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At least 73 records · Page 4Linked to original sources
PubMed1980

Histologic observations of pleomorphic corynebacterium-like microorganisms in diabetic scleredema adultorum.

Diabetic scleredema adultorum is a rare connective tissue disorder characterized by scleroderma-like changes, usually affecting the skin of the neck, shoulders, and upper back in diabetics. Presented herein are two cases in which pleomorphic, but predominantly coccoid forms, were observed in acid-fast, Giemsa, and Gram-stained tissue, sections of the affected skin. Culture of the skin was positive for a microaerophilic, corynebacterium-like organism in one case and an anaerobic Propionibacterium (Corynebacterium) sp in the other case. The morphologic appearance of the bacterial isolates greatly resembled the morphologic forms observed in the tissue sections, suggesting that bacteria, possibly in a cell-wall-deficient phase, may play a role in the pathogenesis of this disorder. Findings of similar bacteria in previously reported cases of other connective tissue disorders such as sclerodermiformis, and rheumatoid arthritis, might support our idea that bacteria could supply the antigenic stimulus for the production of scleredema.

Adult↗
PubMed1997

Systemic involvement in scleredema of Buschke associated with IgG-kappa paraproteinaemia.

Scleredema is a rare primary cutaneous mucinosis. Systemic involvement is uncommon and histological confirmation is often lacking. We report a case of a 60-year-old man with scleredema and evidence of mucin deposition on biopsies from multiple extracutaneous sites. The bone marrow, nerve, hepatic and salivary gland involvement seen on histology in our patient has not, to our knowledge, been previously reported in this condition.

Bone Marrow↗
PubMed1998

[Systemic Buschke's scleredema with cardiomyopathy, monoclonal IgG kappa gammopathy and amyloidosis. Case report with autopsy].

A 73 year old retired truck driver and blacksmith was studied in June 1996 for thoracic pain and was diagnosed as acute pericarditis which responded well to steroid treatment. In January 1997, he noted swelling of the abdominal skin, genitalia and limbs, sparing the feet. He was euthyroid, did not have evidence of diabetes or a Raynaud's phenomenon. His proteinogram showed an IgG-Kappa monoclonal paraprotein M component, 1.31 g/oo. TSH and tetraiodotironine were normal; ESR 16 mm in the first hour. As he did not respond to treatment he was referred to our hospital in March 1997. On physical examination the most relevant findings were a non-pitting edema of the abdomen and lower limbs, sparing the feet. An echocardiogram was consistent with an infiltrative cardiomyopathy. Soon after his hospitalization his condition worsened suddenly with severe bradicardia (28/minute) due to a junctional rhythm and righ bundle branch block. He suffered a cardiac arrest and died. The autopsy findings favoured the diagnosis of systemic scleredema adultorum of Buschke. Amyloid deposits were also found although not abundant, with a similar distribution except in the skin. In this article the clinical and autopsy findings are presented in a patient showing coexistence of systemic Buschke's scleredema with an infiltrative cardiomyopathy, IgG Kappa gammopathy and amyloidosis.

Aged↗
PubMed1994

A case of Hutchinson-Gilford progeria syndrome mimicking scleredema in early infancy.

We report a case of Hutchinson-Gilford progeria syndrome (HGPS). The patient showed the characteristics of scleredema at the age of 2.5 months but developed all the manifestations of HGPS gradually until 10 months old. The possibility of development of HGPS should by considered in any case of scleredema at birth or in early infancy.

Alopecia↗
PubMed1999

[A case report of diabetic scleredema in a patient with non-insulin-dependent diabetes mellitus].

A 70-year-old Japanese man with non-insulin-dependent diabetes mellitus showed scleredema diabeticorum. The patient complained of edematous feelings on his hands, both arms and face. The time of onset of these symptoms was not known. He had typical clinical and histopathological findings of diabetic scleredema. His diabetic control was poor and HbA1c level was 8.2% under insulin treatment. The patient had many complications such as diabetic retinopathy, diabetic nephropathy with benign nephrosclerosis, hyperlipidemia accompanied with a low level of high density lipoprotein cholesterol, hypertension and coronary heart disease. There were no abnormal laboratory findings except the diabetic controls and mild abnormal renal functions; however erythrocyte sedimentation rate was high.

Aged↗
PubMed1998

Scleredema in Chinese patients: a local retrospective study and general review.

The records from 12 Chinese adult patients with scleredema, who had attended the Social Hygiene Service of the Hong Kong Department of Health between 22 January 1990 and 19 March 1996, were retrieved and analysed. The neck was the commonest site of involvement (75%), followed by the back (42%), and the shoulder (17%). The vast majority (83%) of scleredema cases were associated with diabetes mellitus; half of these were insulin-dependent. Most of the patients (92%) had hypertension for which medical treatment was needed. No cases of skin disease were preceded by acute infection, and none had any associated paraproteinaemia. The degree of skin involvement did not affect the daily activities of most of the patients. This study revealed differences between the disease in our locality and those described in the western literature.

Journal Article↗
PubMed2005

Scleredema of Buschke in pediatric age group.

Two cases of scleredema of Buschke are described, which occurred in pediatric age group--an uncommon occurrence after febrile illness. Both cases were self-limiting. Characteristic features are described.

Child↗
PubMed1998

Bath-PUVA therapy in three patients with scleredema adultorum.

BACKGROUND: Scleredema adultorum (SA) is a rare connective tissue disorder for which no treatment has proven to be effective. OBJECTIVE: Our purpose was to determine the effect of bath-PUVA therapy on SA. METHODS: Three patients were treated. Clinical evaluation of skin induration and thickness as well as ultrasonography were performed at baseline and after treatment. RESULTS: All three patients showed substantial clinical improvement with bath-PUVA therapy (median of 59 treatments and a cumulative UVA dose of 245.7 J/cm2). Ultrasonography showed significant reduction in both skin thickness and density. CONCLUSION: Bath-PUVA therapy appears to be effective in the treatment of SA.

Humans↗
PubMed2000

Dosimetric evaluation of a specialized radiotherapy treatment technique for scleredema.

Scleredema adultorum of Buschke is an unusual manifestation of diabetes mellitus that can result in painful indurations and thickening of the skin with associated limitation of motion, and has been previously reported to be responsive to radiotherapy. We report on the method employed to treat a severely affected patient by means of opposed photon fields and multiple electron fields encompassing his entire neck, arms, and thorax.

Humans↗
PubMed2000

Biomechanical measurement of skin distensibility in scleredema of Buschke associated with multiple myeloma.

We report a case of scleredema of Buschke associated with IgG kappa monoclonal hypergammaglobulinaemia. After myeloma polychemotherapy an improvement in skin involvement was observed and confirmed by means of noninvasive skin elasticity measurements. This suggests a relationship between the two diseases. The bioengineering method used can be useful for early detection and monitoring the skin involvement in patients with this disease association.

Biomechanical Phenomena↗
PubMed1997

Diabetic scleredema: a case report and biochemical analysis for glycosaminoglycans.

We report a patient with the typical lesions of diabetic scleredema. Histological findings of the involved skin were thickening of the dermis, depositions of mucins, and fibrosis. Biochemical analysis revealed an increase in glycosaminoglycans in the involved skin as well as in the cutaneous lupus mucinosis. Mucinous materials were composed of hyaluronic acid.

Chondroitin Sulfates↗
PubMed1994

A fatal case of scleredema of Buschke.

Scleredema of Buschke is a rare disorder characterized by the development of areas of skin induration which usually resolve spontaneously. It is occasionally associated with a benign gammopathy, and rarely with myelomatosis. We describe a 60-year-old woman with extensive skin changes, who developed IgA myeloma. Unusually, her skin disease did not respond to conventional myeloma therapy. Death occurred as a consequence of the progressive skin disease.

Esophagus↗
PubMed1989

Scleredema.

We report a classical case of scleredema which pursued a chronic progressive course and was associated with poorly controlled diabetes mellitus.

Collagen↗
PubMedDate not supplied

Tumid lupus erythematosus: an unusual scleredema-like presentation.

A 43-year-old white man presented with an 8-month history of redness and swelling on the back of the neck. He also noted a decrease in range of motion of his upper body. There was no improvement with a 4-week course of topical corticosteroids. Review of systems was negative for polydipsia, polyuria, polyphagia, and bone pain. His medical history included depression, gastroesophageal reflux disease, and microdiscectomy. His medications included sertraline and omeprazole. Physical examination revealed a 20-cm erythematous, indurated plaque on the posterior part of the upper back and neck (Figure 1). A lack of skin wrinkling was noted with lateral pressure. Biopsy revealed a periadnexal and mild interface dermatitis with an increase in connective tissue mucin as demonstrated with colloidal iron (Figures 2A and 2B). Serum protein electrophoresis, hemoglobin A1C, and antinuclear antibody titer were within normal limits. A diagnosis of tumid lupus erythematosus mimicking scleredema was made. Hydroxychloroquine therapy was started at a dose of 200 mg and, at 2 months' follow-up, the patient's symptoms and appearance were improved.

Adult↗
PubMed1990

Acid mucopolysaccharide staining in scleredema.

We studied 26 formalin-fixed biopsy specimens taken from 22 patients with scleredema and found positive staining in 67% of the tissues when both colloidal iron and alcian blue methods were used. Single stains were positive in only half the tissues. Positive findings were noted in both patients with diabetes (11 patients) and in those without it (11 patients). Our experience indicates that multiple biopsies may be necessary to demonstrate mucin in the dermis. Specimens obtained by incisional biopsy are preferred.

Glycosaminoglycans↗
PubMed1983

Buschke's scleredema with right-sided heart failure. Echocardiographic and clinical observations.

Heart failure as a complication of Buschke's scleredema was not reported in the literature on this rare disease. A young girl with a severe form of Buschke's disease is described. She developed severe right-sided heart failure--proven by the typical clinical, auscultatory, electrocardiographic and echocardiographic findings; was treated successfully with penicillin, anticongestive measures and steroids, and showed a remarkable complete recovery.

Child, Preschool↗
PubMed1994

Diabetic scleredema and scleroderma-like changes in a patient with maturity onset type diabetes of young people.

A 21-year-old housewife with maturity onset type diabetes of young people developed scleredema diabeticorum, scleroderma-like skin thickness on forearms and dorsum of hands, digital sclerosis and cheiroarthropathy. She had diabetes mellitus since the age of 11 years. Her grandfather on the mother's side, her mother and 3 of 5 her mother's brothers and sisters have diabetes mellitus. Blood glucose was 295 mg/dl. Urinary glucose was 5.3 g/day. Nail fold capillary microscopy revealed a progressive systemic sclerosis pattern. Histologically, hematoxylin and eosin sections from back and forearm skin demonstrated broad collagen bundles separated by widened clear spaces throughout the thickened dermis.

Adult↗