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PubMedDate not supplied

[Localized scleroderma (morphea) and septic arthritis. Clinical manifestations of Lyme borreliosis seen in El Ferrol].

Two cases of Lyme's disease seen at El Ferrol (Spain) were described. One of them developed a recurrent knee arthritis and the other had a localized sclerodermia (morphea) syndrome. Diagnosis was established by means of clinical picture and serologic tests (enzyme-linked analysis and/or indirect immunofluorescence tests). Joint involvement has often been described in patients diagnosed of having Lyme's disease in Spain, however, the relationship between morphea and borreliosis is still a matter of controversy. We believe that patients with localized sclerodermia and high significant titers of specific antibodies against B. burgdorferi should be treated with antimicrobial agents.

Adult↗
PubMed1984

Localized scleroderma (morphoea): thickness of sclerotic plaques as measured by 15 MHz pulsed ultrasound.

The thickness of morphoea plaques was measured by A-mode ultrasound and compared to regional control measurements in the same individuals. The thickness of morphoea plaques was increased by 18-310% in 17 patients with one or a few morphoea plaques (p less than 0.01), and by 13-145% in 6 patients with generalized morphoea (p less than 0.05). The increase in thickness of morphoea plaques was local confined to the plaques. Ipsilateral and contralateral control measurements were not different, and measurements in a standard region (forearm) were not different from those in a group of healthy controls matched for sex and age. Plaques of clinically 'advanced' scleroderma were more thickened (p less than 0.01) than plaques of 'slight' scleroderma. The relative increase in thickness was larger (p less than 0.01) in skin with a habitual thickness of 0.8-1.1 mm. The habitual skin thickness on the extremities (mean 1.0 mm) was less (p less than 0.01) than on the trunk (mean 1.5 mm), and, consistently, plaques with 'advanced' scleroderma were more frequent (p less than 0.05) on the extremities. Ultrasound measurement of skin thickness was accurate with SD form 0.05-0.09 mm and coefficients of variation from 3-7% in reproducibility studies of typical morphoea plaques as well as normal appearing skin.

Abdomen↗
PubMed1982

Cutaneous and subcutaneous inflammatory sclerosis syndromes.

Systemic scleroderma and localized scleroderma (morphea) show comparable changes on skin biopsy specimens, and a distinction has often been made on the basis of Raynaud's phenomenon, organ involvement, and laboratory abnormalities characteristically seen in systemic scleroderma. Critical evaluation not only of patients with localized scleroderma but also of those with eosinophilic fasciitis, morphea profunda, and acral pansclerotic morphea has disclosed Raynaud's phenomenon, organ involvement, and laboratory abnormalities typical of systemic scleroderma in a small percentage of patients. Histologically, all five conditions show similar inflammation and sclerosis of the skin, the primary difference being the depth at which these changes occur. These conditions may possibly be related, and the clinical and laboratory differences observed may result from variations in depth, nature, and intensity of the cutaneous and subcutaneous inflammatory sclerosis.

Child↗
PubMed2002

Development of histologic features of scleroderma in congenital lesions.

BACKGROUND: Localized scleroderma has been reported after radiation therapy, but has never been reported to occur at the site of a congenital lesion. CASE REPORT: We present two patients, both with family histories of autoimmune disease, who reported unilateral hypopigmented areas on the trunk since birth. The areas remained asymptomatic and grew with the patients until adulthood when the areas became indurated then firm and showed hyperpigmentation. HISTOLOGY: Histologically, both lesions showed features of localized scleroderma with diffuse sclerosis of collagen and loss of periadnexal fat. There was a perivascular lymphoplasmocytic infiltrate with occasional eosinophils extending into the subcutaneous fat predominantly along fat septae, and diffuse loss of CD34+ stromal cell populations within the lesions. CONCLUSION: We propose that somatic mutations affecting vessels may predispose to increased endothelial cell apoptosis. This could lead to the development of an autoimmune response in some individuals, and the areas of localized scleroderma may be markers of susceptibility to autoimmune disease.

Adult↗
PubMed1990

[Detection of pulmonary hypertension in patients with local circumscribed scleroderma].

In three patients with local circumscribed sclerodermia (duration of the disorder 6 months to 3 years), a latent or manifest pulmonary hypertension of the precapillary type in the presence of otherwise unremarkable lung function, radiological and ergometric data, was observed. The pulmonary involvement in this disease would appear to have a relatively early onset.

Biopsy↗
PubMed1999

Expression of type XVI collagen in human skin fibroblasts: enhanced expression in fibrotic skin diseases.

Abundance of type XVI collagen mRNA in normal human dermal fibroblasts explanted from different horizontal layers was determined using RNase protection assays. Type XVI collagen mRNA level in the fibroblasts explanted from the upper dermis was greater than those of the middle and lower dermis. The antibody raised against the synthetic N-terminal noncollagenous region reacted with approximately 210 kDa collagenous polypeptide in the culture medium of fibroblasts. Immunohistochemical study of normal human skin demonstrated that the antibody reacted preferentially with the fibroblasts and the extracellular matrix in the upper dermis rather than those in the middle and lower dermis. Type XVI collagen mRNA level was elevated 2.3-fold in localized scleroderma and 3.6-fold in systemic scleroderma compared with keloid and normal controls. Immunofluorescent study revealed that an intense immunoreactivity with the antibody was observed in the upper to lower dermal matrix and fibroblasts in the skin of systemic scleroderma as compared with normal skin. The results suggest that expression of type XVI collagen, a member of fibril-associated collagens with interrupted triple helices, in human skin fibroblasts can be heterogeneous in the dermal layers and can be modulated by some fibrotic diseases.

Adolescent↗
PubMed1981

Human skin fibroblasts in culture: procollagen synthesis in the presence of sera from normal human subjects and from patients with dermal fibroses.

Various dermal fibrotic conditions, such as progressive systemic sclerosis, localized morphea and familial cutaneous collagenoma, are characterized by excessive deposition of collagen in the skin. In the present study, we examined the possibility that a circulating serum factor(s) is responsible for increased collagen production in these diseases. The effects of human serum on the synthesis of procollagen were examined by incubating normal human dermal fibroblasts with [3H]proline and varying concentrations of dialyzed heat-inactivated serum. The synthesis of procollagen was measured as formation of nondialyzable [3H]hydroxyproline and collagenase-digestible 3H]polypeptides. In the absence of serum little procollagen was formed but the synthesis was markedly stimulated by the addition of normal serum in a concentration-dependent manner. THe ratio of genetically distinct 3H-procollagens of type I and type III, assayed by DEAE-cellulose chromatography and SDS-polyacrylamide gel electrophoresis after limited pepsin proteolysis, was unaffected by the addition of serum. Thus, normal human serum contains a nondialyzable factor(s) which stimulates the synthesis of procollagens type I and type III equally. Sera from 5 patients with progressive systemic sclerosis, 3 with localized scleroderma, and 2 with familial cutaneous collagenoma were also tested. Sera from these patients failed to stimulate 3H-procollagen production more than sera from healthy age-matched controls. Therefore, no increased quantities or qualitatively aberrant factors were shown to be present in the sera of these patients.

Blood Physiological Phenomena↗
PubMed1993

Incidental epidermolytic hyperkeratosis in progressive systemic sclerosis.

Incidental epidermolytic hyperkeratosis (EH) has been reported in a variety of lesions. We describe here incidental EH in a patient with progressive systemic sclerosis (PSS). We reviewed 108 skin biopsies from our cases of PSS and localized scleroderma, but this finding was seen only in the present case. Focal acantholytic dyskeratosis is analogous to EH, but we believe that this case is most consonant with EH. Since there has been no other report of EH in PSS, this association may be coincidental.

Adult↗