Chronic ulcerative stomatitis

Abstract

Chronic ulcerative stomatitis (CUS) has recently been described as a new disease entity characterized by chronic ulceration of oral mucosa which responds to treatment with hydroxychloroquine. It has a particular type of stratified epithelium-specific, antinuclear autoantibody as an immunological marker. Twelve cases have been reported in the literature. We present a 40-year-old woman with an 11-year history of chronic oral ulcerations. Other dermatological diseases, including oral lichen planus, pemphigus vulgaris and cicatricial pemphigoid, as well as bullous lupus erythematosus, were excluded. The clinical diagnosis of CUS was confirmed on the grounds of the immunological and ultrastructural findings. The lesions initially responded to high doses of systemic corticosteroids but relapsed promptly after dose reduction. Dapsone was ineffective. Hydroxychloroquine, given at a dosage of 200-400 mg/day, led to a complete and long-lasting remission.

The diagnostic challenges of separating chronic ulcerative stomatitis from oral lichen planus

“…None of the 10 patients had any significant skin lesions 17 The microscopic findings were generally nonspecific but demonstrated lichenoid features, such as ulceration, hydropic degeneration of basal layer cells and reduplication of the basement membrane, variation in epithelial thickness (with some lesions having a flat interface between the epithelium and connective tissue), saw-tooth rete ridges, and colloid bodies. The band-like infiltration of inflammatory cells varied in composition, with some infiltrates composed purely of lymphocytes and others having a mixture of lymphocytes and plasma cells.…”

Desquamative gingivitis: A sign of mucocutaneous disorders‐a review

“…Treatment is with potent topical steroids or hydroxychloroquine, the latter reportedly bringing about complete and long lasting remission. 13,40,61,62 …”

Chronic ulcerative stomatitis: A comprehensive review and proposal for diagnostic criteria

“…Other lesions may appear non‐specific: a few patients have shown an asymmetrical distribution and single‐site involvement. In nine cases, vesicles and Nikolsky's sign were observed (Beutner et al, ; Chorzelski et al, ; Ko et al, ; Reddy et al, ; Wörle et al, ).…”

“…To date, 72 cases have been reported (Alshagroud, Neiders, Kramer, & Suresh, ; Beutner et al, ; Chorzelski, Olszewska, Jarząbek‐Chorzelska, & Jablońska, ; Church & Schosser, ; Fourie, van Heerden, McEachen, & van Zyl, ; Islam et al, ; Jaremko et al, ; Kapińska‐Mrowiecka, Czubak‐Macugowska, Michcik, Chomik, & Wlodarkiewicz, ; Ko, Danciu, Fullen, & Chan, ; Lewis, Beutner, Rostami, & Chorzelski, ; Lorenzana, Rees, Glass, & Detweiler, ; Molenda & Kozlowski, ; Parodi & Cardo, ; Qari et al, ; Reddy, Fitzpatrick, Bhattacharyya, Cohen, & Islam, ; Solomon et al, ; Wörle et al, ). However, a literature review has indicated that its prevalence in the general population could be underestimated (Solomon, ).…”