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Chronic ulcerative stomatitis: A comprehensive review and proposal for diagnostic criteria.
Oral Dis. 2019 Sep; 25(6):1465-1491.OD

Abstract

Chronic ulcerative stomatitis (CUS) is an immune-mediated disorder characterized by oral erosions and ulcers usually refractory to conventional treatments. The disease often involves middle-aged and older women with painful lesions sometimes resembling those of erosive oral lichen planus (OLP). The most affected sites are the buccal mucosa, the gingiva and the tongue, but the skin is involved in 22.5% of cases. Histopathologic features in CUS are non-specific and indistinguishable from those of OLP, with the exception of the presence of a mixed infiltrate composed of lymphocytes and plasma cells. Direct immunofluorescence (DIF) analysis reveals the presence of stratified epithelium-specific antinuclear antibodies (SES-ANA) in the lower third of the epithelium. The IgG antibodies detected on DIF are directed against the ∆Np63α isoform of p63 expressed in the nuclei of the epithelial basal cells. A distinguishing feature of CUS is the low response to conventional corticosteroid therapy and the good outcome with hydroxychloroquine at the dosage of 200 mg/day or higher dosages. This paper presents a comprehensive review of CUS and is accompanied by a new case report (the 73rd case) and a proposal for updated diagnostic criteria.

Authors+Show Affiliations

Unit of Oral Pathology, Dental Clinic, ASST Sette Laghi, Ospedale di Circolo, Department of Medicine and Surgery, University of Insubria, Varese, Italy.Unit of Anatomical Pathology, ASST Sette Laghi, Ospedale di Circolo, Varese, Italy.Unit of Dermatological Diseases, ASST Sette Laghi, Ospedale di Circolo, Varese, Italy.Unit of Anatomical Pathology, ASST Sette Laghi, Ospedale di Circolo, Varese, Italy.Unit of Oral Pathology, Dental Clinic, ASST Sette Laghi, Ospedale di Circolo, Department of Medicine and Surgery, University of Insubria, Varese, Italy.Unit of Oral Pathology, Dental Clinic, ASST Sette Laghi, Ospedale di Circolo, Department of Medicine and Surgery, University of Insubria, Varese, Italy.Unit of Oral Pathology, Dental Clinic, ASST Sette Laghi, Ospedale di Circolo, Department of Medicine and Surgery, University of Insubria, Varese, Italy.Unit of Oral Pathology, Dental Clinic, ASST Sette Laghi, Ospedale di Circolo, Department of Medicine and Surgery, University of Insubria, Varese, Italy.Department of Dermatology, Medical University of Warsaw, Warsaw, Poland.

Pub Type(s)

Journal Article
Systematic Review

Language

eng

PubMed ID

30457193

Citation

Azzi, Lorenzo, et al. "Chronic Ulcerative Stomatitis: a Comprehensive Review and Proposal for Diagnostic Criteria." Oral Diseases, vol. 25, no. 6, 2019, pp. 1465-1491.
Azzi L, Cerati M, Lombardo M, et al. Chronic ulcerative stomatitis: A comprehensive review and proposal for diagnostic criteria. Oral Dis. 2019;25(6):1465-1491.
Azzi, L., Cerati, M., Lombardo, M., Pellilli, M., Croveri, F., Maurino, V., Tagliabue, A., Tettamanti, L., & Olszewska, M. (2019). Chronic ulcerative stomatitis: A comprehensive review and proposal for diagnostic criteria. Oral Diseases, 25(6), 1465-1491. https://doi.org/10.1111/odi.13001
Azzi L, et al. Chronic Ulcerative Stomatitis: a Comprehensive Review and Proposal for Diagnostic Criteria. Oral Dis. 2019;25(6):1465-1491. PubMed PMID: 30457193.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Chronic ulcerative stomatitis: A comprehensive review and proposal for diagnostic criteria. AU - Azzi,Lorenzo, AU - Cerati,Michele, AU - Lombardo,Maurizio, AU - Pellilli,Maria, AU - Croveri,Fabio, AU - Maurino,Vittorio, AU - Tagliabue,Angelo, AU - Tettamanti,Lucia, AU - Olszewska,Malgorzata, Y1 - 2019/01/06/ PY - 2018/03/27/received PY - 2018/09/23/revised PY - 2018/10/11/accepted PY - 2018/11/21/pubmed PY - 2019/12/31/medline PY - 2018/11/21/entrez KW - CUSP antigen KW - SES-ANA KW - chronic ulcerative stomatitis KW - hydroxychloroquine KW - oral lichen planus KW - ∆Np63α SP - 1465 EP - 1491 JF - Oral diseases JO - Oral Dis VL - 25 IS - 6 N2 - Chronic ulcerative stomatitis (CUS) is an immune-mediated disorder characterized by oral erosions and ulcers usually refractory to conventional treatments. The disease often involves middle-aged and older women with painful lesions sometimes resembling those of erosive oral lichen planus (OLP). The most affected sites are the buccal mucosa, the gingiva and the tongue, but the skin is involved in 22.5% of cases. Histopathologic features in CUS are non-specific and indistinguishable from those of OLP, with the exception of the presence of a mixed infiltrate composed of lymphocytes and plasma cells. Direct immunofluorescence (DIF) analysis reveals the presence of stratified epithelium-specific antinuclear antibodies (SES-ANA) in the lower third of the epithelium. The IgG antibodies detected on DIF are directed against the ∆Np63α isoform of p63 expressed in the nuclei of the epithelial basal cells. A distinguishing feature of CUS is the low response to conventional corticosteroid therapy and the good outcome with hydroxychloroquine at the dosage of 200 mg/day or higher dosages. This paper presents a comprehensive review of CUS and is accompanied by a new case report (the 73rd case) and a proposal for updated diagnostic criteria. SN - 1601-0825 UR - https://www.unboundmedicine.com/medline/citation/30457193/Chronic_ulcerative_stomatitis:_A_comprehensive_review_and_proposal_for_diagnostic_criteria L2 - https://doi.org/10.1111/odi.13001 DB - PRIME DP - Unbound Medicine ER -