Peripheral ameloblastic fibroma: a rare case report.

CASE REPORT: Ameloblastic fibroma is a rare mixed odontogenic tumour which involves mostly the posterior mandible. Its peripheral variant is very rare. Only eight cases have been reported worldwide. In this report, we described a case of peripheral ameloblastic fibroma occurring in the maxillary gingiva in a 10 year-old child. The lesion was excised with a conservative surgical approach and no recurrence has occurred. Peripheral ameloblastic fibroma should be considered in the differential diagnosis of a slow growing lesion involving the gingiva.

Clinical characterization of gingival type of burning mouth syndrome: a cross-sectional study.

BACKGROUND: The aim of this study is to investigate the prevalence of localized intraoral neuropathic pain in a cluster of patients who reported the involvement of gingival site as only clinical manifestation of dysesthesia, analysing type and distribution of symptoms. MATERIAL AND METHODS: Burning mouth syndrome (BMS) patients were enrolled in the study. Patients were screened through laboratory test and a conventional oral examination with periodontal chart. A questionnaire to collect data on symptoms, oral site involved, quality of sleep, anxiety was submitted to all the patients. RESULTS: A total of 236 patients were recruited. Seventy-six patients (32.2%) presented generalized type, whereas 160 (67.8%) had localized type. In the localized BMS group, the gingiva was involved in 70 patients and in 33 of these it was the only site involved. In the gingival subgroup, 35 patients reported burning, 29 pain and 24 xerostomia. CONCLUSIONS: To best our knowledge, this study is the first that analyses gingival involvement as the only site in BMS and it could encourage further investigations to understand the etiopathogenesis of gingival BMS.

The association between burning mouth syndrome and sleep disturbance: A case-control multicentre study.

OBJECTIVES: To investigate the quality of sleep and the psychological profiles of a large cohort of Italian patients with burning mouth syndrome (BMS) and to clarify the relationships between these variables and pain. METHODS: In this case-control study, 200 patients with BMS vs an equal number of age- and sex-matched healthy controls, recruited in 10 universities, were enrolled. The Pittsburgh Sleep Quality Index (PSQI), Epworth Sleepiness Scale (ESS), Hamilton Rating Scale for Depression (HAM-D), Hamilton Rating Scale for Anxiety (HAM-A), Numeric Pain Intensity Scale (NRS) and Total Pain Rating Index (T-PRI) were administered. Descriptive statistics, including the Mann-Whitney U test and hierarchical multiple linear regression analysis, were used. RESULTS: Poor sleep quality (PSQI ≥ 5) was present in 78.8% (160) patients with BMS. BMS patients had statistically higher scores in all items of the PSQI and ESS than the healthy controls (p < .001). A depressed mood and anxiety correlated positively with sleep disturbance. The Pearson correlations were 0.570 for the PSQI vs HAM-D (p < .001) and 0.549 for the PSQI vs HAM-A (p < .001). Pain intensity (NRS) poorly correlated to sleep quality; the Pearson correlation was 0.162 for the PSQI vs NRS (p = .021). CONCLUSIONS: The BMS patients showed a poor sleep quality, anxiety and depression, as compared with the controls, highlighting the relationships between oral burning, sleep and mood.

Time to onset of bisphosphonate-related osteonecrosis of the jaws: a multicentre retrospective cohort study.

OBJECTIVES: Osteonecrosis of the jaw (ONJ) is a potentially severe adverse effect of bisphosphonates (BP). Although the risk of ONJ increases with increasing duration of BP treatment, there are currently no reliable estimates of the ONJ time to onset (TTO). The objective of this study was to estimate the TTO and associated risk factors in BP-treated patients. SUBJECTS AND METHODS: Retrospective analysis of data from 22 secondary care centres in seven countries relevant to 349 patients who developed BP-related ONJ between 2004 and 2012. RESULTS: The median (95%CI) TTO was 6.0 years in patients treated with alendronate (n = 88) and 2.2 years in those treated with zoledronate (n = 218). Multivariable Cox regression showed that dentoalveolar surgery was inversely associated, and the use of antiangiogenics directly associated, with the TTO in patients with cancer treated with zoledronate. CONCLUSIONS: The incidence of ONJ increases with the duration of BP therapy, with notable differences observed with respect to BP type and potency, route of administration and underlying disease. When data are stratified by BP type, a time of 6.0 and 2.2 years of oral alendronate and intravenous zoledronate therapy, respectively, is required for 50% of patients to develop ONJ. After stratification by disease, a time of 5.3 and 2.2 years of BP therapy is required for 50% of patients with osteoporosis and cancer, respectively, to develop ONJ. These findings have significant implications for the design of future clinical studies and the development of risk-reduction strategies aimed at either assessing or modulating the risk of ONJ associated with BP.

Comparison of views on the need for continuing education on oral cancer between general dentists and oral medicine experts: A Delphi survey.

OBJECTIVES: The role of dental professionals in screening for oral cancer has been limited. The purpose of this study was to investigate and compare the educational priorities of oral medicine specialists, general dental practitioners, and doctors of dental surgery with regards to the diagnosis and management of oral cancers and potential malignant disorders. STUDY DESIGN: This was a longitudinal survey. MATERIALS AND METHODS: A Delphi survey was directed to a panel of 25 oral medicine specialists asking them to identify the major difficulties in diagnosing and managing patients with oral cancer or suspected malignancy. In a second phase, two groups of generalists were asked to express their ratings on the issues identified by experts. RESULTS: The response rate of the experts to the survey was 84%, while only 44% of the generalists participated. Although the three groups agreed on most of the issues, there were significant differences of opinions on 10 of the items proposed by specialists (P < 0.05 from the Kruskal-Wallis test), which were observed mainly between experts and general dental practitioners (P < 0.017 from the Mann-Whitney U test). The opinion of the participants about future investments in the field of education resulted in similar results (P > 0.05 from the Chi-square test), with the specialists ranking highest on mandatory annual thematic courses, while the generalists prioritizing more interactive and extensive pre-graduation courses on oral cancer detection. CONCLUSION: This study confirms a clear need to improve the educational foundation on oral cancer by a didactic process starting with pre-graduation courses that should involve National Health Care Services, National Dental Associations, and academia.

Primary oral leishmaniasis mimicking oral cancer: a case report.

Primary mucosal leishmaniasis is a rare infectious disease, particularly in immunocompetent patients. We present a 50-year-old patient with a 6-week history of a painful lesion of the left buccal mucosa that mimicked cancer. The exophytic lesion looked invasive, and we took an incisional biopsy specimen to exclude cancer. The diagnosis of leishmaniasis was unexpected, and the patient was successfully treated with amphotericin B for five weeks. After five months the patient had a visceral recurrence. Chronic exophytic and ulcerated mucosal lesions that do not heal within 3-4 weeks should be regarded as the first signs of oral cancer, but primary oral leishmaniasis can easily mimic it.

The GOCCLES® medical device is effective in detecting oral cancer and dysplasia in dental clinical setting. Results from a multicentre clinical trial.

The purpose of this study is to demonstrate that the GOCCLES® medical device allows proper autofluorescence examination of the oral mucosa in a dental care setting. This is a non-randomised multicentre clinical trial on consecutive patients at risk for oral cancer. Patients underwent a classical naked eye inspection of the oral cavity followed by autofluorescence examination wearing the GOCCLES® spectacles while the light from a dental curing light irradiated the oral mucosa. Lesions were defined as visible potentially malignant lesions and/or fluorescence loss areas. All persisting lesions underwent excisional or incisional biopsy. Sixty-one patients were enrolled. Data from 64 biopsies were analysed. Of the 62 lesions identified by the device, 31 were true positives. The device identified 31 of 32 true positive lesions. One lesion (an invasive carcinoma) was not visible to the naked eye. The device identified all lesions classified as moderate dysplasia to invasive cancer. In 56.7% of cases, true positive lesions showed greater extension when observed through the device. The GOCCLES® medical device allowed the direct visualisation of fluorescence loss in patients suffering from mild to severe dysplasia and in situ to invasive oral cancer. It allowed autofluorescence examination with each source of light used during the study. These results suggest that the role of the autofluorescence visualisation is that of a complementary inspection following naked eye examination when dealing with patients at risk for oral cancer. The device allows detection of otherwise invisible lesions and otherwise impossible complete resections.

Mucosal leishmaniasis with primary oral involvement: a case series and a review of the literature.

OBJECTIVE: To analyze retrospectively a case series of primary oral leishmaniasis and to review the literature on head-neck primary mucosal leishmaniasis (ML) in immunocompetent patients. SUBJECTS AND METHODS: A PUBMED search was carried out from 1950 to 2013. Clinical records of patients with primary head-neck mucosal manifestations of leishmaniasis were analyzed. In addition, clinical records between 2001 and 2012 of patients with primary oral manifestations were collected in two independent hospitals. RESULTS: Our multicenter case series revealed seven patients with oral leishmaniasis. The most commonly affected site was the tongue (four patients, 57%), and the most common clinical presentation was an exophytic lesion (six patients, 85%). The literature review showed 11 reports published between 2005 and 2013, describing 13 patients (100% male) affected by head-neck primary ML (54% laryngeal, 31% oral, 23% pharyngeal, and 15% endonasal). The most common clinical presentation was an exophytic lesion (69%). CONCLUSIONS: The literature analysis revealed that in immunocompetent patients, the oral mucosa is the second most frequently affected site of the head and neck region. In the oral cavity, the tongue is the most affected site. Diagnosis of oral leishmaniasis represents a challenge but must be considered in any differential diagnosis of exophytic lesions of oral mucosa.

Sleep disturbances, anxiety and depression in patients with oral lichen planus: a case-control study.

BACKGROUND: The psychological factors and their association with chronic inflammatory disease, aren't well recognized, yet their importance in oral lichen planus is still debated. AIMS: The aim of this study was to investigate the prevalence of sleep disturbances, anxiety, depression and their association in patient with oral lichen planus. MATERIALS AND METHODS: 50 patients with oral lichen planus vs. equal number of age and sex-matched healthy controls were enrolled. Questionnaires examining insomnia symptoms, excessive daytime sleepiness (Pittsburgh sleep quality index and Epworth aleepiness scale) depression and anxiety (The Hamilton rating scale for Depression and Anxiety) were used. RESULTS: The patients with oral lichen planus had statistically higher scores in all items of the Pittsburgh sleep quality index, the Hamilton rating scale for depression and anxiety and Epworth sleepiness scale than the healthy controls. The median and inter-quartile range of the Pittsburgh sleep quality index was 5-2 and for the oral lichen planus patients and 4-2 for the healthy controls (P < 0.011). In the study group, a depressed mood and anxiety correlated positively with sleep disturbances. The Pearson correlations were 0.76 for Pittsburgh sleep quality Index vs. Hamilton rating scale for depression (P < 0.001) and 0.77 for Pittsburgh sleep quality Index vs. Hamilton rating scale for anxiety (P < 0.001). DISCUSSION: Oral lichen planus patients report a greater degree of sleep problems, depressed mood and anxiety as compared with controls. CONCLUSION: We suggest to screen sleep disturbances in patients with oral lichen planus because they could be considered a prodromal symptoms of mood disorders.

Fine needle aspiration cytology of lymphoproliferative lesions of the oral cavity.

OBJECTIVE: Oral cavity non-Hodgkin lymphoma (OCL) is a rare condition that may be clinically and radiologically indistinguishable from other pathologies of the mouth. A complete excision or adequate biopsy of the OCL may be difficult. Fine needle aspiration (FNA) cytology has been successfully utilized in the pre-operative diagnosis of oral masses and in lymphoma involving other anatomical areas. Our experience with FNA pre-operative cytological diagnosis of 16 OCLs is reported herein. METHODS: The results of FNA cytology on 16 consecutive lymphoproliferative lesions of the oral cavity collected over an 8-year period in three institutions were retrieved. Sampled lesions were submucosal masses of different sizes bulging into the oral cavity. Rapid on-site evaluation (ROSE) and routine cytological staining were performed. Immunocytochemistry (ICC), flow cytometry (FC) and polymerase chain reaction (PCR) of the IGH (immunoglobulin heavy) locus were performed on additional passes according to ROSE. RESULTS: Fourteen OCLs, one myeloma and one florid reactive lymphoid hyperplasia (FRLH) were diagnosed by FNA. OCLs were diagnosed as large B-cell (eight cases) and small B-cell (six cases) lymphomas. Histology revealed eight diffuse large B-cell lymphomas (DLBCL), four lymphomas of mucosa-associated lymphoid tissue (MALT), two follicular lymphomas and one FRLH; no false-negative or false-positive results were diagnosed, but accurate subclassification was obtained in four cases only. CONCLUSIONS: FNA diagnosis of OCLs may be hampered by the rare incidence, anatomical context and difficulties in obtaining a sufficient amount of cells. Ancillary techniques should be used according to ROSE; a pre-operative FNA cytology diagnosis can avoid unnecessary extensive surgery and speed up the institution of therapeutic procedures.

Adjuvant triamcinolone acetonide injections in oro-pharyngeal pemphigus vulgaris.

BACKGROUND: High-potency topical and perilesional/intralesional corticosteroids are becoming increasingly useful as adjuvant to treat autoimmune blistering diseases. OBJECTIVE: We sought to evaluate the role of perilesional/intralesional triamcinolone acetonide (PITA) injections in reducing the time for first complete clinical remission and the total amount of systemic corticosteroids in oro-pharyngeal pemphigus vulgaris (OPV) patients, and also the compliance of PITA injections, in terms of satisfaction, pain and discomfort. METHODS: Thirty-five OPV patients were treated with conventional immunosuppressive therapy (CIST) and received high potency topical corticosteroids (clobetasol and/or methylprednisolone) and/or PITA injections. Patients were grouped as follows: (i) a group of 16 patients was treated with PITA injections and (ii) a group of 19 patients without PITA injections. RESULTS: Sixteen patients treated with PITA injections and 19 without PITA injections reached complete clinical remission within 126.6 days (SD: 41; 95% CI: 104.7-148.8) and 153.2 days (SD: 97.4; 95% CI: 106.2-200.1) (P = 0.4) respectively. The total amount of corticosteroids in patients treated with PITA and without PITA was 4894 mg (SD: 2832; 95% CI: 3385-6403) and 5312 mg (SD: 4009; 95% CI: 3380-7245) (P = 0.4) respectively. Patients treated with PITA reported a satisfaction score significantly higher than pain (P = 0.0007) and discomfort score (P = 0.0006). CONCLUSION: Perilesional/intralesional triamcinolone acetonide injections seems to represent a helpful clinical tool to successfully join CIST, in terms of shortening the time of complete clinical remission, reducing the total amount of corticosteroids and obtaining an acceptable compliance.

Oral pemphigus.

Pemphigus is a group of potentially life-threatening autoimmune diseases characterized by cutaneous and/or mucosal blistering, due to the presence circulating IgG antibodies directed against desmoglein 1 and 3 (Dsg 1 and 3). Differences in the particular distribution of these result in different clinical manifestations of the disease. The most common variant is pemphigus vulgaris (PV). There is a fairly strong genetic background to pemphigus with linkage to HLA class II alleles and ethnic groups such as Ashkenazi Jews and those of Mediterra-nean and Indian origin are especially liable. Oral lesions are commonly characterized by the presence of vesiculobullous and ulcerative lesions. Diagnosis is achieved via three different parameters: perilesional tissue biopsy, histological and immunological examinations. Serum autoantibodies to either Dsg1 or Dsg3 are best detected using both normal human skin and monkey esophagus or by enzyme-linked immunosorbent assay. The main aim of treatment is to reduce inflammatory re-sponse and autoantibody production, in order to achieve disease remission in a short time. Before the advent of corticosteroids, PV was typically fatal due to dehydration or secondary systemic infections. Current treatment is largely based on systemic immunosuppression using corticosteroids, with azathioprine or other adjuvants or alternatives. Nonetheless, newer therapies, such as intravenous immunoglobulins (IVIg) or anti-CD20 monoclonal antibodies (Rituximab), with potentially fewer adverse effects also appear promising.

Analysis of thromboembolic risk related to high-dose intravenous immunoglobulin treatment: a preliminary clinical study of 10 patients with autoimmune mucocutaneous blistering diseases.

BACKGROUND: Intravenous immunoglobulin (IVIg) treatment is a well-known treatment that has been used successfully in a broad spectrum of autoimmune diseases. Currently no data are available in the literature about the role of IVIg in the pathogenesis of thromboembolic events in patients with autoimmune blistering diseases refractory to conventional immunosuppressive treatment. AIM: To determine the relationship between IVIg and thromboembolism in patients with autoimmune blistering diseases and to establish a protocol to deal with the thromboembolic risk. METHODS: In our preliminary clinical study, 10 patients with autoimmune blistering diseases underwent IVIg cycles to a total of 133 cycles in all (total number of infusions in the patient group: 399), at a standard dose of 2 g/kg/infusion accompanied by an accurate and a complete clinical and laboratory screening for thromboembolism. Preventive measures, such as hydration before and after IVIg, and administration of 100 mg of acetyl salicylic acid (aspirin) or 1000 IU of subcutaneous heparin calcium per day for 3 weeks, were introduced to reduce the thromboembolic risk. RESULTS: Throughout the 2 years of IVIg treatment, no patient developed a superficial and/or deep venous or arterial thrombosis, even though some of the patients had underlying thromboembolic risk factors and had tested positive for some congenital and acquired thrombophilia markers. CONCLUSIONS: Our results indicate that thromboembolic events are uncommon, despite the presence of risk factors. However, as these disorders are very rare and the percentage of nonresponder patients is very low, further investigations are needed to better understand whether IVIg alone is able to trigger these fatal events in blistering disorders.

Macroscopic classification of superficial neoplastic lesions of the oral mucosa: a preliminary study.

BACKGROUND: Careful endoscopic classification of macroscopic appearance of superficial neoplasias of the gastrointestinal tract is essential in the early detection and appropriate treatment of cancer. It has contributed to introduce minimally invasive endoscopic surgery and has significantly improved survival of patients with colon and gastric cancers. Squamous cell carcinoma of the oral cavity has been characterized, in past and present times, by poor prognosis and lack of progress in treatment outcome. Failure in diagnosing oral malignancy at an early stage is a major culprit, and the lack of a worldwide adopted classification of its macroscopic appearance, similar to that of gastrointestinal neoplasias, may have contributed to it. AIM: To test the hypothesis that is possible to extend the diagnostic benefit of a classification based upon the macroscopic appearance of superficial tumours of the digestive tract to superficial carcinoma of the oral mucosa. METHODS: We retrospectively examined a group of patients who developed multiple intraepithelial and early invasive malignant oral neoplasias, applying a modified version of the Paris classification of superficial GI neoplastic lesions. RESULTS: One hundred and ten out of 116 (94.9%) of the study lesions had morphological features that accorded with the prepared classification. CONCLUSIONS: These preliminary data suggest that most superficial early neoplastic lesions of the oral cavity can be easily detected using a well-defined classification system similar to that employed in the diagnosis of early gastrointestinal malignancy.