An unusual case of superficial lymphangioma of the right foot

Superficial lymphangioma is a microcytic lymphatic malformation that is usually present already at birth. Women are more commonly affected than men are. It occurs because of the deep lymphatic circulation obstruction. Superficial lymphangioma is most commonly found in the mouth, especially the tongue, on the shoulders, neck, limbs, armpits and groin. It appears as a cluster of small firm blisters filled with clear or hemorrhagic lymph fluid, resembling frogspawn. We are presenting an unusual case of superficial lymphangioma of the right foot in an adult female patient.     

DEPIGMENTATION ALONG LYMPHATIC CHANNELS FOLLOWING INTRALESIONAL CORTICOSTEROID INJECTION

A 39-year-old female with a pruritic verrucous plaque over dorsal aspect of great toe was treated with 4 intralesional corticosteroid injections each at an interval of 2 weeks. Three months later, she developed depigmentation at the injection site and in a network-like distribution radiating away from the site. The depigmentation corresponded to the lymphatic drainage channels of the great toe.     

Hyperkeratotic variant of porokeratosis Mibelli with dermal amyloid deposits

We report a case of hyperkeratotic variant of porokeratosis Mibelli with dermal amyloid deposits. A 66‐year‐old man presented with multiple brownish keratotic lesions on the lower extremities, a verrucous nodule on the third toe of the left foot and brownish verrucous plaques on the buttocks for several years. Histopathological examination of the hyperkeratotic plaque in the right gluteal region revealed extreme hyperkeratosis and cornoid lamella. In the papillary dermis, there were prominent eosinophilic amorphous materials which were positive to Dylon staining. Treatment with oral etretinate resulted in a remission of the skin lesions in this case.     

Acquired progressive lymphangioma occurring following femoral arteriography

Our case report concerns an unusual case of acquired progressive lymphangioma (APL) which developed in a 52-year-old Japanese man following femoral artery catheterization for angiography. The histological examination revealed many irregularly shaped and dilated lymphatic channels that were lined by a single layer of endothelial cells. Immunohistochemical staining for von Willebrand factor was negative in the endothelial cells of the dilated channels. Electron microscopic study demonstrated no Weibel-Palade bodies in the cytoplasm of the endothelial cells. The endothelial cells had no distinct basement membrane.     

Eccrine porocarcinoma with extensive cutaneous metastasis

BACKGROUND: Porocarcinoma is a malignant tumour of the eccrine sweat duct, arising from acrosyringium. The tumoral lesions involve the deep dermal tissue. CASE REPORT: We report the case of an 84 year-old woman, suffering from a porocarcinoma, extensively involving the major part of the left lower limb. The first symptoms appeared two years ago. Hundreds of metastatic papules and small nodules were present, isolated or confluent into large plaques. The clinical picture was very close to lymphangioma. Diagnosis was confirmed by histopathologic examination. Radiotherapy was useful short-term, allowing partial flattening of the lesions and improving lymphatic drainage, thus providing comfort for the patient. It did not prevent a later progression of the tumoral process. DISCUSSION: Porocarcinoma is a rare tumour that usually appears as a single nodule or a plaque, arising from a preexistent eccrine poroma, or developing de novo. Two histopathological variants are described: trabecular or epidermotropic. This latter form, observed in the present case, is more aggressive, leading to frequent local recurrences and/or metastases. Our report is exceptional: the literature shows only one other case with such widespread cutaneous involvement. The clinical course of our case is discussed.     

Verrucous porokeratosis of Mibelli on the buttocks mimicking psoriasis

The typical presentation of porokeratosis of Mibelli is of a solitary plaque with a prominent raised border cleaved by a central furrow. The central portion of the plaque is usually slightly atrophic. The plaques vary in size from a few millimeters to several centimeters in diameter and tend to be acrally distributed, though they can occur on any part of the body. We report an unusual case of verrucous porokeratosis of Mibelli, localized to the natal cleft, that mimicked psoriasis. This entity, though unusual, is not unique. Two similar cases of verrucous porokeratosis of Mibelli limited to the natal cleft region and resembling psoriasis have been reported in the British literature. Verrucous porokeratosis of Mibelli localized to the natal cleft appears to be a distinct clinical entity that can mimic psoriasis. Better recognition of this form of porokeratosis of Mibelli may result in earlier diagnosis and initiation of appropriate therapy.     

Case of dystrophic calcinosis cutis in epidermal cyst arising from verrucous epidermal nevus

Dystrophic calcinosis cutis is diagnosed when calcium is deposited into previously damaged tissue by connective tissue disease, panniculitis, pseudoxanthoma elasticum or trauma. We report a case of dystrophic calcinosis cutis arising from the lesion of an epidermal cyst on the verrucous epidermal nevus. A 20‐year‐old woman presented with a polypoid pinkish tumor on a brownish, verrucous plaque. Histopathological findings of the pinkish tumor showed calcium deposits as amorphous, basophilic material lining the true epidermis in the upper dermis, which were compatible with dystrophic calcinosis cutis and the plaque was diagnosed as a verrucous epidermal nevus.     

"Occult cutaneous lymphangiectasis": an unusual case of cutaneous lymphangioma

An unusual case of cutaneous lymphatic abnormality is described and named as "occult cutaneous lymphangietasis". A 26-year-old man had noticed pigmented maculae in the left inguinofemoral region and waist, which had gradually increased in number for as long as he could remember. The peculiar distribution of the eruptions and their transient saccular dilatation due to infection suggested that they were of lymphatic origin. Lymphangiography showed the presence of dilated lymphatics in the left inguinofemoral area and correspondence of the distribution of dermal backflow with the locations of the pigmented maculae. The histology is consistent with a diagnosis of lymphangioma. We could find no other reports of cases of cutaneous lymphangioma featuring pigmented maculae as the sole manifestation, although whether the pigmentation is an original clinical feature of this type of lymphatic abnormality is still an open question.     

Elephantiasis nostras verrucosa in a patient with systemic sclerosis

Elephantiasis nostras verrucosa (ENV) is an unusual skin condition characterized by dermal fibrosis and hyperkeratotic verrucous lesions resulting from chronic nonfilarial lymphoedema. The condition is similar to ‘elephantiasis tropica’, in which elephantiasis develops secondary to filariasis. Lymphatic obstruction can be primary or due to various causes such as surgery, tumour, radiation, congestive heart failure or obesity. Recurrent attacks of cellulitis lead to further impairment of lymphatic drainage, causing permanent swelling, dermal fibrosis and epidermal thickening. We report a case of a 56‐year‐old man with systemic sclerosis (SS), who presented with painful lesions on both legs, consistent with ENV. He developed extensive, fungating, papillomatous lesions on the skin of the legs, toes and dorsa of the feet over a period of 3 years. Histology revealed dense dermal fibrosis, oedema of the papillary dermis and extensive pseudo‐epitheliomatous changes. To our knowledge, this is the first report of ENV in which SS was considered to be the primary cause for the impairment of lymphatic flow.     

Tumor form of lymphostatic papillomatosis of the skin

The case of a patient with two plantar papillomatous tumours that had had a relapsing course over 10 years is presented. The tumours were located on the right forefoot and the left great toe. Several operations were performed for different diagnoses, i.e. plantar verrucae, verrucous carcinoma and pyoderma vegetans. Histologically, the papillomas proved to be pseudocarcinomatous hyperplasias with multiple ectatic dermal lymph plexuses. Indirect lymphography revealed a circumscribed lymphatic dysplasia with cystic-ectatic cutaneous vessels within the tumour area. Draining lymphatic collectors were conspicuously reduced in number. Isotope lymphography showed a normal lymph transport rate from the foot to the groin. It is concluded that the circumscribed disturbance of lymphatic drainage is the decisive cause of the reactive papillomatous hyperplasia. Thus, these papillomas clinically resembling verrucous carcinoma represent the tumour variant of a papillomatosis cutis lymphostatica.     

Extramammary Paget's disease not only mimicking but also accompanying condyloma acuminatum. A case report

Although its significance remains unknown, it has recently been reported that epidermal papillomatous hyperplasia could be frequently found in extramammary Paget's disease (EMPD). The simultaneous occurrence of EMPD and various human-papillomavirus (HPV)-associated neoplasms has also been reported. We report a case of a perianal EMPD with multiple verrucous papules on its surface. Histologically, the verrucous papules consisted of two distinct histological patterns; one showed numerous Paget's cells within the hyperplastic epidermis with papillomatosis, and the other showed the features compatible with condyloma acuminatum (CA). HPV-6 DNA was demonstrated in koilocytic keratinocytes of the CA by in situ hybridization. No positive signal was obtained for targeted HPV type 6/11/16/18/31/33 DNA in either the verrucous or plaque EMPD. Since the verrucous papules were localized on the plaque of EMPD, our case suggests that some undetermined EMPD-related factors may contribute to the development of epithelial hyperplasia, including HPV-associated neoplasms.     

Extensive annular verrucous late secondary syphilis

We report an illustrative case of an apparently healthy 38-year-old man with a past history of alopecia universalis who developed extensive, slightly pruritic, infiltrated annular verrucous lesions of the scalp, perioral, lumbar, perianal and genital areas over a 6-month period. The combination of an unusual clinical presentation, positive syphilis serology and rapid response to penicillin therapy was consistent with a diagnosis of extensive annular and verrucous late secondary syphilis. We present this case to illustrate a rare and potentially misleading clinical feature of late secondary syphilis, a disease considered to be of the past but still present in today's practice.     

Hyperkeratotic lesion of the nipple revealing cutaneous leiomyoma

BACKGROUND: Leiomyoma of the nipple and areola is a rare benign neoplasm. We report the case of a patient with leiomyoma of the nipple presenting as a hyperkeratotic plaque. OBSERVATION: A 23-year-old patient presented with a five year history of a papillomatous, hyperkeratotic, painful plaque originating in her right nipple. Histological examination of a punch biopsy showed hyperkeratosis of the epidermis with dilatation of the lymphatic vessels within the dermis. Surgical excision revealed a proliferation of smooth muscle fibres, leading to diagnosis of leiomyoma. DISCUSSION: The clinical and histological features were initially consistent with idiopathic naevoid hyperkeratosis of areola. However, associated pain is uncommon in idiopathic lesions. This unusual feature led us to surgical excision enabling the diagnosis of leiomyoma. A hyperkeratotic lesion of the nipple may be associated with benign or malignant neoplasms, hamartoma or chronic dermatoses, or it may be idiopathic. In the present case, the hyperkeratotic lesion revealed subareolar leiomyoma. This is an uncommon clinical presentation not previously seen in medical observations, since leiomyoma usually presents as a firm, painful lump in the subareolar region.