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At least 703 records · Page 39Linked 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↗
PubMed1993

Localized bullous pemphigoid: report of a case with an immunofluorescence and electron microscopical studies on the lesional distribution of 180-KD bullous pemphigoid antigen, beta 4 integrin, and type VII collagen.

A 67-year-old woman with a left-sided hemiplegia had localized bullous pemphigoid demonstrating typical clinical lesions on the left pretibial skin and the radial-side skin of the right forearm. The histology showed a subepidermal blister with extensive hyperkeratosis, hypergranulosis, and acanthosis. Direct immunofluorescence revealed distinct linear deposits of IgG and C3 at the dermo-epidermal junction in the perilesional skin and in the roof of the blisters, but few deposits in nonlesional skin. Electron microscopy revealed separation in the lamina lucida. Indirect immunofluorescence of type VII collagen showed its localization in the blister floor. The distribution of the 180-KD bullous pemphigoid antigen (BPA) and beta 4 integrin, hemidesmosomal transmembrane proteins, were studied in the lesional skin by indirect immunofluorescence. Both 180-KD BPA and beta 4 integrin were localized in the blister roof. By immunoelectron microscopy, beta 4 integrin was detected in small groups on the cell surface facing the blister cavity. Since the epitope of the monoclonal antibody to 180-KD BPA used here is known to be localized at a distance of 20 to 50 nm from the membrane surface and this epitope retained in the blister roof, it appears that the blister was produced in the deep lamina lucida. The lesions were cleared with topical 0.05% clobetasole propionate ointment.

Aged↗
PubMed1993

Acral ichthyosiform mucinosis in association with Sjögren's syndrome: a peculiar form of pretibial myxedema?

Two Japanese women developed well-demarcated ichthyosiform plaques on the lateral aspect of their lower legs. Deposition of mucin was demonstrated throughout the papillary dermis, unlike the site of mucin deposition seen in pretibial myxedema. Their thyroid function was normal. The condition of both women was complicated by Sjögren's syndrome. One of them who presented with positive anti-microsomal and anti-thyroglobulin antibodies had goiter, suggesting that her malady was also complicated by Hashimoto's thyroiditis. Their skin manifestations differed from those described in cutaneous mucinosis including pretibial myxedema, specifically with regard to the well-demarcated ichthyosiform appearance, the mucin deposition in the papillary dermis, and the association with Sjögren's syndrome. To the best of our knowledge, our cases may thus be considered to be a previously undescribed form of cutaneous mucinosis associated with Sjögren's syndrome.

Adult↗
PubMed1993

A case of pretibial dystrophic epidermolysis bullosa: decreased expression of the non-helical domain of type VII collagen molecule.

A 27-year-old man with an ataxic gait due to infantile cerebral paralysis exhibited recurrent blistering caused by mechanical stimuli on the pretibial area of both legs from the age of 20. His parents were not consanguineous, and he had no relatives who suffered from blistering. The histology showed a subepidermal bulla due to dermolytic epidermal-dermal separation. The anchoring fibrils were sparse and rudimentary in the predilection area. An LH 7:2 monoclonal antibody against the non-helical domain of the type VII collagen molecule stained the basement membrane zone of the patient's skin at a weaker intensity than the staining of normal human skin, but at a distinctively stronger intensity than the staining of skin from a patient with recessive dystrophic epidermolysis bullosa. Immunoelectron microscopy revealed that LH 7:2-immunoreactants were distributed irregularly within the lamina densa and sparsely in the sublamina densa region. The patient was diagnosed with pretibial dystrophic epidermolysis bullosa.

Adult↗
PubMed1995

A case of pretibial myxedema associated with Graves' disease: an immunohistochemical study of serum-derived hyaluronan-associated protein.

The myxomatous materials in cutis from a patient with pretibial myxedema (PTM) with Graves' disease were found to be mainly hyaluronic acid (HA), which had accumulated extensively in the upper dermis. Fibroblasts were increased in number in the mid to lower dermis. To clarify the mechanism of the deposition of HA in the dermis, we employed an antibody against serum-derived hyaluronan-associated protein (SHAP). This immunohistochemical study disclosed that the greater part of the fibroblasts in the mid to lower dermis stained positively, while the fibroblasts surrounded by fairly large amounts of HA in the upper dermis stained faintly or not at all. From these results, we suspect that the accumulation of HA progresses from the upper to the low dermis and that the interaction of fibroblasts and SHAP leads to development of the physiophathological cutaneous changes seen in our case.

Fibroblasts↗
PubMed1996

Pemphigoid and pemphigus foliaceus successfully treated with topical corticosteroids.

Six patients with pemphigoid and three patients with pemphigus foliaceus were successfully treated with topical corticosteroids. This was especially effective in cases of pretibial localized pemphigoid and in mild cases of bullous pemphigoid and pemphigus foliaceus with negative or low-titer circulating autoantibodies.

Administration, Topical↗
PubMed1996

Inflammatory linear verrucose epidermal nevus coexisting with lichen amyloidosus.

The clinical and histopathological findings in a case of inflammatory linear verrucose epidermal nevus coexisting with lichen amyloidosus are presented. A 33-year-old woman had had linear verrucose lesions which were histopathologically compatible with ILVEN, and with which no amyloid deposits were associated, on her left lower leg for 30 years. She had noticed keratotic papules around the verrucose lesions on the left leg for the last 3 years. Histopathology of these papules revealed amyloid deposits in the upper dermis under the hyperkeratotic and acanthotic epidermis. Topical application of DMSO liniment with dexamethasone successfully treated the ILVEN. This remission of both lesions and itching also alleviated the keratotic papules on the left leg.

Administration, Topical↗
PubMed1997

Hypergammaglobulinemic purpura associated with Sjögren's syndrome and chronic C type hepatitis.

Although hypergammaglobulinemic purpura usually occurs secondarily on the lower legs in several disorders, purpura has also recently been reported to be associated with chronic C type hepatitis (HCV). To define the differences in the clinical, histological and laboratory findings and the prognosis of hypergammaglobulinemic purpura associated with those two disorders, we examined ten patients with hypergammaglobulinemic purpura, 6 patients with hypergammaglobulinemic purpura associated with Sjögren's syndrome (SjS) and 4 patients with hypergammaglobulinemic purpura with chronic C type hepatitis. Five of 6 patients with SjS were female. Attacks of purpura occurred in the pretibial area in all cases. Triggering factors included long walks and prolonged standing. The mean duration of attacks was 6.4 days. No systemic manifestations were associated. Anti-Ro/SS-A and anti-La/SS-B antibodies were expressed in one case. Spontaneous regression was noted in all cases; however, recurrence was noted in one. On the other hand, all the 4 patients with hypergammaglobulinemic purpura associated with HCV were men. Purpura was indurated in a few cases. Involved sites included the knee, forearm, abdomen and thigh in addition to the lower leg. The mean duration of attacks was 12.6 days. Recurrent purpura was noted in one case. Cryoglobulin was positive in three cases. In one patient with severe recurrent purpura, attacks stopped with prednisolone 10 mg/day. Histologically, leukocytoclastic vasculitis was detected in three cases associated with SjS and two cases with HCV. In conclusion, hypergammaglobulinemic purpura associated with HCV appears to occur unilaterally with a sex predilection for men and the manifestations last longer than those associated with SjS. Severe palpable purpura was also noted in association with HCV; systemic prednisolone resulted in good control.

Aged↗
PubMed1998

Cutaneous disseminated actinomycosis in a patient with acute lymphocytic leukemia.

Actinomycosis is an uncommon infectious disease caused predominantly by Actinomyces israelii. The cutaneous disseminated form is usually caused by hematogenous dissemination from a primary extra-cutaneous lesion. We report here cutaneous disseminated actinomycosis without any detectable extra-cutaneous lesions in a 42-year-old Japanese woman with acute lymphocytic leukemia. Multiple soft nodules developed on her upper and lower extremities. Histopathological examination revealed "sulfur granules", which are a specific finding for actinomycosis. Cultures from biopsy specimens were not successful. There were no cervicofacial, thoracic, nor abdominal lesions. These findings suggest that cutaneous disseminated actinomycosis in our patient developed primarily in the skin. Although the patient was immunocompromised, antibiotic treatment with minocycline was effective.

Actinobacillus↗
PubMed1998

Pretibial myxedema with Graves' disease: a case report and review of Japanese literature.

We report a case of pretibial myxedema with Graves' disease in an 18-year-old Japanese woman. The physical examination revealed waxy indurated plaques with prominent hair follicle openings and nonpitting edema disseminated on her lower legs. Histology from an edematous lesion revealed that the dermis was markedly thickened with abundant mucin, especially hyaluronic acid, and the collagen fibers in this portion were splitting up into fibrils. We also reviewed 112 cases of pretibial myxedema reported in the Japanese literature.

Adolescent↗
PubMed1998

Clinical phenotype of Bart's syndrome seen in a family with dominant dystrophic epidermolysis bullosa.

BACKGROUND: Bart's syndrome is one type of dominant dystrophic epidermolysis bullosa (EB). It is known that, in some familial cases of dominant dystrophic EB, the symptoms differ depending on the individual. We observed the way Bart's syndrome affected four generations in the same family. The proband was a newborn boy who showed congenital localized absence of skin (CLAS) and bullae on the anterior aspects of both legs. Histologically, the bullae were located subepidermally. The CLAS and bullae disappeared within 4 months after birth, leaving scars. His father retained scarring and scaling from the knees down along the anterior aspect of the legs, and the nails of the toes were either lacking or deformed. His paternal grandmother and great-grandmother also presented deformed nails of the toes, although they had not had CLAS or bullae on the legs at birth. The individuals in this family thus showed some heterogeneity depending on the sex: blistering and CLAS were seen on the legs soon after birth in the male family members, but the female members did not share this pattern of symptoms, suggesting that the expression of symptoms may differ depending on the sex of the affected individual.

Epidermolysis Bullosa Dystrophica↗
PubMed1998

Necrobiosis lipoidica-like skin lesions in systemic sarcoidosis.

A 62-year-old woman with systemic sarcoidosis developed erythematous plaques on her lower legs. Clinically, two kinds of skin lesions were distinguished; one type formed brownish-red plaques with induration suggesting plaque-type skin sarcoid, and the other formed purplish erythematous plaques with atrophic centers resembling necrobiosis lipoidica. In spite of this clinical appearance, a biopsy specimen from one of the latter lesions revealed typical skin sarcoid histology composed of discrete non-caseating granulomas, while that from one of the other lesions showed necrobiotic changes of collagen bundles surrounded by epitheloid histiocytes and foreign-body giant cells. Because cutaneous involvement of sarcoidosis may mimic necrobiosis lipoidica clinically and/or histologically, we diagnosed her skin lesions as necrobiosis-like skin sarcoid.

Collagen↗
PubMed1998

An unusual panniculitis appearing in the winter with good response to tetracycline.

Cold panniculitis is a form of physical panniculitis due to exposure of skin to severe cold. It usually appears on the cheeks of infants and children. It has also been reported on the thighs and buttocks of young females. Its clinical manifestations include red, cold, indurated plaques or nodules which appear one to three days after exposure to low temperatures and resolve spontaneously within several weeks without scarring. The histopathological picture shows a perivascular infiltrate of lymphoid and histiocytic cells at the dermal-subcutaneous junction in the early phase of the reaction (1). After 48 to 72 hours, a well developed panniculitis appears. We report an unusual case of an adult female patient with recurrent panniculitis on her legs appearing in the winter but without any preceding repeated or prolonged exposure to cold. She responded dramatically to oral tetracycline. This drug was successful as a prophylactic agent as well.

Administration, Oral↗
PubMed2002

Co-existence of variants of porokeratosis: a case report and a review of the literature.

Rarely, different variants of porokeratosis may coexist in an individual patient or their family members. A patient with the linear form of porokeratosis present since birth subsequently developed the disseminated superficial actinic form at a later age. A review of the literature pertaining to the coexistence of variants of porokeratosis suggests a significant association between the linear and disseminated superficial actinic forms. Genetic linkage between different variants and the basis for their association is discussed.

Adult↗