Keratoacanthoma centrifugum marginatum: a rare atypical variant of keratoacanthoma

Keratoacanthoma centrifugum marginatum (KCM) is an extremely rare variant of keratoacanthoma (KA), with about 30 cases reported since it was first described in 1962. Clinically, KA is an exoendophytic lesion of 10-25 mm with a horn-filled crater that resolves spontaneously within 6 months. In contrast, KCM is characterized by a larger diameter continuous centrifugal spread, concurrent central atrophy and lack of spontaneous remission. Histologically, KCM is similar to KA, with a central keratin-filled crater, overhanging lips of epithelium, a sharp outline between the tumour nests and stroma, and lack of anaplasia and stroma desmoplasia. We describe a 63-year-old agricultural worker with a 9-month history of a multinodular tumour, 70-75 mm in size, on his right hand. The clinical diagnosis of KCM was confirmed by histological examination. Local radiotherapy proved effective, with no recurrence during a 4-year follow-up.     

Keratoacanthoma centrifugum marginatum arising from a scar after skin injury.

Among the variants of solitary keratoacanthoma, keratoacanthoma centrifugum marginatum (KCM) is characterized by the lack of a tendency toward spontaneous remission and by continuous centrifugal spread. We describe a case of KCM arising from the scar after an old skin injury. The lesion appeared on the dorsum of the right hand, grew peripherally for 30 months, and became a tumor with a multinodular margin and central atrophy. A biopsy specimen from the edge of the tumor showed features resembling typical solitary keratoacanthoma.     

Keratoacanthoma centrifugum marginatum—A rare variant of keratoacanthoma: Case report and literature review

Keratoacanthoma centrifugum marginatum (KCM) is a rare variant of keratoacanthoma and is characterized by progressive peripheral growth with accompanying central healing. Here, we report a case of multiple KCM. A 53-year-old man presented with multiple erythematous papulonodules on both upper limbs and neck for >2 years. His skin lesions enlarged in an annular manner with central residual cribriform scarring that eventually formed confluent plaques (2–8 cm in diameter) with elevated hyperkeratotic borders. Skin biopsy of a developed matured nodule on the right forearm was consistent with that of classical keratoacanthoma. KCM was diagnosed on the basis of clinical and pathological presentation. Low-dose acitretin (0.7 mg/kg/day) was administered and the skin lesions improved significantly within 3 months after the treatment. In this case, we present the clinical and histological features of KCM and discuss the different effective treatment modalities.     

A case of Ferguson-Smith type multiple keratoacanthomas associated with keratoacanthoma centrifugum marginatum: response to oral acitretin

Keratoacanthoma centrifugum marginatum (KCM) is a rare entity, usually classified as solitary keratoacanthoma (KA). The Ferguson-Smith type is the most common form of multiple KAs. Because development of multiple KAs and KCM in a single patient has rarely been reported, this association presents a therapeutic challenge. We report a 46-year-old man with Ferguson-Smith multiple KAs and KCM, who was successfully treated with acitretin.     

Multiple keratoacanthoma centrifugum marginatum

Keratoacanthoma centrifugum marginatum (KCM) is a rare variant of keratoacanthoma characterized by a progressive peripheral growth with concomitant central healing. We report here a case of multiple KCM of the lower legs in a 48-year-old man. The lesions had progressively evolved over 3 years. They were multiple asymptomatic and confluent annular plaques of 5 to 20 cm, having papulo-nodular with hyperkeratotic and crusted borders and cicatricial center. Within the centers were numerous firm and pigmented minipapules of 1 to 2 mm. The typical clinical aspect, together with characteristic histological features confirmed the diagnosis of KCM. Herein we will highlight the clinical and histological features of KCM, as well as the different effective treatments. We will also briefly discuss KCM among the other types of keratoacanthomas.     

Keratoacanthoma centrifugum marginatum after photodynamic therapy with good response to oral retinoids and topical 5‐fluorouracil

Keratoacanthoma centrifugum marginatum (KCM) is a rare variant of keratoacanthoma (KA), characterized by progressive peripheral growth, and usually devoid of deep invasion. Different systemic (oral retinoids) or topical treatments have been reported, but there is not a well‐defined therapeutic protocol. We report the case of a KCM developing after photodynamic therapy (PDT) on the right leg of a 64‐year‐old woman. It was treated successfully with oral acitretin combined with topical 5‐Fluorouracil + salicylic acid for 5 months. This is the first case of KCM developing after PDT and successfully treated with oral retinoid combined with topical treatment.     

Keratoacanthoma centrifugum marginatum associated with mechanical trauma: Response to acitretin—A case report and review of the literature

Keratoacanthoma centrifugum marginatum (KCM) is a very rare variant of keratoacanthoma, characterized with progressive centrifugal growth, central healing, and atrophy. Due to its rarity and lack of distinctive histopathological features, KCM often raises diagnostic and therapeutic challenge. We present a case of a 76‐year‐old Caucasian woman with a single large tumor on her right shin that responded to oral retinoids. The patient presented history of local trauma. The tumor developed over the course of 20 months from a scar. To the best of our knowledge, this is the fifth case of KCM associated with mechanical trauma as a possible triggering factor.     

Efficacy of oral retinoids for keratoacanthoma centrifugum marginatum

Keratoacanthoma centrifugum marginatum (KCM) is a rare variant of keratoacanthoma. This condition is difficult to diagnose because of its large size and expansive nature and may be diagnosed as a malignant tumor. There are various treatments such as surgery and oral retinoids; however, limited studies have verified their effectiveness. Here, we report a case of KCM on the anterior chest of a 50‐year‐old woman and evaluate the efficacy of oral retinoids. In this case, oral retinoids were highly effective for KCM treatment. A total of 55 cases of KCM, including 54 previously reported cases, were reviewed, and their clinical characteristics and treatment were examined. In this report, 14 of 16 patients were effectively treated with oral retinoids, resulting in a treatment rate of 87.5%. Furthermore, even low‐to‐medium doses were sufficient for treatment and prevention. KCM can be misdiagnosed as a malignant disease based on its clinical features. Due to its large size and expansive nature, a wide excision may be performed; however, because oral retinoids have a very high response rate, an accurate diagnosis will help avoid an unnecessary wide excision.     

Immune response to human keratoacanthoma

Immune reactivity to human keratoacanthoma was investigated by microcytotoxicity tests and immunofluorescence. IgM and complement were consistently present in lesions; IgG and fibrin were infrequent. No evidence of in vivo bound immunoglobulin was found on the surface of keratoacanthoma cells by membrane immunofluorescence. Neither patients' sera nor peripheral blood leukocytes showed significant cytotoxicity against autochthonous tumour cells in microtitre assays. This study fails to support the view that regression of human keratoacanthoma is mediated by immunological mechanisms.     

A case of multiple keratoacanthoma centrifugum marginatum: response to oral etretinate

The patient was a 48-year-old man who had a history of colonic polyps. About 18 months before, a dark reddish nodule had appeared on his right forearm. By peripheral growth, this lesion had formed a crateriform prominence with central healing. Similar symptoms multiplied and expanded, the largest being about the size of a child's head. Given both the clinical and histologic evidence, a diagnosis of keratoacanthoma centrifugum marginatum (KCM) was made. Oral etretinate was administered, and an involution of verrucous plaque began after 4 weeks. After 8 weeks, the lesions faded, leaving pigmentation in some areas. This is a rare disorder, only 30 such cases having been reported worldwide, and the present case was the third in which oral etretinate was found to be effective.     

Giant keratoacanthoma arising in hypertrophic lichen planus

A 45-year-old man presented with a rapidly enlarging tumour in an area of long-standing hypertrophic lichen planus of the lower leg. Histological examination of the resected specimen showed it to be a giant keratoacanthoma measuring 37 x 57 mm. Neoplastic change is a rarely reported complication of chronic variants of cutaneous lichen planus. To date there have been only two reports of keratoacanthoma development in association with lichen planus.     

Keratoacanthoma developing in previous cryotherapy site for solar keratosis

Keratoacanthoma is a relatively common benign squamous neoplasm that may show spontaneous clearing. Although the cause of the tumour is unknown, there are some reports describing keratoacanthoma following various types of trauma or secondary to other skin lesions. Thermal burns are reported to be important preceding traumas in the development of keratoacanthomas. whereas there is only one report of cold trauma of cryotherapy. We report a rare case of keratoacanthoma arising in the site of cryotherapy applied for solar keratosis.     

Keratoacanthoma and multiple carcinomas

A case of keratoacanthoma, preceding four primary malignant tumours without metastasis, is presented. Two similar cases from the literature with two and three multiple primary carcinomas, are reviewed. They all have the following findings in common: (1) keratoacanthoma, (2) adenocarcinoma of the colon, (3) additional primary malignant tumour or tumours, and (4) absent or delayed metastasis.     

Keratoacanthoma in a smallpox vaccination site

Tumours arising at the site of smallpox vaccination are rare. A case of keratoacanthoma developing within 2 months at the site of vaccination is described and a likely role of vaecinia virus in the causation of such a tumour is discussed. Complications of varying type and severity may follow smallpox vaccination. Except for encephalitis, these are not officially notifiable diseases, and thus available statistics on their incidence are often un-reliable; however, a primary tumour developing in the site of vaccination is certainly rare. Lane et al. (1970) in 1,648,000 vaccinations against smallpox described one patient who developed melanoma. We describe here a case of keratoacanthoma developing within 2 months at the site of a successful primary vaccination.     

Recurrent distal digital keratoacanthoma of the periungual region treated with Mohs micrographic surgery

We report a 42-year-old man who presented with a recurrent painful nodule on the proximal nail fold of the left fifth finger. The tumour had been surgically excised at another department a month earlier, and histological examination had shown a keratoacanthoma. The patient underwent Mohs micrographic surgery, and no recurrence was evident 16 months later. Mohs micrographic surgery appears to be an effective option in the management of this unusual variant of keratoacanthoma, which is often difficult to eradicate.     

鳞癌及角化棘皮瘤中P53表达的研究

Ｐ５３基因突变是人类癌症中最常见的癌基因之一。采用ＤＯ－１单克隆抗体免疫组化方法检测１３例鳞癌及１３例角化棘皮瘤的Ｐ５３表达。结果６１．５％鳞癌Ｐ５３染色阳性，Ｐ５３阳性细胞广泛分布于肿瘤全层。角化棘皮瘤的Ｐ５３阳性率仅１５．４％，Ｐ５３阳性细胞局限于肿瘤边缘。提示Ｐ５３表达与鳞癌的恶性程度相关。Ｐ５３表达对鉴别鳞癌及角化棘皮瘤有一定意义。     

Conditioned medium from cultured human keratinocytes has growth stimulatory properties on different human cell types.

Evidence for growth-stimulatory properties of keratinocyte-conditioned medium (KCM) on human fibroblasts, endothelial cells, keratinocytes, smooth muscle cells, and a mouse fibroblast cell line (3T3 cells) is presented. On human fibroblasts KCM caused an increase of over 400% in DNA synthesis as revealed by 3H-thymidine incorporation and autoradiography. The proliferative effect was comparable to that of platelet-derived growth factor (PDGF), but was not inhibited by PDGF antibodies and exceeded that of transforming growth factor-alpha (TGF-alpha), epidermal growth factor (EGF), insulin-like growth factor-I (IGF-I), and basic fibroblast growth factor (bFGF). Furthermore, KCM was found to stimulate smooth muscle cells, keratinocytes, and endothelial cells more potently than PDGF, EGF/TGF-alpha, and bFGF, respectively. KCM was also potent in stimulating thymidine incorporation in 3T3 cells, whereas EGF showed a twenty-fold weaker stimulatory effect. Because keratinocytes have been shown to secrete TGF-alpha, which binds to the EGF receptor, binding of factors in KCM to the EGF receptor was assayed. The displacement of radiolabeled EGF by KCM corresponded to a low concentration of EGF (0.5 ng/ml), implying that the growth-stimulatory effect of KCM was not mediated via activation of EGF receptors. Taken together, these results suggest the presence of hitherto unidentified growth-stimulatory factor(s), expressed and secreted by cultured human keratinocytes.     

Spontaneous resolution of keratoacanthoma centrifugum marginatum

Keratoacantnoma centrifugum marginatum (KCM) is a rare variant of keratoacantnoma, with > 40 cases reported world wide. Spontaneous resolution of KCM is very rare. To our knowledge, this is the first case of KCM with spontaneous resolution as documented by serial photographs.     

Giant keratocanthoma on the inner surface of the prepuce.

Gian keratoacanthoma on the inner surface of the prepuce was observed in a 45-year-old man. First symptoms had appeared three months earlier. The tumour was surgically removed. The main histological features of the tumour were enormous, but relatively regular, acanthosis of rete pegs revealing no similarity to the squamous-cell carcinoma, and an exclusively parakeratottic eleidine-containing central plug. The name parakeratoacanthoma is suggested for this type of tumour.     

Ineffectiveness of radiotherapy for the treatment of keratoacanthoma centrifugum marginatum: A case report

Keratoacanthoma centrifugum marginatum (KCM) is a rare variant of keratoacantoma (KA). Although KCM shares histological and clinical features with KA, KCM has no tendency of spontaneous regression, and presents with progressive peripheral expansion with a bank‐shaped outer wall and concurrent central healing. As such, early diagnosis and proper treatment of the patient are required. However, because of its rarity, previous reports are insufficient to evaluate which treatment should be selected. Here, we report a case of KCM that responded to radiotherapy, but relapsed 6 months later.