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PubMed1983

Localized scleroderma 'en coup de sabre' and iridopalpebral atrophy at the same line.

A case is reported of scleroderma 'en coup de sabre' in a 13-year-old girl with atrophy of the nasal part of the iris and loss of cilia on the upper eyelid. The lesions of the front and eye were located on precisely the same line. The line did not follow the innervation fields of the cranial, peripheral or autonomous nerves. Nor did it follow the tension lines of the skin (Langer), or underlying anatomical structures. It is discussed that the predisposition to the 'coup de sabre' line seen in localized scleroderma was laid down in the mesenchyme in early foetal life before the differentiation of the anatomical structures.

Atrophy↗
PubMed1990

[Histological study on localized scleroderma].

To define stage-specific and type-specific histological findings in morphea, 21 (14 plaque, 2 linear, and 5 en coup de sabre type) patients were examined. A) Fibrotic changes; 1) Every case had the fibrotic change of various degrees. 2) Nodular sclerotic fibrosis was found in 7 cases with morphea, namely in 5 of 11 cases with a plaque type and 2 of 4 en coup de sabre type morphea. And the above mentioned 7 cases were within two years since the onset, and no nodular fibrosis was found in the morphea with the longer duration than two years in history. Nodular fibrosis was located in the middle or lower dermis adjacent to the typical sclerotic dermis. It was strongly suggested that this nodular fibrosis is as an initial change of localized scleroderma. 3) Nodular fibrosis expanded to the neighboring area and made a typical histological feature of morphea which is completely different from that of diffuse scleroderma. B) Inflammatory findings; 1) Inflammatory changes were divided into, a perivascular, a diffuse or non-perivascular, and a mixed type. 2) The pure perivascular type was found in 10, the pure diffuse type only in one, the mixed type in 6 cases, and the other 4 cases had no inflammation. 3) Marked or moderate inflammation was found in cases with short history of the disease except for one case. 4) Inflammatory cells in the morphea were mainly composed of lymphocytes and histiocytes, but occasionally of plasma cells in 11 of 17 cases. C) Pigmentary changes; Incontinentia pigmenti was found in 18 of 21 cases.

Adolescent↗
PubMedDate not supplied

Circulatory system in children with localized scleroderma.

The circulatory system was studied in 43 children (ages 3-18 years, mean 10.6 years; 32 girls, 11 boys) suffering from localized cutaneous forms of scleroderma. The following studies were undertaken: general pediatric examination, cardiologic examination including routine electrocardiogram (ECG), 24-h Holter ECG monitoring, echocardiography plus Doppler study, and a treadmill exercise test. Three children found to have congenital heart malformations were excluded from the study. Children with localized scleroderma were often lighter and shorter than their appropriate controls. The most common ECG abnormality was incomplete right bundle branch block, but other ECG and Holter abnormalities were found as well. Abnormal echocardiographic results were obtained in 16 cases. The abnormalities concerned valvar function (in all heart valves but predominantly in the mitral valve). It was found that the children suffering from scleroderma had different indices than controls for left ventricular mass index, mitral valve function and left ventricular filling. During the exercise test (conducted according to Bruce's protocol) we found no difference between patients and controls.

Adolescent↗
PubMed1994

Clinical aspects of systemic and localized scleroderma.

Continued efforts are being made to better define the clinical course, disease subsets, and predictors of outcome in scleroderma. Data suggest that the course of the skin disease is triphasic, with the most active thickening phase in the first 12 months of disease. The presence of specific autoantibodies may predict clinical course more precisely than any clinical feature. Antipolymerase I, II, and III antibodies seem specific for scleroderma and, if present, may predict aggressive disease. Early detection of lung involvement provides an opportunity to select patients who may be responsive to drug treatment. Renal crisis in scleroderma is still important and may occur in the absence of significant signs of cutaneous fibrosis. Renin plasma levels do not appear helpful in predicting renal crisis. Significant gastrointestinal reflux disease with delayed acid clearance and esophagitis is associated with aperistalsis of the lower esophagus. Evidence for widespread structural and functional abnormalities of the microvascular circulation have been reemphasized. The psychosocial impact of scleroderma has been studied, demonstrating both the importance of depression and the need for social support. The etiology of localized scleroderma remains unknown despite efforts to link these lesions to Borrelia burgdorferi infection.

Autoantibodies↗
PubMed1997

General and local scleroderma in children and dermatomyositis and associated syndromes.

Scleroderma in children virtually always manifests in the form of localized disease, whereas adult scleroderma usually manifests as systemic disease. The recent literature on childhood scleroderma is limited almost exclusively to localized disease. The treatment of this disease remains a dilemma, especially with regard to which patients should be treated, how they should be treated, and how the treatment response should be monitored. Our current therapeutic approach is reviewed. The recent literature regarding juvenile dermatomyositis is discussed. New understanding of the pathobiology of this disease should lead to improved, less toxic treatments in the future.

Child↗
PubMed1998

[Madecassol treatment of systemic and localized scleroderma].

AIM: The trial of efficacy of 6-month therapy with madecassol (tablets, ointment, powder) of patients with systemic and focal scleroderma (SS and FS). MATERIALS AND METHODS: 54 patients (49 females and 5 males) aged 15 to 70 years with scleroderma running from 3 months to 15 years entered the study. 30 patients had typical SS, 24 patients had FS. Tablets were given to 18 patients, ointment was applied in 42 patients, powder in 3 and tablets + ointment in 9 patients. Madecassol 10 mg tablets were taken 3 times a day by patients with SS and advanced FS. The ointment was preferred in ulcers and scars on fingers and toes in SS and vascular trophic lesions in FS. In active focal scleroderma the ointment was applied to the skin lesions. The ointment was used 2 times a day (in the morning and evening) for 1-6 months. Madecassol powder was employed rarely, primarily of anal and vulval lesions. RESULTS: 6-month oral course (30 mg/day) in 12 SS patients brought about a decrease of indurative lesions, hyperpigmentation (8), vascular trophic disorders (6) and improvement of general condition (5). Subjective response was good in 10 patients and corresponded to absence of progression. In progressive disease and diffuse skin lesions the drug was ineffective. The best response was obtained in local application of madecassol ointment on digital ulcers in SS. CONCLUSION: Madecassol is effective and well tolerated and therefore recommended for oral and local use in combined treatment of SS adn FS. Indications for per os utelization are: chronic or subchronic SS with limited skin involvement, advanced and/or prone to progression FS in which combined administration of the tablets and ointment is proposed.

Adolescent↗
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↗