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At least 19 recordsLinked to original sources
PubMed1991

Transepidermal elimination of thrombi in three cases of thrombotic angiokeratoma: an incidental histopathologic finding of angiokeratoma.

Transepidermal elimination (TEE) is a well known phenomenon by which foreign or altered constituents are removed from the dermis. A thrombosis in the dilated dermal blood vessels of angiokeratoma is not unusual, hence the term thrombotic angiokeratoma. We recently reviewed the histopathologic findings of previously diagnosed cases of angiokeratoma and found that multiple histologic sections of surgical biopsy materials disclosed changes indicating TEE of thrombi in three cases. Special stains searching for an altered substance in those thrombi failed. In this report of three cases, we suggest that this phenomenon could be an incidental and long missed histopathologic finding of angiokeratoma which might explain the fate of these thrombi.

Adult↗
PubMed1992

Angiokeratoma of the clitoris: a subtype of angiokeratoma vulvae.

A 24-year-old married woman had an angiokeratoma of the vulva measuring 3 x 3 x 4 mm localized at the clitoris. The nodule was noticed when she was pregnant with her first child. This tumor grew with occasional tenderness and bleeding during pregnancy, but its size did not change after delivery. The growth of this tumor might be attributable to an increase in venous pressure or serum progesterone levels; therefore this disorder might be classified as a subtype of angiokeratoma of the vulva. This is the first case of angiokeratoma localized at the clitoris.

Adult↗
PubMed1998

[Multiple Fordyce-type angiokeratomas of the scrotum. An iatrogenic case].

BACKGROUND: Angiokeratoma is a wart-like vascular lesion of the skin. There are five types of angiokeratoma: the Mibelli-type, the Fordyce-type, the solitary and multiple (papular) types, the angiokeratoma circumscriptum, and the angiokeratoma corporis diffusum. The "Mibelli-type" occurs on the acral sites, mainly digits, of young people affected by repeated attacks of chilblain, which result in a deleterious effects on vessel walls. The "Fordyce-type" occurs on the scrotal skin of young and adults as a secondary effect to an increased blood pressure in scrotal veins. An equivalent form affecting adult females and occurring analogously on the skin of the vulva is also on record. The "solitary and multiple papular types" of young individuals affect the lower extremities and is considered a consequence of a congenital deficiency of elastic tissue in regional veins. We suggest the term "acquired angiokeratomas" for these three above mentioned clinical forms of angiokeratomas, leaving apart the other two types which are essentially congenital. In fact the "angiokeratoma circumscriptum" is a nevoid hamartomatous lesion arising early in life during infancy or childhood, sometimes in association with other congenital malformation of other sites, while the "angiokeratoma corporis diffusum" almost always occurs in association with enzyme disorders, usually alpha-galactosidase A enzyme deficiency (Anderson-Fabry disease), an X-linked recessive disorder affecting homozigous male patients in their adulthood. CASE REPORT: A case of multiple angiokeratomas of the scrotum (Fordyce-type) arisen in a 62-year old male patient following surgical amputation of the penis and bilateral inguino-crural lymphadenectomy for carcinoma of the penis is reported on. RESULTS AND CONCLUSION: Although they are well on record cases of angiokeratomas of the scrotum arising after surgical injuries to the outer vein pudenda (mainly following inguinocrural hernioplasty), based on a computerized search of the literature on theme this case represents the first iatrogenic example of such an occurrence. The pathogenetic mechanism leading to the the rise of angiokeratomas mediated by the increase of the blood pressure in the superficial scrotal veins is discussed. This example represents an additional case report of inevitable and sometimes unsuspected iatrogenic pathology in medicine.

Amputation, Surgical↗
PubMedDate not supplied

[Intergluteal angiokeratoma circumscriptum. Report of one case].

BACKGROUND: The term angiokeratoma is applied to several diseases with cutaneous vascular lesions. CLINICAL CASE: A 26-year-old male presented with an intergluteal tumor from childhood until the present time, which was treated on several occasions without results. The patient presented to the hospital complaining of a painful and bleeding intergluteal tumor. DISCUSSION: Five types of angiokeratomas have been described (angiokeratoma corporis diffusum, angiokeratoma of Mibelli, angiokeratoma of Fordyce, solitary angiokeratoma and angiokeratoma corporal circumscriptum). This patient was identified with an angiokeratoma circumscriptum by pathological and physical characteristics of the tumor. Differential diagnosis is with verrucous hemangioma and the Fabry disease type II (angiokeratoma corporis diffusum). CONCLUSION: Pathogenesis is unknown and surgery is the recommended treatment.

Adult↗
PubMed1996

Angiokeratomas: an update.

Angiokeratomas are vascular lesions which are defined histologically as one or more dilated blood vessel(s) lying directly subepidermal and showing an epidermal proliferative reaction. At the center of pathogenesis there is a capillary ectasia in the papillary dermis. The epidermal changes in all forms of angiokeratoma are secondary. The different entities causing vessel ectasia lead to the many clinical variants of angiokeratoma. Current classification distinguishes between widespread forms (angiokeratoma corporis diffusum), which is usually associated with an inborn error of metabolism, and localized forms, which include solitary angiokeratoma, Fordyce's angiokeratoma, angiokeratoma circumscriptum naeviforme and angiokeratoma of Mibelli.

Angiokeratoma↗
PubMed1999

Angiomatosis with angiokeratoma-like features in children: a light microscopic and immunophenotypic examination of four cases.

We have identified three patients with an initial clinical or biopsy diagnosis of angiokeratoma, all of whom were found to have a more extensive vascular lesion within the surgical excision. A fourth patient with identical histologic findings had no specified clinical diagnosis and his first procedure was excisional. The patients ranged in age from 7 to 16 years, and the lesions were located on the buttock, thigh, calf, and foot. Macroscopic appearances included mildly keratotic pink-red or blue-grey macules (three cases) and pink macules with focal ulceration (one case). In three of the four cases, a shave biopsy diagnosis of angiokeratoma had been made, and the extensive and deeply infiltrative nature of the vascular proliferation was recognized only at subsequent resection, at which point angiomatosis was diagnosed. In the fourth case. excisional biopsy was attempted at presentation, and the superficial morphology was angiokeratoma-like, but the vascular proliferation was present in the deep subcutaneous fat. CD31 and CD34 reactivity was present in the superficial and deep vessels in all cases, and lesional vessels were rimmed by a bland population of smooth muscle actin positive pericytes, findings that differentiate these cases from angiokeratoma, which has previously been reported to be CD34 negative. We conclude that the dilated vascular spaces that typify angiokeratoma may also be seen overlying a deep vasoformative process that is not amenable to resection, and suggest that caution should be exercised in evaluating small biopsies with angiokeratoma-like appearance.

Actins↗
PubMed1995

Argon laser treatment of cutaneous multiple angiokeratomas.

BACKGROUND: Angiokeratomas are cutaneous vascular lesions with common clinical and histological features: dark red to black papules and ectasia of upper dermis vessels, respectively. Multiple angiokeratomas are difficult to treat because they are often too extensive for surgery and cautery, procedures frequently leading to hemorrhage and scarring. OBJECTIVE: Our report is designed to evaluate the efficacy of argon laser photocoagulation in the treatment of multiple angiokeratomas. METHODS: Two male patients affected by angiokeratoma circumscriptum and angiokeratoma of Fordyce, respectively, were treated using an argon laser under local anesthesia. The laser beam was used as a hemostyptic and to flatten vascular papules by photocoagulation. RESULTS: The cosmetic results of argon laser treatment were good. Posttreatment crusting lasted 2 weeks; 4 months later there was excellent healing without scarring. CONCLUSION: Angiokeratomas respond favorably to argon laser photocoagulation since vascular ectasias are localized in the papillary dermis. Thus, the argon laser offers significant advantages over traditional methods and produces no side effects.

Adult↗
PubMed1995

[Lysosomal storage diseases with angiokeratoma corporis diffusum].

There are 6 well known lysosomal storage diseases which produce angiokeratoma corporis diffusum clinically. The clinical, histological, ultrastructural and biochemical characteristics are discussed. The best known angiokeratoma will be observed in patients with Fabry disease. Angiokeratoma in Fabry disease, however, may be much fewer than thought previously. Fucosidosis and galactosialidosis are next well known diseases to produce angiokeratoma. Approximately 50% of patients with these diseases have angiokeratoma. Recently reported Kanzaki disease, beta-mannosidosis and aspartylglucosaminuria will show angiokeratoma more or less extensively. Ultrastructurally Fabry disease only produce electron dense deposits in lysosomes and others electron lucent. These are summarized in Table 1 in the text.

Endothelium, Vascular↗
PubMedDate not supplied

Angiokeratomas in juvenile dermatomyositis.

Juvenile dermatomyositis (JDM) is an uncommon disease that features muscle weakness, a characteristic rash, and vascular changes in skin, muscle, and other organs. We report a patient with JDM who developed multiple angiokeratomas, one of which was calcified. Electron microscopy of an angiokeratoma revealed altered connective tissue consistent with abnormal collagen. To our knowledge, angiokeratomas occurring in association with JDM have not been previously reported. Abnormal collagen distinguishes the angiokeratomas in our patient from those seen in other conditions. We propose that the angiokeratomas resulted from a progressive compensatory response to the obliterative angiopathy of JDM.

Angiokeratoma↗
PubMed1983

[Angiokeratomas. Apropos of 8 cases].

Eight patients with angiokeratomas are reported; three of them with angiokeratoma circumscriptum present at birth, three angiokeratoma Fordyce type with vulvar location, another one with scrotum location all these appeared by the third decade and the other one a solitary angiokeratoma which age of onset was forty-one years. The clinical and histopathological aspects of the angiokeratoma are studied.

Adult↗
PubMed1993

Angiokeratoma corporis diffusum with glycopeptiduria due to deficient lysosomal alpha-N-acetylgalactosaminidase activity. Clinical, morphologic, and biochemical studies.

BACKGROUND: Angiokeratoma corporis diffusum is a prominent cutaneous feature of certain lysosomal storage diseases. In this article, the clinical, morphologic, and biochemical features of a new, adult-onset lysosomal disease with angiokeratoma are described. OBSERVATIONS: A 46-year-old Japanese woman had diffuse angiokeratoma, mild intellectual impairment, and peripheral neuroaxonal degeneration. The angiokeratoma first appeared on her lower torso when she was 28 years old, and then it became diffusely distributed. Histopathologically, the telangiectasia had localized hyperkeratosis; ultrastructural examination revealed clear cytoplasmic vacuoles in all dermal cells, particularly in vascular and lymphatic endothelial cells and in eccrine sweat gland cells. The lysosomal pathologic features and increased urinary excretion of O-linked glycopeptides suggested the deficiency of a specific glycosidase. Enzyme analyses revealed less than 2% of normal alpha-N-acetylgalactosaminidase activity and the absence of immunodetectable enzyme protein. Her two unaffected children had half-normal alpha-N-acetylgalactosaminidase levels, consistent with the autosomal recessive inheritance of the enzymatic defect. CONCLUSIONS: Since this enzyme deficiency was previously identified in patients with an infantile form of inherited neuroaxonal dystrophy, the occurrence of the enzymopathy in the 46-year-old proband described herein represents an adult-onset form of alpha-N-acetylgalactosaminidase deficiency. This newly recognized entity should be considered in the differential diagnosis of angiokeratoma corporis diffusum.

Fabry Disease↗