Ultrastructural Aspects of Mucosas in Endemic Pemphigus Foliaceus

Guedes, Antônio Carlos Martins; Rotta, Osmar; Leite, Henrique Vítor; Leite, Virgínia Hora Rios

doi:10.1001/archderm.138.7.949

Cited by 11 publications

(5 citation statements)

References 15 publications

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“…Intercellular widening is defined as 40.5 μm gap between two neighboring cells, which are still in contact. Until recently, this widening was considered to be the first evidence of acantholysis; however, it is also present in basal and suprabasal layers of the skin and mucosa of PF patients where acantholysis is absent (Guedes et al, 2002;Oktarina et al, 2011;van der Wier et al, 2012). Thus, nonjunctional Dsg1 apparently is, in a yet-to-be uncovered way, important for interdesmosomal cohesion as loss is associated with widening.…”

Section: Discussionmentioning

confidence: 99%

Large-Scale Electron Microscopy Maps of Patient Skin and Mucosa Provide Insight into Pathogenesis of Blistering Diseases

Sokol

Kramer

Diercks

et al. 2015

Journal of Investigative Dermatology

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Large-scale electron microscopy ("nanotomy") allows straight forward ultrastructural examination of tissue, cells, organelles, and macromolecules in a single data set. Such data set equals thousands of conventional electron microscopy images and is freely accessible (www.nanotomy.org). The software allows zooming in and out of the image from total overview to nanometer scale resolution in a 'Google Earth' approach. We studied the life-threatening human autoimmune blistering disease pemphigus, using nanotomy. The pathomechanism of cell-cell separation (acantholysis) that underlies the blistering is poorly understood. Ultrastructural examination of pemphigus tissue revealed previously unreported findings: (i) the presence of double-membrane structures between cells in all pemphigus types; (ii) the absence of desmosomes around spontaneous blisters in pemphigus foliaceus (PF); (iii) lower level blistering in PF when force induced; and (iv) intercellular widening at non-acantholytic cell layers. Thus, nanotomy delivers open-source electron microscopic maps of patient tissue, which can be analyzed for additional anomalies from any computer by experts from different fields.

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Section: Discussionmentioning

confidence: 99%

Large-Scale Electron Microscopy Maps of Patient Skin and Mucosa Provide Insight into Pathogenesis of Blistering Diseases

Sokol

Kramer

Diercks

et al. 2015

Journal of Investigative Dermatology

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“…Possible explanations for this finding are either epitope spreading (shift from PF to PV in progression) or ultrastructural changes in the uterine cervix preceding acantholysis. 19 Relevant findings of this study include: vulvar lesions were the second most frequent site of PV mucosal involvement; symptomatic genital involvement (especially vulvar) may be a PF clinical presentation; gynecologic evaluation of patients with pemphigus should be performed; and a careful evaluation of Pap smears in patients with pemphigus is needed to avoid misdiagnosis and potentially drastic clinical consequences (eg, hysterectomy).…”

Section: Discussionmentioning

confidence: 87%

Vulvo-cervico-vaginal manifestations and evaluation of Papanicolaou smears in pemphigus vulgaris and pemphigus foliaceus

Barbosa

Aguiar

Maruta

et al. 2012

Journal of the American Academy of Dermatology

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“…Other authors studied patients with FS and 4 control subjects, examining the oral mucosa using electron microscopy. In addition to showing clinically normal oral mucosa, electron microscopy showed widening of the intercellular spaces between keratinocytes [ 6 – 11 ].…”

Section: Discussionmentioning

confidence: 99%

“…Previous studies have shown that patients affected by EPF in Brazil have some oral findings [ 7 , 8 ]. Selected authors have described the presence of autoantibodies using hematoxylin and eosin (H&E) staining, direct and indirect immunofluorescence (DIF, IIF), and electron microscopy studies [ 9 – 11 ]. In the current study, our aim was to search for oral clinical lesions and an oral autoimmune response in patients affected by EPF in El Bagre, Colombia (El Bagre-EPF) [ 1 – 5 ] and to compare our findings with those described in the medical literature for Brazilian EPF patients.…”

Section: Introductionmentioning

confidence: 99%

Subclinical oral involvement in patients with endemic pemphigus foliaceus

Velez

Howard

Padilla³

et al. 2018

Dermatol Pract Concept

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BackgroundWe have described a variant of endemic pemphigus foliaceus (EPF) in El Bagre area known as pemphigus Abreu-Manu. Our previous study suggested that Colombian EPF seemed to react with various plakin family proteins, such as desmoplakins, envoplakin, periplakin BP230, MYZAP, ARVCF, p0071 as well as desmoglein 1.ObjectivesTo explore whether patients affected by a new variant of endemic pemphigus foliaceus (El Bagre-EPF) demonstrated oral involvement.Materials and MethodsA case-control study was done by searching for oral changes in 45 patients affected by El Bagre-EPF, as well as 45 epidemiologically matched controls from the endemic area matched by demographics, oral hygiene habits, comorbidities, smoking habits, place of residence, age, sex, and work activity. Oral biopsies were taken and evaluated via hematoxylin and eosin staining, direct immunofluorescence, indirect immunofluorescence, confocal microscopy, and immunohistochemistry.ResultsRadicular pieces and loss of teeth were seen in in 43 of the 45 El Bagre-EPF patients and 20 of the 45 controls (P < 0.001) (confidence interval [CI] 98%). Hematoxylin and eosin staining showed 23 of 45 El Bagre-EPF patients had corneal/subcorneal blistering and lymphohistiocytic infiltrates under the basement membrane zone and around the salivary glands, the periodontal ligament, and the neurovascular bundles in all cell junction structures in the oral cavity; these findings were not seen in the controls (P < 0.001) (CI 98%). The direct immunofluorescence, indirect immunofluorescence, confocal microscopy, and microarray staining displayed autoantibodies to the salivary glands, including their serous acini and the excretory duct cell junctions, the periodontal ligament, the neurovascular bundles and their cell junctions, striated muscle and their cell junctions, neuroreceptors, and connective tissue cell junctions. The autoantibodies were polyclonal. IgA autoantibodies were found in neuroreceptors in the glands and were positive in 41 of 45 patients and 3 of 45 controls.ConclusionsPatients affected by El Bagre-EPF have some oral anomalies and an immune response, primarily to cell junctions. The intrinsic oral mucosal immune system, including IgA and secretory IgA, play an important role in this autoimmunity. Our data contradict the hypothesis that pemphigus foliaceus does not affect the oral mucosa due to the desmoglein 1-desmoglein 3 compensation.

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Ultrastructural Aspects of Mucosas in Endemic Pemphigus Foliaceus

Cited by 11 publications

References 15 publications

Large-Scale Electron Microscopy Maps of Patient Skin and Mucosa Provide Insight into Pathogenesis of Blistering Diseases

Large-Scale Electron Microscopy Maps of Patient Skin and Mucosa Provide Insight into Pathogenesis of Blistering Diseases

Vulvo-cervico-vaginal manifestations and evaluation of Papanicolaou smears in pemphigus vulgaris and pemphigus foliaceus

Subclinical oral involvement in patients with endemic pemphigus foliaceus

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