Chronic Ulcerative Stomatitis (CUS) as an Interdisciplinary Diagnostic Challenge: A Literature Review.

Chronic ulcerative stomatitis (CUS) is a rarely reported disease affecting the oral cavity, most often affecting middle-aged Caucasian females. The aim of the present study is to present the diagnosis, differentiation, and interdisciplinary treatment of this rare disease. CUS is characterized by the presence of an oral erosive or ulcerative lesion. The autoimmune pathogenesis of CUS includes affecting the antigen's activity by DNA-breaking and protein-hydrolyzing enzymes. The stratified epithelium-specific antinuclear antibodies (SES-ANA) are associated with CUS development. Clinically, the lesions presented in oral mucosa might resemble an erosive form of oral lichen planus, whereas gingival lesions seem to be similar to desquamative gingivitis related to dermatological diseases manifested in the oral cavity. Patients often report subjective symptoms related to oral mucosa and general symptoms. Histopathological presentation of CUS is often non-specific and includes sub-epithelial separation from underlying connective tissue, atrophic epithelium, and inflammatory infiltrate with an increased number of plasma cells and lymphocytes. Direct immunofluorescence (DIF) might be used in CUS diagnostics. CUS generally remains nonsusceptible to corticosteroid treatments; however, antimalarial drugs and calcineurin inhibitors are more effective. Further research should be conducted in order to implement a diagnostic protocol and observe the long-term results of CUS management.

Clinical and immunological features of chronic ulcerative stomatitis: A systematic review.

Chronic ulcerative stomatitis (CUS) is a rare disease of the mucous membranes with characteristics similar to other autoimmune diseases. The aim of this study was to conduct a systematic review of the literature to recover all reported cases of CUS in order to summarize what are the clinical, demographic, microscopic, immunological features of CUS and its therapeutic response to different drugs. A systematic review of the literature was carried out following the statements of preferred reporting items for systematic reviews and meta-analyses (PRISMA). The searches were performed in the electronic databases PubMed, Scopus, EMBASE, LILACS, Opengrey, and Google scholar. Inclusion criteria were articles or abstracts reporting at least one case with a final diagnosis of CUS. A total of 696 records were identified through databases, and 25 studies were selected reporting 81 cases. CUS affects more females (92%), and a greater number of cases are reported in Caucasian patients (53%). The age of patients ranged from 20 to 86 years with a mean age of 60 years (±13.86), and 15% of cases reported concomitantly skin lesions. The clinical and histopathological characteristics of CUS are very similar to those of oral lichen planus. The direct immunofluorescence (DIF) remains the gold-standard diagnostic resource and was performed in 69 cases, revealing a dotted pattern of deposition of stratified epithelium-specific antibodies (SES-ANA). The serum of 38 patients was collected for the performance of the indirect immunofluorescence (IIF), and the use of epithelial substrates such as monkey and guinea pig esophagus often resulted in positive SES-ANA IgG. Most patients were treated with antimalarials, and the treatment of choice that proved to be effective is hydroxychloroquine (HCQ). This entity must be considered in the differential diagnosis of other autoimmune diseases, as it may be underreported.

Bactericidal and clinical efficacy of photochemotherapy in acute necrotizing ulcerative gingivitis.

AIM: This study was designed to investigate the clinical and microbiological parameters with photochemotherapy (PCT) against conventional debridement (CD) among patients with necrotizing ulcerative gingivitis (NUG). MATERIALS AND METHODS: Patients with a diagnosis of NUG were divided into two groups: 'Group-PCT' that underwent indocyanine green-mediated PCT with adjunctive mechanical debridement, and Group-CD that underwent mechanical debridement with adjunctive 3 % hydrogen peroxide/chlorhexidine rinse. Clinical inflammatory gingival parameters including full-mouth plaque scores (FMPS), bleeding scores (FMBS), and probing depth (PD) were measured. Counts of Treponema microdentium, Fusobacterium nucleatum and Prevotella intermedia were assessed using polymerase chain reaction technique. All assessments were done at baseline, 3 weeks and 6 weeks. RESULTS: Twenty-six participants were contacted and agreed to participate in the trial. The mean age of participants in group-PCT was 24.5 years while the mean age of patients in group-CD was 26.7 years. FMPS showed significant but equal reduction in both the groups at 3 weeks and 6 weeks (p < 0.01). The reduction in mean FMBS was higher in the group-PCT at 6 weeks follow-up (p < 0.05). All bacterial levels reduced from baseline to follow-up with both PCT and CD groups (p < 0.05). Group-PCT showed significantly reduced counts of T. microdentium and F. nucleatum at 3 weeks and 6 weeks compared with group-CD (p = 0.024). Equal reduction was shown for P. intermedia between both the groups at 3 and 6 weeks, respectively. CONCLUSION: Application of adjunctive photochemotherapy was both clinically and microbiologically effective in the treatment of NUG.

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

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.

Clinical diagnosis and treatment of necrotizing ulcerative gingivitis in the orthodontic patient: a case report / Diagnóstico clínico y tratamiento de la gingivitis ulcerativa necrotizante en el paciente ortodóntico: reporte de un caso

Abstract: Introduction: about 0.1 percent of the population suffers from necrotizing ulcerative gingivitis, a disease of rapid progression and acute manifestation, which may progress to necrotizing ulcerative periodontitis and eventually to bone sequestration and loss of gingival tissue. Case report: A 21-year-old female patient undergoing orthodontic treatment for six months, diagnosed with necrotizing ulcerative gingivitis due to acute pain in the gingival tissue, spontaneous bleeding, halitosis and abundant plaque. The treatment was conservative and effective, obtaining total remission of the lesion after seven days and three months of postoperative follow-up. Conclusion: Today there are no epidemiological or clinical reports that support the relationship of necrotizing ulcerative gingivitis and orthodontic treatment. Prevention is critical to the success of the treatment, which is why the dentist should recognize the clinical features of necrotizing ulcerative gingivitis to raise awareness of its risks in the orthodontic patient.

Resumen: Introducción: alrededor del 0.1 por ciento de la población padece de gingivitis ulcerativa necrotizante, una enfermedad de progresión rápida y de presentación aguda que puede progresar a periodontitis ulcerativa necrotizante llegando a desarrollarse secuestros óseos y la pérdida de tejido gingival. Reporte del caso: Paciente femenino de 21 años de edad bajo seis meses de tratamiento ortodóntico, quien fue diagnosticada con gingivitis ulcerativa necrosante, debido a dolor agudo en el tejido gingival, sangrado espontáneo, halitosis y abundante placa bacteriana. El tratamiento fue llevado a cabo de manera conservadora y efectiva, obteniendo la remisión total de la lesión al término de siete días y los tres meses de seguimiento postoperatorio. Conclusión: Hoy en día no existen reportes epidemiológicos ni clínicos que sustenten la relación de la gingivitis ulcerativa necrotizante y el tratamiento ortodóntico. La prevención es decisiva para el éxito del tratamiento, es por ello que el odontólogo debe conocer las características clínicas de la gingivitis ulcerativa necrotizante para hacer conciencia en el paciente ortodóntico.

Necrotizing Periodontal Diseases in Children: A Literature Review and Adjustment of Treatment.

Necrotizing ulcerative gingivitis, sometimes observed in young children, may lead to necrotizing stomatitis and noma. Therefore, its interception is a necessity and a challenge for the paediatric practitioners. First, this article aims to propose a systematic review of recent literature on the use of local antiseptic and antibiotic prescription in this particular periodontal condition. Then, a protocol is proposed to have a simple, costless and reproducible treatment on children.

Acute necrotising ulcerative gingivitis in an immunocompromised young adult.

Acute necrotising ulcerative gingivitis is an acute onset disease characterised by ulceration, necrosis, pain and bleeding in gingival surfaces. It is predominantly seen in severely malnourished children and young adults with advanced HIV infection. We present a unique presentation in a young adult with high-grade osteogenic sarcoma.

Antibiotics in the treatment of periodontal disease: a guide for the general dental practitioner.

The objective of the present paper is to discuss the appropriate use of systemic and local adjunctive antibiotics/antimicrobials in the management of periodontal diseases using a number of case studies to illustrate an evidence-based approach to treatment.

Prescribing for periodontal disease.

With concerns about the ever-increasing development of antimicrobial resistance, it is imperative that antimicrobials are prescribed responsibly and used appropriately. This article provides an overview and simple guidelines for antimicrobial prescribing in the management of periodontal diseases.

Oral ulcers and necrotizing gingivitis in relation to HIV-associated neutropenia: a review and an illustrative case.

An unusual case of necrotizing gingivitis and neutropenic oral ulcers in an HIV-seropositive patient is presented. In spite of a very low CD4(+) T cell count and severe neutropenia, the necrotizing gingivitis responded favorably to standard periodontal treatment, and the oral ulcers healed after administration of granulocyte colony-stimulating factor (G-CSF). Nonspecific oral ulcers in HIV-seropositive subjects with neutropenia should be regarded as neutropenic ulcers. The term nonspecific ulcers should be restricted to those ulcers with nonspecific histopathological features in patients without neutropenia or a nutritional deficiency such as iron, folic acid, and vitamin B.

Chronic ulcerative stomatitis: a distinct clinical entity?

Chronic ulcerative stomatitis (CUS) is a mucocutaneous disorder which is characterised by persistent oral mucosal ulceration. The clinical appearance is often reminiscent of oral lichen planus (OLP) leading to erroneous diagnoses. The immune mediated inhibition of the CUS protein (CUSP) is implicated in the pathogenesis of CUS. CUSP acts as an anti-apoptotic protein and when its action is prevented it may result in significant epithelial injury. The objective or this article is to present the first documented case of CUS in South Africa, with relevant reference to current international literature. CUS should be considered in patients previously diagnosed with OLP but who are unresponsive to glucocorticosteroid therapy. The condition can be successfully managed using hydroxychloroquine.

The role of inferior alveolar nerve involvement in bisphosphonate-related osteonecrosis of the jaw.

PURPOSE: Hypesthesia or anesthesia of the lower lip (Vincent's symptom) is a common sign in patients with osteomyelitis of the mandible, especially in severe cases. PATIENTS AND METHODS: We observed an involvement of the inferior alveolar nerve in patients with bisphosphonate-related osteonecrosis of the jaw (BRONJ). Surprisingly, we found Vincent's symptom also in patients with limited and early stages of BRONJ. RESULTS: These patients were successfully treated by surgical removal of the necrotic bone combined with preoperative and postoperative administration of antibiotics. We report on the occurrence and management of an involvement of the inferior alveolar nerve in patients with BRONJ and discuss possible causes. CONCLUSION: We conclude that impairment of inferior alveolar nerve function can be an important early symptom or even the presenting symptom of BRONJ that is also easily detectable by bisphosphonate-prescribing physicians. Concerning the management of BRONJ, we conclude that surgical removal of necrotic bone combined with antibiotics is an adequate treatment in patients with osteonecrosis of the jaw.

Calcifying epithelial odontogenic tumor of the maxilla with ulcerative stomatitis: a case report.

Calcifying epithelial odontogenic tumor (CEOT) is a rare benign odontogenic tumor, known as Pindborg tumor. Although ulcer formation was reported in one previously involving the peripheral maxilla, such change of the overlying mucosa has been reported in intraosseous CEOT. We report maxillary CEOT in a patient who complained of spontaneous pain due to extensive ulcer formation of the oral mucosa.

Is there a role for antibiotics in periodontal treatment?

UNLABELLED: Plaque-induced inflammatory periodontal disease affects a significant number of the population, and raises the question as to why antibiotics are not universally used to treat a disease in which bacteria are the main aetiological agent. This article describes the reasons why antibiotics may not be effective in treating periodontitis, and why their use is not more widespread. However, antibiotics have been shown to be helpful in periodontal treatment in some cases, and evidence for this is presented and suggestions where their use may be indicated are made. CLINICAL RELEVANCE: To rationalize the selective use of antibiotics in the treatment of periodontitis and avoid inappropriate prescribing.

Validation of the oral mucositis assessment scale in pediatric cancer.

BACKGROUND: Our objective was to examine the construct validity of the Oral Mucositis Assessment Scale (OMAS) in children receiving doxorubicin chemotherapy. METHODS: Children between 6 and 18 years of age with cancer receiving doxorubicin-containing chemotherapy were included. OMAS was measured on days 7, 10, 14, and 17 after chemotherapy. Other measures of mucositis obtained concurrent with OMAS were the World Health Organization (WHO) mucositis scale and pain visual analogue scale (VAS). We also recorded analgesia administration. RESULTS: Sixteen children were studied for 45 post-chemotherapy cycles and 156 OMAS assessments were performed. OMAS was moderately correlated with WHO scores (r = 0.56; P = 0.0006) whereas correlation with the pain VAS was fair (r = 0.37; P = 0.002). OMAS also had fair correlation with the number of doses of topical analgesia (r = 0.43; P = 0.001) and with the cumulative dose of opioid analgesia (r = 0.38; P = 0.003). CONCLUSIONS: The OMAS is valid for use in mucositis clinical trials for children at least 6 years of age.

Necrotizing ulcerative periodontal diseases in children and young adults in Medellín, Colombia, 1965--2000.

Previous findings that necrotizing ulcerative gingivitis (NUG) is a precursor to noma or cancrum oris were confirmed by the follow-up of these clinical conditions in a study of 45 Colombian patients: necrotizing ulcerative gingivitis (NUG, n = 29), necrotizing ulcerative periodontitis (NUP, n = 7) and noma (n = 9). Patients were diagnosed at the outpatient clinic of the School of Dentistry at the University of Antioquia, at the University Hospital Saint Vincent of Paul, at the Luz Castro de Gutierrez University Hospital, at the Red Cross Hospital and at the private office of one of the authors (Jiménez L., M) in Medellín, Colombia, from 1965 until 2000. Almost all the patients came from low socioeconomic groups and presented with predisposing and/or contributing factors, such as acute herpetic gingivostomatitis, measles, and leukemia (including acute lymphoblastic and chronic lymphoid leukemia). Malnutrition and poor oral hygiene were associated with the necrotizing process and favored progression from the gingiva to deeper periodontal tissues and other structures within the oral cavity or the facial tissues. No patients had human immunodeficiency virus (HIV) or AIDS, which makes these findings different from other reports. Noma can be prevented by vaccinating children against infectious diseases, by controlling malnutrition and by improving their oral hygiene. It is arrested by mechanical lesion debridement, improving oral hygiene and antibiotic therapy. Necrotizing ulcerative gingivitis may progress in some cases to ulcerative necrotizing stomatitis, necrotizing ulcerative periodontitis, and, finally, to noma. Microbial studies among new Colombian NUG, NUP and noma patients are necessary, using bacterial culturing and identification methods and molecular techniques such as PCR for viruses and bacteria, in order to establish the exact nature of these lesions.