Skip to main content
Top
Published in: Head and Neck Pathology 3/2019

01-09-2019 | Original Paper

Seventeen New Cases of Chronic Ulcerative Stomatitis with Literature Review

Authors: Rekha Reddy, Sarah G. Fitzpatrick, Indraneel Bhattacharyya, Donald M. Cohen, Mohammed N. Islam

Published in: Head and Neck Pathology | Issue 3/2019

Login to get access

Abstract

Chronic ulcerative stomatitis (CUS) is a poorly understood disease with clinical and histologic overlap with lichen planus (LP). Unlike classic LP, direct immunofluorescence (DIF) studies in cases of CUS exhibit a granular pattern of IgG in nuclei of basal and parabasal cells. This study assesses the demographic, clinical, histologic, and DIF features of CUS. It is important to differentiate CUS from LP and other vesiculobullous diseases (VBD) because lesions of CUS are resistant to steroid therapy, which is typically used to control LP and VBD. A literature review and IRB-approved retrospective search of CUS was performed within the archives of the University of Florida (UF) Oral Pathology Biopsy Service from 2007 to 2017. Fifty-two cases were identified from the literature and seventeen new cases were identified in our series. All UF patients were female and the median age was 64-years. The majority of patients were Caucasian and the most common location was buccal mucosa. Frequent clinical presentations were pain, erythema, leukoplakia, and ulcerations. Histologic features included epithelial separation, atrophic epithelium, and a chronic inflammatory infiltrate. All cases were confirmed with DIF testing that showed a speckled pattern of IgG staining in basal and parabasal cell nuclei. Fibrinogen was present in eleven cases and two cases were positive for C3. The results of our series are in accordance with the literature. Since CUS has overlapping features with LP and VBD, clinicians and pathologists should consider this entity and confirm diagnosis with DIF testing when recalcitrant oral ulcerative diseases are encountered.
Literature
1.
go back to reference Jaremko WM, Beutner EH, Kumar V, Kipping H, Condry P, Zeid MY, Kauffmann CL, Tatakis DN, Chorzelski TP. Chronic ulcerative stomatitis associated with a specific immunologic marker. J Am Acad Dermatol. 1990;22(2 Pt 1):215–20.CrossRefPubMed Jaremko WM, Beutner EH, Kumar V, Kipping H, Condry P, Zeid MY, Kauffmann CL, Tatakis DN, Chorzelski TP. Chronic ulcerative stomatitis associated with a specific immunologic marker. J Am Acad Dermatol. 1990;22(2 Pt 1):215–20.CrossRefPubMed
2.
go back to reference Feller L, Khammissa RAG, Lemmer J. Is chronic ulcerative stomatitis a variant of lichen planus, or a distinct disease? J Oral Pathol Med. 2017;46(10):859–63.PubMed Feller L, Khammissa RAG, Lemmer J. Is chronic ulcerative stomatitis a variant of lichen planus, or a distinct disease? J Oral Pathol Med. 2017;46(10):859–63.PubMed
3.
go back to reference Islam MN, Cohen DM, Ojha J, Stewart CM, Katz J, Bhattacharyya I. Chronic ulcerative stomatitis: diagnostic and management challenges—four new cases and review of literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endodontol. 2007;104(2):194–203.CrossRef Islam MN, Cohen DM, Ojha J, Stewart CM, Katz J, Bhattacharyya I. Chronic ulcerative stomatitis: diagnostic and management challenges—four new cases and review of literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endodontol. 2007;104(2):194–203.CrossRef
4.
go back to reference Carlson MW, Garlick JA, Solomon LW. Chronic ulcerative stomatitis: evidence of autoimmune pathogenesis. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2011;111(6):742–8.CrossRefPubMed Carlson MW, Garlick JA, Solomon LW. Chronic ulcerative stomatitis: evidence of autoimmune pathogenesis. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2011;111(6):742–8.CrossRefPubMed
5.
go back to reference Solomon LW, Stark PC, Winter L, Kumar V, Sinha S. ELISA test for p63 antibodies in chronic ulcerative stomatitis. Oral Dis. 2010;16(2):151–5.CrossRefPubMed Solomon LW, Stark PC, Winter L, Kumar V, Sinha S. ELISA test for p63 antibodies in chronic ulcerative stomatitis. Oral Dis. 2010;16(2):151–5.CrossRefPubMed
6.
go back to reference Neville BW, Damm DD, Allen CM, Chi AC. Oral and Maxillofacial Pathology. 4th ed. St. Louis: Saunders; 2015. Neville BW, Damm DD, Allen CM, Chi AC. Oral and Maxillofacial Pathology. 4th ed. St. Louis: Saunders; 2015.
7.
go back to reference Qari H, Villasante C, Richert J, Rees T, Kessler H. The diagnostic challenges of separating chronic ulcerative stomatitis from oral lichen planus. Oral Surg Oral Med Oral Pathol Oral Radiol. 2015;120(5):622–7.CrossRefPubMed Qari H, Villasante C, Richert J, Rees T, Kessler H. The diagnostic challenges of separating chronic ulcerative stomatitis from oral lichen planus. Oral Surg Oral Med Oral Pathol Oral Radiol. 2015;120(5):622–7.CrossRefPubMed
8.
go back to reference Parodi A, Cardo PP. Patients with erosive lichen planus may have antibodies directed to a nuclear antigen of epithelial cells: a study on the antigen nature. J Invest Dermatol. 1990;94(5):689–93.CrossRefPubMed Parodi A, Cardo PP. Patients with erosive lichen planus may have antibodies directed to a nuclear antigen of epithelial cells: a study on the antigen nature. J Invest Dermatol. 1990;94(5):689–93.CrossRefPubMed
9.
go back to reference Solomon LW, Neiders ME, Zwick MG, Kirkwood KL, Kumar V. Autoimmunity to deltaNp63alpha in chronic ulcerative stomatitis. J Dent Res. 2007;86(9):826–31.CrossRefPubMed Solomon LW, Neiders ME, Zwick MG, Kirkwood KL, Kumar V. Autoimmunity to deltaNp63alpha in chronic ulcerative stomatitis. J Dent Res. 2007;86(9):826–31.CrossRefPubMed
10.
go back to reference Lee LA, Walsh P, Prater CA, Su LJ, Marchbank A, Egbert TB, Dellavalle RP, Targoff IN, Kaufman KM, Chorzelski TP, Jablonska S. Characterization of an autoantigen associated with chronic ulcerative stomatitis: the CUSP autoantigen is a member of the p53 family. J Invest Dermatol. 1999;113(2):146–51.CrossRefPubMed Lee LA, Walsh P, Prater CA, Su LJ, Marchbank A, Egbert TB, Dellavalle RP, Targoff IN, Kaufman KM, Chorzelski TP, Jablonska S. Characterization of an autoantigen associated with chronic ulcerative stomatitis: the CUSP autoantigen is a member of the p53 family. J Invest Dermatol. 1999;113(2):146–51.CrossRefPubMed
11.
go back to reference Parodi A, Cozzani E, Chorzelski TP, Beutner EH, Rebora A. A molecule of about 70 kd is the immunologic marker of chronic ulcerative stomatitis. J Am Acad Dermatol. 1998;38(6 Pt 1):1005–6.CrossRefPubMed Parodi A, Cozzani E, Chorzelski TP, Beutner EH, Rebora A. A molecule of about 70 kd is the immunologic marker of chronic ulcerative stomatitis. J Am Acad Dermatol. 1998;38(6 Pt 1):1005–6.CrossRefPubMed
12.
go back to reference Romano RA, Solomon LW, Sinha S. Tp63 in oral development, neoplasia, and autoimmunity. J Dent Res. 2012;91:125–32.CrossRefPubMed Romano RA, Solomon LW, Sinha S. Tp63 in oral development, neoplasia, and autoimmunity. J Dent Res. 2012;91:125–32.CrossRefPubMed
14.
go back to reference Solomon LW, Aguirre A, Neiders M, Costales-Spindler A, Jividen GJ Jr, Zwick MG, Kumar V. Chronic ulcerative stomatitis: clinical, histopathologic, and immunopathologic findings. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2003;96:718–26.CrossRefPubMed Solomon LW, Aguirre A, Neiders M, Costales-Spindler A, Jividen GJ Jr, Zwick MG, Kumar V. Chronic ulcerative stomatitis: clinical, histopathologic, and immunopathologic findings. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2003;96:718–26.CrossRefPubMed
15.
go back to reference Fourie J, van Heerden WF, McEachen SC, van Zyl A. Chronic ulcerative stomatitis: a distinct clinical entity? SADJ. 2011;66(3):119–121.PubMed Fourie J, van Heerden WF, McEachen SC, van Zyl A. Chronic ulcerative stomatitis: a distinct clinical entity? SADJ. 2011;66(3):119–121.PubMed
16.
go back to reference Worle B, Wollenberg A, Schaller M, Kunzelmann KH, Plewig G, Murer M. Chronic ulcerative stomatitis. Br J Dermatol. 1997;137(2):262–5.CrossRefPubMed Worle B, Wollenberg A, Schaller M, Kunzelmann KH, Plewig G, Murer M. Chronic ulcerative stomatitis. Br J Dermatol. 1997;137(2):262–5.CrossRefPubMed
17.
go back to reference Church LF Jr, Schosser RH. Chronic ulcerative stomatitis associated with stratified epithelial specific antinuclear antibodies: a case report of a newly described disease entity. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1992;73(5):579–82.CrossRef Church LF Jr, Schosser RH. Chronic ulcerative stomatitis associated with stratified epithelial specific antinuclear antibodies: a case report of a newly described disease entity. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1992;73(5):579–82.CrossRef
18.
go back to reference Beutner EH, Chorzelski TP, Parodi A, Schosser R, Guin J, Cardo PP, Maciejowska E, Valeski JE, Kumar V. Ten cases of chronic ulcerative stomatitis with stratified epithelium–specific antinuclear antibody. J Am Acad Dermatol. 1991;24(5 Pt 1):781–2.CrossRefPubMed Beutner EH, Chorzelski TP, Parodi A, Schosser R, Guin J, Cardo PP, Maciejowska E, Valeski JE, Kumar V. Ten cases of chronic ulcerative stomatitis with stratified epithelium–specific antinuclear antibody. J Am Acad Dermatol. 1991;24(5 Pt 1):781–2.CrossRefPubMed
19.
go back to reference Lewis JE, Beutner EH, Rostami R, Chorzelski TP. Chronic ulcerative stomatitis with stratified epithelium-specific antinuclear antibodies. Int J Dermatol. 1996;35(4):272–5.CrossRefPubMed Lewis JE, Beutner EH, Rostami R, Chorzelski TP. Chronic ulcerative stomatitis with stratified epithelium-specific antinuclear antibodies. Int J Dermatol. 1996;35(4):272–5.CrossRefPubMed
20.
go back to reference Chorzelski TP, Olszewska M, Jarzabek-Chorzelska M, Jablonska S. Is chronic ulcerative stomatitis an entity? Clinical and immunological findings in 18 cases. Eur J Dermatol. 1998;8(4):261–5.PubMed Chorzelski TP, Olszewska M, Jarzabek-Chorzelska M, Jablonska S. Is chronic ulcerative stomatitis an entity? Clinical and immunological findings in 18 cases. Eur J Dermatol. 1998;8(4):261–5.PubMed
21.
go back to reference Lorenzana ER, Rees TD, Glass M, Detweiler JG. Chronic ulcerative stomatitis: A case report. J Periodontol. 2000;71(1):104–11.CrossRefPubMed Lorenzana ER, Rees TD, Glass M, Detweiler JG. Chronic ulcerative stomatitis: A case report. J Periodontol. 2000;71(1):104–11.CrossRefPubMed
22.
go back to reference Alshagroud R, Neiders M, Kramer JM, Suresh L. Clinicopathologic significance of in vivo antinuclear autoantibodies in oral mucosal biopsies. Oral Surg Oral Med Oral Pathol Oral Radiol. 2017;124(5):475–82.CrossRefPubMed Alshagroud R, Neiders M, Kramer JM, Suresh L. Clinicopathologic significance of in vivo antinuclear autoantibodies in oral mucosal biopsies. Oral Surg Oral Med Oral Pathol Oral Radiol. 2017;124(5):475–82.CrossRefPubMed
23.
go back to reference Waranun B, Nis O, Supanee T, Titikarn L. Direct immunofluorescence in oral lichen planus. J Clin Diagn Res. 2015;9(8):ZC34–7. Waranun B, Nis O, Supanee T, Titikarn L. Direct immunofluorescence in oral lichen planus. J Clin Diagn Res. 2015;9(8):ZC34–7.
24.
go back to reference Ebrahimi M, Wahlin YB, Coates PJ, Wiik A, Roos G, Nylander K. Detection of antibodies against p63 and p73 isoforms in sera from patients diagnosed with oral lichen planus. J Oral Pathol Med. 2007;36(2):93–8.CrossRefPubMed Ebrahimi M, Wahlin YB, Coates PJ, Wiik A, Roos G, Nylander K. Detection of antibodies against p63 and p73 isoforms in sera from patients diagnosed with oral lichen planus. J Oral Pathol Med. 2007;36(2):93–8.CrossRefPubMed
25.
go back to reference Cozzani E, Cacciapuoti M, di Marco E, Zerega B, Descalzi Cancedda F, Parodi A. Patients with oral erosive and cutaneous lichen planus may have antibodies directed against the chronic ulcerative stomatitis protein antigen of 70-kDa. Acta Dermatovenerol Alp Pannonica Adriat. 2008;17(3):120–4.PubMed Cozzani E, Cacciapuoti M, di Marco E, Zerega B, Descalzi Cancedda F, Parodi A. Patients with oral erosive and cutaneous lichen planus may have antibodies directed against the chronic ulcerative stomatitis protein antigen of 70-kDa. Acta Dermatovenerol Alp Pannonica Adriat. 2008;17(3):120–4.PubMed
Metadata
Title
Seventeen New Cases of Chronic Ulcerative Stomatitis with Literature Review
Authors
Rekha Reddy
Sarah G. Fitzpatrick
Indraneel Bhattacharyya
Donald M. Cohen
Mohammed N. Islam
Publication date
01-09-2019
Publisher
Springer US
Published in
Head and Neck Pathology / Issue 3/2019
Electronic ISSN: 1936-0568
DOI
https://doi.org/10.1007/s12105-018-0982-7

Other articles of this Issue 3/2019

Head and Neck Pathology 3/2019 Go to the issue