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PubMed1976

[Diagnostic problems associated with the coincidence of bullous dermatosis and lupus erythematosus visceralis. Demonstration of a case with Duhring's dermatitis herpetiformis in association with visceral lupus erythematosus].

This report describes a patient, whom we observed for 3 years, exhibiting a coincidence of dermatitis herpetiformis Duhring and systemic Lupus erythematosus. The difficulties involved in demarcating bullous lupus erythematosus from the more rare association of bullous pemphigoid or dermatitis herpetiformis with systemic lupus erythematosus are discussed in connection with differential, histological, direct and indirect immunofluorescence diagnostic aids.

Adult↗
PubMed2000

Detection of autoantibodies against tissue transglutaminase in patients with celiac disease and dermatitis herpetiformis.

OBJECTIVE: Endomysial autoantibodies (EmA) are specific for celiac disease. The target antigen has been identified as tissue tranglutaminase (tTG). Our aim was to study the accuracy of a newly developed enzyme-linked immunosorbent assay (ELISA) for easy detection of tTG autoantibodies. METHODS: Thirty-one sera from patients with histologically proven celiac disease and 23 healthy controls were examined for EmA using monkey esophagus and human umbilical cord as substrate. IgA-tTG autoantibodies were determined by newly developed ELISA. Additionally, sera from patients with dermatitis herpetiformis (n = 20), inflammatory bowel disease (IBD; n = 32), chronic liver disease (n = 36), and diabetes mellitus (n = 19) were tested. RESULTS: The sensitivity of the tTG autoantibody ELISA accounted for 90% detection in patients with untreated celiac disease. The specificity was 76% owing to positive values in the lower range in patients with IBD (15%), chronic liver disease (36%), and diabetes (22%), all of whom were negative for EmA. In dermatitis herpetiformis patients 90% were EmA-positive. Of these, only 47% showed elevated tTG autoantibodies. Preincubation of sera from dermatitis patients with tTG abolished immunofluorescent staining of endomysial structures. CONCLUSION: Detection of mid- to high-titer tTG autoantibodies is highly specific for celiac disease. However, in the low-titer range, overlap exists with liver disease, IBD, and diabetes. Tissue transglutaminase autoantibodies may evolve as a new screening and follow-up method for celiac disease. Although tTG seems to be a major autoantigen in dermatitis herpetiformis, the low sensitivity of both tTG ELISA and immunofluorescence using human umbilical cord suggests differential involvement of tTG in this disease.

Animals↗
PubMed1993

[The Köbner phenomenon in Duhring dermatitis herpetiformis].

We report on four female patients with confirmed dermatitis herpetiformis DH and Köbner phenomenon in identical localizations. All four had typical bilateral lesions in a band-like arrangement along the straps of their brassieres, and two patients also had additional lesions in other sites prone to pressure. Obviously, the Köbner phenomenon is not as rare in DH as would be expected in view of a single previous case report.

Adult↗
PubMed1984

Vesicular pemphigoid vs dermatitis herpetiformis.

We describe a patient with clinical features of dermatitis herpetiformis (DH) and histopathologic features suggestive of both DH and bullous pemphigoid (BP). Immunofluorescent (IF) studies of skin biopsy revealed IgG and C3 deposits along the basement membrane zone (BMZ) in a linear pattern and circulating BMZ antibodies in the serum consistent with BP. The patient's condition evolved over 5 years into a typical case of DH characterized by histologic findings of papillary edema and neutrophilic infiltrates and evidence by IF findings of granular IgA deposits in the dermal papillae and the presence of antigliadin antibodies in the serum.

Adult↗
PubMed1978

Dermatoglyphics in dermatitis herpetiformis.

In a dermatoglyphic study of 101 patients with dermatitis herpetiformis, comparison was made with their normal relatives and several random normal series. The results of the several comparisons are quite consistent, considering the nature of the data, for the all point to an attenuation of qualitative and quantitative digital and palmar traits in the patients. It is argued that these differences are associated with the disease itself, and that some of the female relatives have an inherited tendency to the disorder, but do not express it. The findings suggest the involvement of genetic factors in the aetiology, and possibly intrauterine environmental influences as well.

Dermatitis Herpetiformis↗
PubMed1993

Major histocompatibility complex susceptibility genes for dermatitis herpetiformis compared with those for gluten-sensitive enteropathy.

Dermatitis herpetiformis (DH) shares some clinical features and major histocompatibility complex (MHC) markers with gluten-sensitive enteropathy (GSE). We compared MHC haplotypes in 27 patients with DH, 35 patients with GSE, and normal controls. As in GSE, the frequencies of two extended haplotypes, [HLA-B8, SC01, DR3] and [HLA-B44, FC31, DR7], were increased in patients with DH. Distributions of fragments of extended haplotypes, consisting of some but not all of the elements of complete extended haplotypes, were analyzed to attempt to localize a susceptibility gene. Besides complete extended susceptibility haplotypes, (DR3, DQ2) and (DR7, DQ2) fragments were most common in GSE. In contrast, DH showed only a few such fragments but many instances of the fragment (SC01). The differences in distribution of these fragments in the two diseases were highly significant (P < 0.002). HLA-DQ2 and DR3 had the highest odds ratios for GSE, but the highest odds ratio for DH was for the complotype SC01. These findings suggest that the MHC susceptibility gene for DH is between class II and complotype regions, closest to the complotype, whereas that for GSE is in the class II region.

Celiac Disease↗
PubMed2004

A new model for dermatitis herpetiformis that uses HLA-DQ8 transgenic NOD mice.

Dermatitis herpetiformis (DH) is an autoimmune blistering skin disorder that is associated with gluten sensitivity. It presents as a papulovesicular rash and is often associated with enteropathy. The rash resolves when the patient is placed on a gluten-free diet and/or dapsone. DH, as well as celiac disease, is tightly associated with DQ2 and DQ8. A novel mouse model for DH is described that utilizes the NOD background and the HLA-DQ8 transgene. The addition of DQ8 contributes sensitivity to gliadin, and the addition of the NOD background contributes to autoimmunity and pathogenesis. Fifteen NOD DQ8+ mice of 90 that were sensitized to gluten developed blistering pathology similar to that seen in DH. Neutrophil infiltration of the dermis, deposition of IgA at the dermal-epidermal junction, and a complete reversal of the blistering phenomenon with the administration of a gluten-free diet with or without dapsone were observed. None of the 3 blistering mice examined had small-bowel pathology. This animal model of DH will be useful to determine the specificity of the IgA deposits, as well as the pathogenic mechanisms that occur in the skin as a result of gluten ingestion.

Animals↗
PubMed1991

IgA-binding structures in dermatitis herpetiformis skin are independent of elastic-microfibrillar bundles.

Dermatitis herpetiformis (DH) is characterized in part by the presence of granular deposits of IgA in the papillary dermis just beneath the dermal-epidermal junction. The nature of the structures to which IgA binds in DH skin, however, has not been clearly demonstrated. Previous immunoelectron-microscopy studies using the peroxidase-antiperoxidase technique have concluded that the IgA may bind to abnormal elastic microfibrillar bundles. Recently, antibodies have been developed against a major component of the elastic microfibril bundles, fibrillin. In addition, another dermal matrix protein, hexabrachion, has been characterized and found in normal human skin in a distribution similar to the IgA deposits of DH. Utilizing antibodies against fibrillin, hexabrachion, and human IgA and immunoelectronmicroscopy with immunogold staining techniques, we have examined the skin from patients with DH in order to localize the IgA deposits. Normal-appearing skin from five patients with DH exhibited electron-dense patches within the dermis, which were not seen in skin from normal subjects. These structures were sometimes adjacent to the basement membrane zone, but appeared amorphous and without a well-defined fibrillar structure. The electron-dense patches were labeled with anti-human IgA, but not with antibodies to fibrillin or hexabrachion. The anti-IgA antibody did not label the normal basement membrane. These studies confirm the presence of abnormal electron-dense, amorphous structures in the skin of patients with DH. Due to this lack of association with the elastic microfibril bundles and the lack of labeling with antibodies against fibrillin, we suggest that these deposits are distinct from the microfibrillar bundles of elastic tissue and may represent IgA bound to degraded basement membrane or isolated dermal deposits of IgA.

Actin Cytoskeleton↗
PubMed2002

Neutrophil CD11b, L-selectin and Fc IgA receptors in patients with dermatitis herpetiformis.

BACKGROUND: The skin lesions found in patients with dermatitis herpetiformis (DH) are characterized by the presence of neutrophils at the dermal papillary tips in areas where the diagnostic cutaneous IgA deposits are found. Although the presence of the skin lesions of DH is known to be associated with gluten-sensitive enteropathy, the mechanisms that control the development of skin lesions are not known. OBJECTIVES: To determine if circulating neutrophils from patients with DH have evidence of priming as shown by increased expression of CD11b, decreased expression of L-selectin and increased function of neutrophil Fc IgA receptor. METHODS: Neutrophils from 12 normal subjects and 10 DH patients with active, ongoing disease and 14 DH patients with quiescent disease activity were examined by fluorescence-activated cell sorter for expression of cell surface CD11b, L-selectin expression, Fc IgA expression (CD89) and for the function of the Fc IgA receptor by determining the binding capacity of neutrophils for monoclonal human IgA. RESULTS: Neutrophils from patients with active, ongoing DH had increased expression of CD11b when compared with patients with inactive DH or normal subjects [mean net geometric mean channel fluorescence (GMCF): active DH, 403.3; inactive DH, 237.8; normal subjects, 290.5; P < 0.05]. L-selectin expression in both groups of DH patients was significantly lower than that seen in normal subjects (mean net GMCF: active DH, 363.2; inactive DH, 375.2; normal subjects, 432.7; P < 0.05). No difference in CD89 expression was seen in any of the groups; however, the function of Fc IgA receptor was increased in patients with active DH when compared with patients with inactive DH and normal subjects. CONCLUSIONS: Neutrophils from patients with active, ongoing DH show an increased expression of CD11b, decreased expression of L-selectin and increased ability to bind IgA, consistent with a pattern of priming of the neutrophils. This priming may occur in the gut as a result of the ongoing mucosal immune response that is present in patients with DH on a gluten-containing diet and may predispose neutrophils to localize in the skin of patients with DH.

Adult↗
PubMedDate not supplied

IgA dermatosis in an adult Chinese population. A 10-year study of linear IgA and dermatitis herpetiformis in Singapore.

During a 10-year period (1976-1985), eight Chinese patients from a population of 2.5 million fulfilled the criteria for dermatitis herpetiformis and linear IgA bullous dermatosis (LABD). These rare diseases among Asians presented during the third decade of life with homogenous linear IgA deposits along the dermoepidermal junction in four of the eight cases. Three had classic granular deposits in the papillary dermis, while one was negative. There has been a remission in two of the patients. Dermatitis herpetiformis among the Chinese differs from experience elsewhere in that there was no associated gluten-sensitive enteropathy. The response to a very low-dose dapsone was excellent in most cases.

Adult↗
PubMed1985

[Childhood benign chronic bullous dermatosis. Differential diagnosis with dermatitis herpetiformis and bullous pemphigoid].

We review the literature on benign chronic bullous dermatosis of childhood and the criterions for its diagnosis and differentiation from dermatitis herpetiformis and bullous pemphigoid. The diagnosis among these conditions should be based on immunofluorescence data. BCBDC shows linear IgA on the basal membrane zone. Its course is benign and there is not enteropathy. In dermatitis herpetiformis we find granular IgA and complement in the tips of dermal papillae. It is a chronic illness and the patients have a gluten-sensitive enteropathy. The bullous pemphigoid is a severe condition which needs corticosteroids for its control. Direct immunofluorescent studies reveal IgG and complement in a linear pattern in the membrane basal zone. It is associated with circulating antibasement membrane zone IgG.

Child↗
PubMed1983

Pharmacokinetic observations on dapsone in dermatitis herpetiformis.

The pharmacokinetics of dapsone (DDS) and monoacetyldapsone (MADDS) following an oral dose of 150 mg DDS were studied in sixteen patients with dermatitis herpetiformis and seven normal subjects. No differences in DDS disposition were observed between the two groups. The maintenance dose of DDS for individual patients was not significantly correlated with jejunal biopsy morphology, DDS or MADDS half-lives, or the area under the plasma concentration-time curves for DDS or MADDS. DDS plasma protein binding was normal in patients and did not apparently determine the concentration of DDS in skin biopsies, for which the skin/plasma DDS concentration ratio was approximately unity. There was no undue representation of acetylator phenotype in the patient group and no correlation between maintenance dose and MADDS/DDS ratio was noted. The determinants of the maintenance DDS dose have not been found. This may relate to pharmacodynamic differences, but alternatively the concentration of oxidative metabolites rather than DDS or MADDS could be responsible for the therapeutic activity in dermatitis herpetiformis.

Adult↗
PubMed1989

Bullous pemphigoid and dermatitis herpetiformis: mixed bullous disease or coexistence of two separate entities?

We present a 73-year-old man with a 5-year history of dermatitis herpetiformis who developed lesions with the clinical, histologic, and immunologic features of bullous pemphigoid. Direct immunofluorescence testing of a skin biopsy demonstrated both granular deposition of IgA, predominantly in the papillary bodies, and linear deposition of IgG and C3 at the basement membrane zone. This mixed direct immunofluorescence pattern, typical for dermatitis herpetiformis in the type of IgA deposits, but also typical for pemphigoid in the linear localization of IgG and C3, is unusual. This case emphasizes that even after a specific diagnosis has been established, if the clinical morphology or response to therapy changes, repeat histologic and immunofluorescence studies may be indicated in diagnosis and management of patients with bullous disease.

Aged↗
PubMed1990

Patients with dermatitis herpetiformis, acne, psoriasis and Darier's disease have low epidermal zinc concentrations.

Zinc concentration was determined in epidermis, papillary dermis and serum in patients with dermatitis herpetiformis, acne or psoriasis and in two small groups of patients with ichthyosis vulgaris and Darier's disease. Except in ichthyosis vulgaris the zinc level in epidermis was decreased in all these disorders. The mean serum zinc concentration was, however, significantly decreased only in men with dermatitis herpetiformis. There was no correlation between the concentration of zinc in epidermis or dermis and that in serum. The decreased epidermal zinc concentration indicates that many of the patients have a zinc deficiency in spite of a "normal" serum zinc value. Supplementation of zinc might therefore be of value in patients with these disorders.

Acne Vulgaris↗
PubMed1986

IgA endomysium antibody in children with dermatitis herpetiformis treated with gluten-free diet.

The IgA antibody to endomysium of smooth muscle (IgA-EmA) was measured in 32 children with confirmed dermatitis herpetiformis who were eating gluten-free diets. One patient had IgA-EmA before treatment but had a negative test one month later while on the diet. Two so treated for less than one year still had antibody, but of seven children treated for more than one year with gluten-free diet, none had detectable antibody. It was present in 13 of 20 children not adhering to the prescribed diet. The antibody was absent in 4 children with linear IgA bullous dermatosis and 43 children with various skin and intestinal diseases. These findings correspond to those in adults with dermatitis herpetiformis and indicate that IgA-EmA is also a marker for gluten-sensitive enteropathy in children.

Child↗
PubMed1985

IgA anti-endomysial antibody detection in the serum of patients with dermatitis herpetiformis following gluten challenge.

This study reports the appearance of IgA-class anti-endomysial antibodies in the serum of 8 out of 12 patients with dermatitis herpetiformis who were challenged with gluten after a number of years of control of the rash with a strict gluten-free diet. Although there was no evidence for the antibodies having any pathogenic role in the rash of dermatitis herpetiformis, their presence may be related to the deterioration in the gluten-sensitive enteropathy.

Adult↗