Daniela Adamo

Anxiety, depression, and pain in burning mouth syndrome: first chicken or egg?

Background.— Burning mouth syndrome (BMS) is an idiopathic and chronic pain condition for which patients may experience high levels of pain, anxiety, and depression. So far, it has not yet been well investigated whether specific psychiatric features (anxious traits, personality disorder, or somatization) may play a role in the BMS pathogenesis or whether some BMS symptoms, or BMS itself, may cause secondary psychiatric symptoms.

Effectiveness of small-volume, intralesional, delayed-release triamcinolone injections in orofacial granulomatosis: A pilot study

BACKGROUND Orofacial granulomatosis (OFG) is an idiopathic disorder characterized by chronic orofacial swellings causing significant cosmetic and functional problems. Treatment with high-volume triamcinolone injections has been shown to be effective but requires nerve block anesthesia and causes a dramatic temporary increase of lip swelling. OBJECTIVE We have performed a noncomparative open-label pilot study in 7 patients with OFG in order to evaluate the effectiveness of small volumes of extended-release high-concentrate triamcinolone injections in reducing lip swelling and preventing recurrences. METHODS Seven patients with OFG were studied. Small-volume, intralesional, high-concentrate, extended-release triamcinolone was injected on the basis of a weekly schedule. A standard cycle consisted of 2 or 3 injection sessions over 14 or 21 days, depending on the clinical response. RESULTS After cycle completion, all patients remained without recurrences or with cosmetically acceptable slight lip enlargement for a mean time of 19 months (range, 8-30 months). No side effects were observed, except in one patient with hypopigmentation of the skin of the upper lip. CONCLUSIONS Slow-volume, intralesional, high-concentrate, extended-release triamcinolone injections appear to be effective in reducing lip enlargement in patients with OFG and do not require nerve block anesthesia or cause a temporary troublesome increase of swelling. A long disease-free period is generally obtained.

Mucormycosis in immunocompetent patients: a case-series of patients with maxillary sinus involvement and a critical review of the literature.

OBJECTIVES To review the current literature on mucormycosis in immunocompentent/otherwise healthy individuals, to which five new cases with maxillary sinus involvement have been added. METHODS We searched in the PudMed database all articles in the English language related to human infections caused by fungi of the order Mucorales, in immunocompetent/otherwise healthy patients, starting from January 1978 to June 2009. In addition, we updated the literature by reporting five new cases diagnosed and treated at the oral medicine unit of our institution. RESULTS The literature review showed at least 126 articles published from 35 different countries in the world, to a total of 212 patients described. The most affected country was India with 94 (44.3%) patients and the most representative clinical form was the cutaneous/subcutaneous with 90 (42.5%) patients. Our five immunocompetent patients with a diagnosed infection of Mucorales localized at the maxillary sinus completely healed with lyposomial amphotericin B. CONCLUSIONS The literature analysis revealed that even in immunocompetent/otherwise healthy individuals mucormycosis infection has a worldwide distribution. What might be the real predisposing factors involved in its pathogenesis in such patients and the real causes of this peculiar geographic distribution still remains unknown. It is likely that, in our cases, a chronic insult of a well-defined and localized body area might have resulted in a local immunocompromission, thus fostering the development of an invasive fungal infection.

Sleep disturbance in patients with burning mouth syndrome: a case-control study.

AIM To examine sleep complaints in patients with burning mouth syndrome (BMS) and the relationships between these disturbances, negative mood, and pain. METHODS Fifty BMS patients were compared with an equal number of healthy controls matched for age, sex, and educational level. The Pittsburgh Sleep Quality Index (PSQI), the Epworth Sleepiness Scale (ESS), the Hamilton Rating Scales for Depression (HAM-D) and Anxiety (HAM-A) were administered. Descriptive statistics, including the Mann-Whitney U test and hierarchical multiple linear regression analyses were used. RESULTS BMS patients had higher scores in all items of the PSQI and ESS than the healthy controls (P < .001). In the BMS patients, a depressed mood and anxiety correlated positively with sleep disturbances. The Pearson correlations were 0.68 for PSQI vs HAM-D (P < .001) and 0.63 for PSQI vs HAM-A (P < .001). CONCLUSION BMS patients reported a greater degree of sleep disorders, anxiety, and depression as compared with controls. Sleep disorders could influence quality of life of BMS patients and could be a possible treatment target.

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.

Burning Mouth Syndrome Responsive to Duloxetine: A Case Report

INTRODUCTION Burning mouth syndrome (BMS) is a chronic, idiopathic, intraoral mucosal pain condition in the absence of specific oral lesions and systemic disease. Among evidence-based pharmacological treatments for this disorder, topical and systemic clonazepam, levosulpiride, selective serotonin reuptake inhibitors have been used with partial results. CASE We report a case of a 65-year-old otherwise healthy woman with a 3-year history of oral burning. Clinical and laboratory evaluations allowed us to make a diagnosis of burning mouth syndrome. She was treated with duloxetine (60 mg p.o. qd), a selective serotonin, and norepinephrine reuptake inhibitor, obtaining a complete remission of symptoms, evaluated via standardized clinical rating scales, and an improvement of her quality of life and level of functioning. DISCUSSION The pathogenesis of BMS still remains unclear. Recently, it has been suggested an underlying neuropathic mechanism, demonstrating a dysfunction in the trigeminal nociceptive pathways at peripheral and/or central nervous system level. The rationale behind the administration of duloxetine resides in its central mechanism of action, and analgesic effects previously demonstrated in diabetic peripheral neuropathy, and fibromyalgia. Also, it has been shown to reduce painful physical symptoms associated with depression. CONCLUSION We hypothesize that duloxetine might represent a useful, effective, and additional therapeutic option in the treatment of BMS.

Extrahepatic manifestations of Hepatitis C virus infection: the slowly unraveling picture of oral lichen planus

To the Editor: Hepatitis C virus (HCV) infection is associated with various extrahepatic manifestations. Nevertheless, although several epidemiological arguments suggest that HCV could be implicated in their pathogenesis, existing studies have not been able to determine whether, and by which mechanisms, it plays a direct causal role. With regard to oral lichen planus (OLP), many studies performed mainly in Italy, Japan, Spain have described a strong epidemiological association with HCV infection. On the contrary other reports from different geographic areas, as well as some from the same countries, have found no significant correlation between these conditions. Thus, controversies still exist as to whether HCV infection can have a role in OLP pathogenesis. However, new epidemiological data on the real prevalence of HCV infection in Southern Italy are now available [1–4]. In this context, we have compared the epidemiological features of HCV infection in 600 OLP patients (390 women and 210 men; mean age 52.6 years, range 18–83 years) with the prevalence of HCV infection in general population in order to extrapolate new results towards the association with OLP. All cases had characteristic oral lichenoid lesions and underwent oral biopsy that confirmed the diagnosis. No patient, on the basis of histopathological features and clinical data, was suspected of having drugor restoration-related lichenoid reactions. Subject were screened for the presence of anti-HCV by a second generation enzyme-linked immunosorbent assay (ELISA II; Orto Diagnostic and Chiron Corp., Emerville CA, USA) according to manufacturer’s instructions. Anti-HCV immunoreactivity by enzyme immunoassay was confirmed by a supplemental second-generation recombinant immunoblot assay (RIBA 2; Orto Diagnostic and Chiron Corp., Emerville CA, USA). Differences in proportion between OLP patients and the control group were evaluated using the chi-squared test. A P-value of ,0.05 was considered significant. Of the total of 600 OLP patients studied, 169 tested anti-HCV reactive with ELISA II; among these, 165 tested positive with RIBA and four had indeterminate results. Thus, the overall anti-HCV ELISA II-positive RIBAconfirmed prevalence was 27.5% (165 of 600 subjects). Categorizing OLP patients in five age groups, the prevalence was 0.0% (0/3) in subjects younger than 30 years, 4.6% (3/65) in 30–39 years age group, 12.5% (15/120) in 40–49 years age group, 27.5% (55/200) in 50–59 years age group and 43.3% (92/212) in those older than 60 years. With regard to the control group, it is important to notice that, in recent years, the real prevalence of HCV infection in the general population of Southern Italy has been newly assessed by three studies performed in the Calabria, Campania and Apulia regions over more than 3800 subjects on the whole [2–4]. They have evidenced one of the highest overall anti-HCV prevalence rates compared with figures from other countries, reporting that the average prevalence of HCV infection in this area ranges from 12.6 to 26.0% with a clear age-distribution. In fact, the age-specific prevalence of HCV infection in Southern Italy ranges from 0 to 1.3% in subjects younger than 30 years, from 2.3 to 10.9% in 30–39 years age group, from 5.0 to 25.9% in 40–49 years age group, from 18.4 to 34.5% in 50–59 years age group and from 33.1 to 42.1% in those older than 60 years. The average prevalence, according to the same age groups, are 1.30, 8.45, 19.9, 30.2, and 37.7%, respectively. Comparing these data with those underlined in our study over 600 OLP patients, the emerging picture is that of a strong similarity, without statistically significant differences (P 1⁄4 0:05) (Fig. 1). So, to understand whether OLP is really epidemiologically associated with HCV infection, we have to avoid the error of looking at the overall anti-HCV prevalence in the general population, and we need to compare the single age groups and the age-specific prevalence of HCV infection. What emerges from our 600 OLP patients, which is to our knowledge the biggest group of OLP patients in Italy, reflects the age-specific average prevalence of HCV infection in the general population of Southern Italy. These epidemiological findings point out a lack of evidence for an association between OLP and HCV infection in Southern Italy and seem to suggest that the data previously reported in Southern Italy and other HCV hyperendemic regions may be explained by the high prevalence of HCV infection in these areas. As consequence, our findings suggest that the reported epidemiological association between OLP and HCV infection may be merely casual and that OLP may not be categorized among extrahepatic manifestations of HCV infection, as reported in previous non-epidemiological studies. Nevertheless, we have to accept that OLP may have a different clinical behavior in patients with HCV infection and/or in those treated with interferon for hepatitis C, as we and other authors have described [5], hypothesizing a role of HCV and/or the interferon therapy in the modulation of the Journal of Hepatology 37 (2002) 412–416

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.

Oral erythema multiforme: trends and clinical findings of a large retrospective European case series.

OBJECTIVE Erythema multiforme (EM) continues to be an underestimated disease with a lack of strict classification and diagnostic criteria. We present the analysis of a case series of 60 oral EM patients from 2 centers and illustrate the range of oral clinical presentations. STUDY DESIGN Clinical data from 60 EM patients with oral involvement, diagnosed and treated between 1982 and 2014, were retrospectively collected from the archives of 2 independent hospitals. Statistical analyses of the data were performed using the Pearson χ-squared test and the Mann-Whitney U test. RESULT Thirty-one patients (51.7%) were male and 29 (48.3%) were female, with a mean (±SD) age of 37.9 years (±18.1). The frequency of previous occurrences ranged from 0 to 10 (mean ± SD: 1.4 ± 2.0). Twenty-nine patients (48%) had no previous occurrence. Medications (particularly antipyretics, food additives, and antibiotics) were the suspected precipitants in 28 patients (46.7%), whereas herpes simplex virus infection was suspected in 18 (30.0%). All but 1 patient had involvement of multiple oral sites, with the buccal mucosa being the most commonly involved oral site (75%), followed by the vermillion border (71.7%). CONCLUSIONS Patients with EM may present initially to oral health care workers. Medications and herpes simplex virus continue to be the most typically involved precipitating factors. Our data highlight the additional role of food-derived antigens. Although laboratory tests can provide support diagnostically, EM diagnosis continues to be based on clinical features. A medication and food diary should be encouraged particularly in patients with recurrent forms.

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.