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.

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.

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 Syphilis: a retrospective analysis of 12 cases and a review of the literature.

OBJECTIVE: To present a retrospective analysis of multicentre case series of oral syphilis and a review of relevant literature. SUBJECTS AND METHODS: A PUBMED search was carried out from 1950 to 2011. Clinical records of patients with exclusive/prevalent oral manifestations of syphilis were collected and examined in three independent hospitals. RESULTS: Of 23 reports describing 34 patients were detected through the review (35% primary, 56% secondary, and 9% tertiary disease), describing unspecific ulcers (59%), mucosal patches (23%), keratosis (6%), pseudomembranes (3%), and gumma (9%). Multicentre case series revealed 12 patients with oral syphilis, of which 17%, 58%, and 25% with, respectively, primary, secondary, and tertiary lesions. Clinically, patients showed white patches (17%), blistering mucositis (8%), chronic unspecific ulcers with/without skin lesions (50%), gumma (17%), and necrosis of the dorsum of the tongue (8%). Oral bullae and tongue necrosis are never described in the current review. CONCLUSIONS: Diagnosis of syphilis remains a challenge because of the multiform and polymorphous clinical pattern at onset and its ability to imitate different diseases. It is mandatory to include syphilis in the differential diagnosis of unusual oral lesions. Diagnosis of oral lesions of syphilis is often difficult, and biopsy is required in controversial cases.

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.

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.

The role of upper endoscopy in identifying oesophageal involvement in patients with oral pemphigus vulgaris.

BACKGROUND AND AIMS: The involvement of oesophagus in pemphigus vulgaris is still debated. The aims of this study were to evaluate the prevalence of oesophageal involvement and the gastro-duodenal mucosa appearance before and after high-dose corticosteroid therapy in a group of patients with oral pemphigus vulgaris. METHODS: We prospectively studied 28 consecutive patients with oral pemphigus by oesophageal symptom standardised questionnaire, upper gastro-intestinal endoscopy, exfoliative cytology and histological biopsy. After clinical remission, all patients underwent new endoscopy. RESULTS: The prevalence of oesophageal symptoms was 57.1%. Endoscopic examination revealed oesophageal involvement with different degrees of severity in 67.8% of patients. After corticosteroid therapy, endoscopy showed normal oesophageal-gastro-duodenal mucosa. No examination-related exacerbations of the oesophageal lesions were seen. CONCLUSIONS: The upper gastro-intestinal endoscopic examination, in oral pemphigus vulgaris patients with oesophageal symptoms, is safe in skilled hands technique and a useful diagnostic tool prior to starting therapy.

Clinical behaviour of malignant transforming oral lichen planus.

AIMS: At present oral lichen planus (OLP) is classified among precancerous conditions but very few data are available in literature regarding prognosis of OLP-related cancers. The aim of this paper is to evaluate clinical long-term behaviour of OLP-related oral squamous cell carcinomas (OSCCs). METHODS: Clinical history and data from follow-up regarding 21 patients undergoing malignant transformation of OLP have been critically revised. RESULTS: In a mean time of 2.6 years from diagnosis of OLP, patients developed OSCCs. Histopathologically, two carinomas showed a moderate degree of differentiation, while the others were well differentiated. Six (28.5%) were in situ OSCC, in eight (30%) was found a microinvasive pattern of infiltration, one was a stage II tumour and the remaining (6 cases) were stage I tumours. During follow up, four patients (19%) have developed another OSCC in a mean time of 11 months from the first occurrence. In two men (10% of cases), multiple OSCCs occurred and in five cases (24%) lymph nodal metastases were detected. CONCLUSIONS: Our study points out an alarming tendency in developing second primary metachronous tumours (33.3%) of the oral cavity and nodal metastases (23.8%), especially if we consider their early stages. Thus we always recommend a strict follow-up of OLP patients with clinical oral and neck examination every 2 months for 6-9 months since OSCC diagnosis is made and, after, three times a year.

Costs and effectiveness in the care of patients with oral and pharyngeal cancer: analysis of a paradox.

Oropharyngeal cancer is estimated to be the ninth most common cancer worldwide. Its prognosis is largely dependent upon tumour-stage at the time of diagnosis. Stage I and II oropharyngeal cancers are characterized by a 5-year survival rate ranging from 70% to 90%, and the management of these early carcinomas is usually of short duration, easy and very cost-effective. On the other hand, the diagnostic evaluation, treatment and management of complications and recurrences of advanced stage oral tumours (stage III and IV) are often very long, complex and costly. They also have very poor prognosis with survival figures dropping to about 20%. Nowadays, most oropharyngeal cancers are detected at a late stage with an overall 5-year survival rate of around 45-50%, and with a conspicuous increase in treatment costs and a worsening of prognosis. Even if formal and comprehensive cost-effectiveness and cost-benefit analyses are not currently available in the oropharyngeal cancer literature, it seems clear that, in the care of these patients, the enormous consumption of resources is not associated with acceptable outcomes. New initiatives should be evaluated, planned and developed for the care of patients with oral and pharyngeal cancer. These strategies should be directed at prevention and early diagnosis in order to increase patient survival and quality of life and decrease the consumption of health care resources.

Clinical guidelines in early detection of oral squamous cell carcinoma arising in oral lichen planus: a 5-year experience.

In recent years, studies on the malignant potential of oral lichen planus (OLP) provided clinical evidence that patients affected by OLP have an increased risk to develop oral squamous cell carcinoma (OSCC); nevertheless, controversies still exist as to whether OLP has inherent predisposition to become malignant, or not. We believe extremely careful management of OLP patient is mandatory, and the aim of this paper is to illustrate our clinical guidelines in evaluating the possible risk of transformation in OLP lesions. Five-hundred and two patients (311 women and 191 men) affected by OLP regularly undergo follow-up examination in our Department. Patients' ages range from 18 to 83 years, with a mean of 55.4 years (57.5 for women and 53.9 for men); minimal follow-up period is 4 months, with a maximum of 12 years. In our group of OLP patients in the past 5 years we detected 24 carcinomas: excluding three cases in which diagnoses of OLP and OSCC were synchronous and three patients who had a history of tobacco use, thus possible malignant transformation of OLP would appear to be 3.7%. Clinical criteria used in our follow-up allowed us to detect 28.5% of tumours as in situ OSCC, 38% as microinvasive OSCC, 28.5% as stage I OSCC and 4.7% as stage II OSCC, with a remarkable improvement in prognosis compared to our previous study in which we adopted different criteria.

Metastases in small thickness oral squamous-cell carcinoma arising in oral lichen planus.

Oral lichen planus (OLP) is classified among precancerous conditions, as it is considered a generalized state associated with a significantly increased risk of cancer. The objective of this study was to discuss ultra-structural aspects of OLP that could play a role in enhancing metastatic potential, thus worsening the prognosis in oral squamous-cell carcinoma (OSCC). We report four cases of microinvasive OSCC which have occurred in OLP patients. All of them were stage I tumors, with a mean thickness of 1.75 mm. Recent studies indicate a tumor thickness over 4 mm as predictive of nodal metastases, but within 6 mo, our four patients with OSCC arising from OLP developed lymphnodal metastases. Our findings suggest that OLP-related OSCC may have a worse prognosis because of increased metastatic potential; obviously, further investigation is required, but this preliminary evidence emphasizes that extremely careful management of OLP patients is mandatory, and in cases of OSCC arising from OLP, a more radical treatment is probably required.

Oral pemphigus: long term behaviour and clinical response to treatment with deflazacort in sixteen cases.

Systemic corticosteroids remain the mainstay of therapy for pemphigus. Their use has transformed what was almost invariably a fatal illness into one whose mortality is now below 10%. Unfortunately, the high doses and prolonged administration of corticosteroids that are often needed to control the disease result in numerous side effects, many of which are serious or even life-threatening. Sixteen patients affected by oral pemphigus vulgaris were retrospectively examined to illustrate the natural course of the disease and to describe the efficacy of the treatment we utilised. Deflazacort, used with azathioprine, is the steroid of first choice in our therapeutic protocols, while cyclophosphamide and methylprednisolone "pulse therapy" are reserved for cases unresponsive to high doses of oral corticosteroids. In addition, the literature on oral pemphigus vulgaris was reviewed with respect to clinical history, signs and symptoms, management, and treatment outcome.

Oral lichen planus: different clinical features in HCV-positive and HCV-negative patients.

BACKGROUND: Hepatitis C virus (HCV) infection induces variable dermatologic manifestations. OBJECTIVE: To determine whether differences exist in the clinical features and behavior of oral lichen planus (OLP) between HCV-positive (HCV+ve) and HCV-negative (HCV-ve) patients. METHODS: Two hundred and sixty three patients (156 women and 107 men), with a mean age of 55.5 years, with OLP (76 HCV+ve and 187 HCV-ve) were clinically evaluated. Previously, all local factors that could modify the clinical characteristics were removed and were monitored carefully following morphology. RESULTS: In both groups, the prevalent clinical form of OLP was the mixed form (33.1% in HCV-ve and 35.5% in HCV+ve patients), in which reticular-plaque lesions coexist with atrophic-erosive ones. The reticular form was more frequent in HCV+ve (25%) than in HCV-ve (18. 7%) patients, whereas plaque lesions were more prevalent in HCV-ve (15.5%) than in HCV+ve (5.2%) patients (P < 0.01, chi-squared test). There were no significant differences in the frequency of erosive (27.2% in HCV-ve and 27.6% in HCV+ve) and atrophic (5.3% in HCV-ve and 5.2% in HCV+ve) forms between the two groups. CONCLUSIONS: Our findings show that there were statistically significant differences between OLP-HCV-ve and OLP-HCV+ve groups for reticular and plaque clinical forms. These findings underline the importance of liver examination in all OLP patients, including cases with mild, asymptomatic keratotic forms of the disease.

Oral tuberculosis: a clinical evaluation of 42 cases.

OBJECTIVES: A retrospective review of a large series of oro-facial cases of tuberculosis to analyse clinical, histopathological, and radiological aspects, as well as those of chemotherapy. MATERIALS AND METHODS: A total of 42 cases of tuberculosis of the oro-facial region were examined. Thirteen patients had a primary form and 29 a secondary form of the disease. Diagnosis was based on careful clinical examination, Mantoux reaction, histopathological examination, microbiological cultures and immunological investigation with the detection of antibodies against Mycobacteria in the patients' serum (ELISA). RESULTS: Cases examined consisted of 27 males and 15 females. The age range was 3 to 73 years (mean age 31 years). Clinical manifestations comprised oral ulcers in 69.1%, bone involvement in 21.4%, and salivary gland and/or lymph node involvement in 14.3%. A total of 79.4% patients with secondary disease had pulmonary lesions, 15 of whom showed clinical and radiological signs of activity; there was one case of bilateral renal lesions and two of skin lesions. CONCLUSIONS: Oro-facial tuberculosis is often difficult to diagnose and it should be an important consideration in the differential diagnosis of lesions that appear in the oral cavity. The most important diagnostic tools remain a careful clinical evaluation, biopsy for histologic study, as well as acid-fast stains, culture, and immunological assays, and skin testing.

Oral manifestations of idiopathic lenticular mucocutaneous pigmentation (Laugier-Hunziker syndrome): a clinical, histopathological and ultrastructural review of 12 cases.

OBJECTIVE: To present a clinical, histopathological and ultrastructural study on a group of patients affected by idiopathic mucocutaneous pigmentation (Laugier-Hunziker syndrome: LHS). MATERIALS AND METHODS: Twelve patients were investigated: clinical examination, laboratory tests, and X-ray studies together with light microscopy and electron microscopy were performed in order to diagnose LHS. RESULTS: All cases showed acquired, benign, macular hyperpigmentation of buccal mucosa lips and nails. Histologically, pigmentations are due to an accumulation of melanin in the basal layer keratinocytes and an increase in the number of melanophages in the submucosa and/or papillary dermis. Ultrastructurally there were increased numbers of normal-appearing melanosomes in keratinocytes of the lower epithelium. No evidence of malignant changes were detected. CONCLUSIONS: The importance of this condition relates to it being included in the differential diagnoses of pigmentary disorders of the oral mucosa with associated nail involvement. It is important to recognize this acquired benign disorder to avoid unnecessary investigations and treatments.