Subclinical oral involvement in patients with endemic pemphigus foliaceus

Background: We 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 a...

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Autores principales:	Ana Maria Abreu-Velez, Michael S. Howard, Hector Jose Lambraño Padilla, Sergio Tobon-Arroyave
Formato:	article
Lenguaje:	EN
Publicado:	Mattioli1885 2018
Materias:	endemic pemphigus foliaceus oral mucosa IgA cell junctions salivary glands secretory immunoglobulin A Dermatology RL1-803
Acceso en línea:	https://doaj.org/article/155b7f547d024ba2860575fb50884e61
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spelling	oai:doaj.org-article:155b7f547d024ba2860575fb50884e612021-11-17T08:29:58ZSubclinical oral involvement in patients with endemic pemphigus foliaceus10.5826/dpc.0804a022160-9381https://doaj.org/article/155b7f547d024ba2860575fb50884e612018-10-01T00:00:00Zhttp://dpcj.org/index.php/dpc/article/view/475https://doaj.org/toc/2160-9381 Background: We 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. Objectives: To explore whether patients affected by a new variant of endemic pemphigus foliaceus (El Bagre-EPF) demonstrated oral involvement. Materials and Methods: A 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. Results: Radicular 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. Conclusions: Patients 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. Ana Maria Abreu-VelezMichael S. HowardHector Jose Lambraño PadillaSergio Tobon-ArroyaveMattioli1885articleendemic pemphigus foliaceusoral mucosaIgAcell junctionssalivary glandssecretory immunoglobulin ADermatologyRL1-803ENDermatology Practical & Conceptual, Vol 8, Iss 4 (2018)
institution	DOAJ
collection	DOAJ
language	EN
topic	endemic pemphigus foliaceus oral mucosa IgA cell junctions salivary glands secretory immunoglobulin A Dermatology RL1-803
spellingShingle	endemic pemphigus foliaceus oral mucosa IgA cell junctions salivary glands secretory immunoglobulin A Dermatology RL1-803 Ana Maria Abreu-Velez Michael S. Howard Hector Jose Lambraño Padilla Sergio Tobon-Arroyave Subclinical oral involvement in patients with endemic pemphigus foliaceus
description	Background: We 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. Objectives: To explore whether patients affected by a new variant of endemic pemphigus foliaceus (El Bagre-EPF) demonstrated oral involvement. Materials and Methods: A 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. Results: Radicular 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. Conclusions: Patients 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.
format	article
author	Ana Maria Abreu-Velez Michael S. Howard Hector Jose Lambraño Padilla Sergio Tobon-Arroyave
author_facet	Ana Maria Abreu-Velez Michael S. Howard Hector Jose Lambraño Padilla Sergio Tobon-Arroyave
author_sort	Ana Maria Abreu-Velez
title	Subclinical oral involvement in patients with endemic pemphigus foliaceus
title_short	Subclinical oral involvement in patients with endemic pemphigus foliaceus
title_full	Subclinical oral involvement in patients with endemic pemphigus foliaceus
title_fullStr	Subclinical oral involvement in patients with endemic pemphigus foliaceus
title_full_unstemmed	Subclinical oral involvement in patients with endemic pemphigus foliaceus
title_sort	subclinical oral involvement in patients with endemic pemphigus foliaceus
publisher	Mattioli1885
publishDate	2018
url	https://doaj.org/article/155b7f547d024ba2860575fb50884e61
work_keys_str_mv	AT anamariaabreuvelez subclinicaloralinvolvementinpatientswithendemicpemphigusfoliaceus AT michaelshoward subclinicaloralinvolvementinpatientswithendemicpemphigusfoliaceus AT hectorjoselambranopadilla subclinicaloralinvolvementinpatientswithendemicpemphigusfoliaceus AT sergiotobonarroyave subclinicaloralinvolvementinpatientswithendemicpemphigusfoliaceus
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Subclinical oral involvement in patients with endemic pemphigus foliaceus

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