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PubMed1978

Verrucous lesion in patients with discoid lupus erythematosus. Clinical, histopathological and immunofluorescence studies.

Seven cases of classical discoid lupus erythematosus of the face associated with hyperkeratotic, papulo-nodular lesions on the arms and hands were studied. Clinically the hyperkeratotic lesions resembled keratoacanthomas or hypertrophic lichen planus. The clinical course was marked by chronicity, an absence regression of the lesions, and resistance to treatment. Histopathologically, a lichenoid cellular reaction seemed to play a key role in the development of the hypertrophic lesions which resembled either keratoacanthomoas or hypertrophic lichen planus. Elastic fibres were prominent in the upper dermis, the lower levels of the epidermis and in the hyperkeratotic horny layer. Immunofluorescence of the papulo-nodular lesions demonstrated the deposition of IgG and IgM in a globular pattern at the dermal-epidermal junction. Discontinuous deposition of these antibodies was also seen at the basement membrane zone. On the basis of the immunofluorescence, histopathological and clinical studies, we suggest that the verrucous, hyperkeratotic lesions on the upper extremities represent an unusual, but distinct, form of discoid lupus erythematosus.

Adult↗
PubMed1985

Squamous cell carcinoma of the lip developing in discoid lupus erythematosus.

Since the substitution of steroids and antimalarials for irradiation in the treatment of discoid lupus erythematosus, squamous cell carcinoma arising in discoid lupus erythematosus is thought by some to be an uncommon occurrence. A review of the recent literature (subsequent to 1945) revealed fifteen cases, of which seven occurred in the lips. In one of twelve of the cases a history of irradiation was documented. In three other cases, there was no evidence of its use. We report an additional case of squamous cell carcinoma occurring in the lower lip of a 24-year-old black woman in the absence of radiation therapy. A review of the literature and a discussion of possible predisposing factors are presented.

Adult↗
PubMed1995

Discoid lupus erythematosus lesions developed on lupus erythematosus profundus nodules.

We describe this case of LEP for its unusual way of presentation. It first appeared with a LEP pattern, followed by a typical DLE of the skin, overlying the nodules only. Moreover, our histologic findings showed the typical pattern of lymphocytic lobular panniculitis, with hyaline necrosis of fat, the lymphoid nodule, and even the lymphocytic nuclear "dust." The epidermal changes, with the liquefaction degeneration of the basal cell layer, a moderate follicular hyperkeratosis, and a perivascular and periappendeal lymphocytic infiltrate, were also observed in the abdominal lesion that developed last without clinically evident DLE.

Adult↗
PubMed1997

[Treatment of discoid lupus erythematosus with sulfasalazine: 11 cases].

INTRODUCTION: Antimalaria agents and thalidomide are two reference drugs for discoid lupus erythematosus. In non-responders or after secondary resistance or contraindications, there are a number of alternative therapeutics which are less effective and more toxic. We therefore conducted an open study in patients with discoid lupus erythematosus treated with sulfasalazine. PATIENTS AND METHODS: Seven men and four women (mean age 40 years) with severe discoid lupus erythematosus (mean duration of disease 14 years) were treated with sulfasalazine (2 g/d). This treatment was initiated after a previous failure or contraindication of antimalarial drugs or thalidomide. The acetylation phenotype was predicted in all patients with N-acetyltransferase 2 genotyping. Genome DNA was tested for mutations causing an N-acetyltransferase deficiency. Homozygous individuals or those with heterozygous composites for the tested mutations were predicted slow acetylators and those with a homozygous or heterozygous genotype for an allele carrying a normal sequence at the mutation sites were predicted rapid acetylators. RESULTS: We had 7 complete responses, 1 partial response and 3 failures. Mean delay to efficacy was 7 weeks, longer for lesions involving the scalp (4 to 5 months). Six of the 8 responders were given sulfasalazine exclusively. The effect was suspensive and dose-dependent; the minimal effective dose was 1.5 g/d. Excepting light sensitization requiring discontinuation, there were no clinically significant side effects. Neutropenia occurred in one patient and moderate and transient live enzyme movements did not require treatment withdrawal. The only immunoallergic side effect (light sensitization) observed occurred in a slow acetylator. All responders except one were rapid acetylators. DISCUSSION: Salazosulfapyridine, or sulfasalazine, is composed of a derivative of 5-aminosalicylic acid and a sulfamide fraction, sulfapyridine. It is only marginally used in dermatology except for psoriasis. Its efficacy in chronic lupus erythematosus has been reported in one case. We confirmed the role of this compound in the treatment of chronic lupus erythematosus. The rare observations of induced lupus and development of antinuclear antibodies are not a contraindication, but require close regular clinical and biological surveillance. The potential risk is that possible hypersensitivity could lead to reserving sulfasalazine for severe resistant chronic lupus erythematosus after failure with antimalarials and thalidomide. Nevertheless, our study demonstrates that the slow acetylator phenotype predicts immunoallergic events, as observed by other authors, and would be a factor predicting nonresponse. If these results are confirmed by other studies, it would be possible to propose sulfasalazine as a treatment for discoid lupus erythematosus in rapid acetylators.

Acetylation↗
PubMed1989

[Immunologic and immunogenetic heterogeneity of systemic and discoid lupus erythematosus].

The immune and endocrine systems and HLA genotype were subjected to a comparative study in patients with systemic and discoid lupus erythematosus (SLE, DLE). The patients suffering from these diseases were found to differ in a number of the parameters of the immune status including the content in blood serum and supernatant of the cultivated mononuclear cells of the soluble molecules HLA-A, HLA-B and HLA-DR. The degree of the SLE and DLE association with the genes and haplotypes of class I HLA complex was different as was the character of the association of HLA-A and HLA-B specificities with the activity of the immune system cells and with hydrocortisone content in plasma. The common immunogenetic syndrome characteristic of SLE and DLE patients has been identified.

Adolescent↗
PubMed1992

Scarring alopecia in discoid lupus erythematosus.

The clinicopathological features of the scarring alopecia of discoid lupus erythematosus (DLE) were studied. Scarring alopecia was present in 34% of 89 patients with DLE and was associated with a prolonged disease course. More than half these patients had scalp involvement at the onset of the disease. There was a significant reduction in size of sebaceous glands in affected scalp. Perifollicular lymphocytic inflammation was maximal around the mid-follicle at the level of the sebaceous gland, which seems to be an important functional level in the follicle. There are changes in the expression of the matrix molecules, the proteoglycans, in the connective tissue sheath and the keratin intermediate filaments in the outer root sheath cells at this level in normal scalp and in diseased scalp. Loss of a population of mid-follicular stem cells may be important in the pathogenesis of scarring alopecia in DLE.

Adolescent↗
PubMed1980

Ultrastructural features of oral discoid lupus erythematosus.

Tissue from buccal and labial mucosal lesions of discoid lupus erythematosus (DLE) was studied in the electron microscope. The material comprised 10 patients, 7 of whom presented skin lesions as well. The epithelium showed a change in differentiation pattern from nonkeratinizing to keratinizing epithelium. Civatte bodies (filamentous bodies) were present intercellularly in the basal and suprabasal cell layers, and in the juxta-epithelial lamina propria. Dyskeratotic cells, suggesting immature stages of Civatte bodies, were recorded in the lower stratae of the epithelium. The epithelium-connective tissue interface was abnormal, presenting detachments, gaps and multiplications of the lamina densa. Cytoplasmic tubular structures were observed in epithelial and endothelial cells, as well as in fibroblasts, lymphocytes and macrophages. The oral tissue changes recorded were common in both groups of patients studied, i.e. with and without skin lesions, and similar to earlier reported changes of DLE skin lesions.

Adult↗
PubMed1981

Cytoplasmic tubular structures in the diagnosis of oral discoid lupus erythematosus.

Biopsies from oral lesions of 5 patients with discoid lupus erythematosus (DLE), 5 with lichen planus (LP), 5 with leukoplakia (LEUK) and 3 patients with uncertain diagnoses termed DLE?, LP? were examined in the electron microscope for presence of cytoplasmic tubular structures (CTS) in vascular endothelium. Five vessels were examined in each of two randomly selected sections from each biopsy, without knowledge of the clinical diagnosis. All five DLE biopsies showed presence of CTS in 4-9 out of 10 vessels, as compared with none of 10 vessels in biopsies from LP or LEUK, the difference being significant. Further, CTS occurred in 4 out of 10 vessels in one DLE?, LP? biopsy from a patient with serologic signs of systemic lupus erythematosus. As CTS seem to be absent in oral lesions of LP and LEUK, which are the most important differential diagnoses for oral DLE, the identification of CTS in vascular endothelium sing electron microscopy may be an auxiliary diagnostic procedure in oral DLE.

Adult↗
PubMed1982

A complete selective C1q deficiency in a patient with discoid lupus erythematosus (DLE).

A 32 year old male patient with discoid lupus erythematosus (DLE) was found to have a complete, selective deficiency of C1q subcomponent of the complement assessed either by haemolytic assay or by protein determination. Addition of highly purified C1q completely restored the complement haemolytic activity of the patient's serum. Neither C1q precipitin nor anti-complementary activity was detected. Lymphocytes isolated from the patient's peripheral blood, however, bound as many C1q molecules as those of healthy control individuals. The patient is in good health except for skin lesions. A low level of circulating immune complexes was detected in his serum, but no C1q molecules were bound. Serum complement activities of the patient's mother and two other siblings were within the normal range. An impaired synthesis of C1q protein was strongly suggested as the cause of C1q deficiency in this patient's serum.

Adult↗
PubMedDate not supplied

Discoid lupus erythematosus in children: clinical, histopathologic, and follow-up features in 27 cases.

Among 27 pediatric patients with a clinicopathologic diagnosis of discoid lupus erythematosus (DLE), 15 had localized cutaneous lesions and 12 had disseminated lesions. During a mean follow-up period of 36 months, seven patients (26%) developed systemic lupus erythematosus (SLE). Four of these patients were less than 10 years of age. No correlation was found between localized and disseminated lesions and evolution to SLE. Three of four patients with a positive family history for rheumatoid disease developed SLE (p < 0.05). Hyperpigmentation was significantly more frequent (p < 0.04) in children less than 10 years of age. There was a female predominance of 5:1 among patients less than 10 years of age. Our findings suggest that onset of DLE prior to 10 years of age does not indicate a greater risk of developing SLE. The occurrence of localized or disseminated lesions does not seem to influence the outcome.

Adolescent↗
PubMedDate not supplied

Childhood discoid lupus erythematosus: a Tunisian retrospective study of 16 cases.

Discoid lupus erythematosus (DLE) is uncommon in children. The clinical features of childhood DLE are similar to those of adult DLE in presentation and chronic course. However, children have a particularly high level of transition to systemic disease. We undertook a retrospective study of 16 children with DLE ranging in age from 2 to 15 years, seen over a 9-year period. Six were less than 10 years old at the onset of the disease. The sex ratio was equal. The frequency of childhood DLE was about 7% of the total number of DLE patients seen in our department. Photosensitivity was defined as a clinical history of induction or exacerbation of discoid lesions following sun exposure, and was present in 81% of patients. There was no progression to systemic lupus erythematosus (SLE); an average follow-up time was 10.5 months (2-30 months). We would like to emphasize the increased frequency of childhood DLE in our country and the importance of photosensitivity. However, follow-up data regarding transition to SLE is lacking, therefore we are unable to offer a prognosis to our patients.

Adolescent↗
PubMed1992

Enhanced normal tissue response to radiation in a patient with discoid lupus erythematosus.

We report the case of a patient with discoid lupus erythematosus who developed a severe skin reaction whilst undergoing mantle irradiation for non-Hodgkin's lymphoma. Widespread moist desquamation occurred after a skin dose of only 17 Gy and was associated with an abscopal response outside the treatment area. The case illustrates the need for extreme caution when administering radiotherapy to patients with discoid or systemic lupus erythematosus.

Female↗
PubMedDate not supplied

Immunological studies in patients with discoid lupus erythematosus.

Disorders of serum proteins, circulating antibodies, immune complexes to the dermo-epidermal junction, B- and T-cell markers from peripheral blood lymphocytes and the suppressor function of peripheral mononuclear cells have been investigated in 106 patients with histologically proven discoid lupus erythematosus (discoid LE). The results confirm that this condition is primarily a localized skin disease with low concentrations of autoantigens and circulating autoantibodies. By contrast, systemic lupus erythematosus (systemic LE) is a multifocal, generalized disease with high concentrations of autoantigens, autoantibodies and immune complexes. The transformation of discoid LE to systemic LE is discussed, and a possible two-hit mechanism is proposed.

Antigen-Antibody Complex↗
PubMed1980

Familial discoid lupus erythematosus associated with heterozygote C2 deficiency.

Two siblings with chronic discoid lupus erythematosus and several family members were found with heterozygous C2 deficiency. An association with histocompatibility markers HLA-B18 and HLA-Dw2 was demonstrated, and the slow allotype of factor B was present. Linkage studies in this family suggested a close linkage between the C2 deficiency gene and genes coding for B18, Dw2, and BfS antigens. One HLA-ACB/DBf recombinant was observed showing closer linkage between HLA-D and Bf than between HLA-B and Bf.

Adolescent↗