-up appointments for routine care over a year and a half, there has been no recurrence or affiliated issues (see Figures 6, 7 and 8). We found it rare within the literature to have bilateral presentations of epithelioma cuniculatum. The presentation does correlate with multifactorial influences of trauma including acute injury to the area as well as former ulceration sites. It was unknown if the patient has been exposed in the past to variants of HPV.
Dr. Bornstein is a Fellow of the American College of Foot and Ankle Surgeons, and is in private practice in Winter Park, Fla.
Dr. McDonough is a third-year podiatry resident at Florida Hospital East Orlando in Orlando, Fla.
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