Chronic ulcerative stomatitis: Case series of an under‐recognized entity

Abstract

Chronic ulcerative stomatitis (CUS) is a mucocutaneous condition characterized by chronic relapsing and remitting oral ulcers and erosions. This condition remains under-recognized among dermatopathologists, possibly because of common misdiagnosis as oral erosive lichen planus (LP). We report five cases of CUS in order to raise awareness of this uncommon condition. All patients presented with desquamative gingivitis and/or oral erosions, with biopsies showing lichenoid mucositis and epithelial nuclear IgG deposition on direct immunofluorescence. Recognition of the characteristic direct immunofluorescence findings allows for distinction of chronic ulcerative stomatitis from oral LP and appropriate therapy.

Correlation of clinicopathological characteristics and direct immunofluorescence studies in oral lichenoid lesion in Thai patients

“…In fact, there are many similarities between OLP and CUS, both clinically and histopathologically. DIF assays that show a granular or speckled pattern of IgG deposits within or surrounding the nuclei of keratinocytes of basal and parabasal layers of the affected stratified squamous cell epithelium are essential for the diagnosis . In our study, ENS was found in one case each in the OLL and OLDR groups.…”

“…Moreover, the lesion showed a good response to a potent topical steroid, fluocinolone acetonide 0.1% in Orabase. In fact, CUS is usually refractory to topical steroids, but improves dramatically when treated with hydroxychloroquine . Another case of a 67‐year‐old woman with OLDR also showed IgG and IgA deposits by ENS with mild intensity.…”

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

“…Gingival lesions manifest in the form of desquamative gingivitis, which is very similar to the clinical appearance of erosive OLP or vesiculobullous diseases (Solomon et al, ). The lesions are usually ulcerative and not preceded by bullae or vesicles, even though a positive Nikolsky's sign has been reported in nine cases in the literature (Beutner et al, ; Chorzelski et al, ; Ko et al, ; Reddy et al, ; Wörle et al, ).…”

“…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, ).…”