Thalidomide use in the management of oromucosal disease: A 10-year review of safety and efficacy in 12 patients.

OBJECTIVE: Thalidomide is an effective systemic agent in the management of ulcerative oromucosal conditions. However, its clinical use is limited because of its known adverse effect profile, including teratogenicity, peripheral neuropathy, and thromboembolic risk. The aim of this study was to review the efficacy and safety of thalidomide over a 10-year period in an Oral Medicine specialty clinic. STUDY DESIGN: Clinical records of the Oral Medicine Department at the Royal National ENT and Eastman Dental Hospitals (London, UK) were retrospectively reviewed for patients prescribed thalidomide between 2009 and 2019 for the management of oromucosal ulceration. Twelve eligible patients were identified. Data on patient response to treatment and major/minor adverse events were obtained from their clinical and electrophysiologic records. RESULTS: A complete remission rate was noted in 50% (6 of 12) patients treated for recurrent aphthous stomatitis, HIV-related ulceration and oral Crohn disease. A thalidomide-induced neuropathy rate of 41.7% (5 of 12) was detected by electrophysiology testing, however clinical symptoms of neuropathy were only described by 3 subjects. No other major adverse effects were reported. CONCLUSIONS: Thalidomide demonstrates a good efficacy-to-safety ratio in the management of oromucosal ulceration over a prolonged treatment period. Interval electrophysiologic testing is essential to monitor for thalidomide-induced neuropathy. In this cohort, neuropathy does not appear to be a dose-dependent outcome.

World Workshop on Oral Medicine VII: Relative frequency of oral mucosal lesions in children, a scoping review.

OBJECTIVE: To detail a scoping review on the global and regional relative frequencies of oral mucosal disorders in the children based on both clinical studies and those reported from biopsy records. MATERIALS AND METHODS: A literature search was completed from 1 January 1990 to 31 December 2018 using PubMed and EMBASE. RESULTS: Twenty clinical studies (sample size: 85,976) and 34 studies from biopsy services (40,522 biopsies) were included. Clinically, the most frequent conditions were aphthous ulcerations (1.82%), trauma-associated lesions (1.33%) and herpes simplex virus (HSV)-associated lesions (1.33%). Overall, the most commonly biopsied lesions were mucoceles (17.12%), fibrous lesions (9.06%) and pyogenic granuloma (4.87%). By WHO geographic region, the pooled relative frequencies of the most common oral lesions were similar between regions in both clinical and biopsy studies. Across regions, geographic tongue (migratory glossitis), HSV lesions, fissured tongue and trauma-associated ulcers were the most commonly reported paediatric oral mucosal lesions in clinical studies, while mucoceles, fibrous lesions and pyogenic granuloma were the most commonly biopsied lesions. CONCLUSIONS: The scoping review suggests data from the clinical studies and biopsy records shared similarities in the most commonly observed mucosal lesions in children across regions. In addition, the majority of lesions were benign in nature.

Bisphosphonate-related osteonecrosis of the jaws and its relevance to children--a review.

Bisphosphonate-related osteonecrosis of the jaws (BRONJ) has been detailed extensively in adults, but to date, there have been no similar cases in children. Members of the dental team may treat children prescribed bisphosphonate therapy often for management of osteogenesis imperfecta (OI). There is uncertainty as to how best treat this patient group. This review explores the background of bisphosphonates, indications for their prescription in children, adverse effects with special emphasis on BRONJ, and protocols available to guide dental management.

Orofacial granulomatosis: clinical features and long-term outcome of therapy.

BACKGROUND: Orofacial granulomatosis (OFG) is a chronic inflammatory disorder characterized by persistent or recurrent soft tissue enlargement, oral ulceration, and a variety of other orofacial features. There remain few detailed reports of the clinical features and long-term response to therapy of substantial groups of patients with OFG. OBJECTIVE: The aim of this study was to determine retrospectively the clinical, hematologic, and histopathological features of a large case series of patients with OFG. In addition the long-term response to therapy was examined. METHODS: Clinically relevant data of 49 patients with OFG who attended a single oral medicine unit in the United Kingdom were retrospectively examined. The analyzed parameters included diagnostic features, clinical manifestations, and outcomes and adverse side effects of therapy. RESULTS: Labial swelling was the most common presenting clinical feature at diagnosis (75.5%), followed by intraoral mucosal features other than ulceration such as cobblestoning and gingival enlargement (73.5%). Mucosal ulceration was observed in 36.7% of patients whereas extraoral facial manifestations such as cutaneous erythema and swelling were present in 40.8% of patients. Of the 45 patients who required treatment, 24 (53.3%) were treated with topical corticosteroids/immunosuppressants only, whereas 21 (46.7%) received a combined therapy (topical plus systemic corticosteroids/immunosuppressants and/or intralesional corticosteroids). The long-term outcome analysis showed complete/partial resolution of tissue swelling and oral ulceration in 78.8% and 70% of patients, respectively. LIMITATIONS: The main limitation of the current study was its retrospective design and methodology including differences in reporting clinical features and outcome. CONCLUSIONS: OFG can show multiple facial and mucosal clinical features. Long-term treatment with topical and/or combined therapy is needed in the majority of patients. Response to therapy is highly variable even though in the long-term complete/partial disease resolution can be obtained in the majority of patients. Mucosal ulceration tends to be more recalcitrant than orofacial swelling. Adverse side effects of therapy are rare.

Management of salivary hypofunction during and after radiotherapy.

Salivary hypofunction, the most common complication of high-dose radiation therapy (RT) to the head and neck, has a significant impact on quality of life, and requires careful planning of long-term dental and oral care. This report documents the results and conclusions of an evidence-based literature review on multidisciplinary team management of salivary hypofunction during and after RT. An update is provided on the pathophysiology of salivary hypofunction during and after RT, and recommendations for clinical management. The paper presents aspects managed by dental professionals (use of cholinergic agonists and other saliva stimulants, prevention of hyposalivation-induced rampant caries, and use of saliva substitutes), as well as the role of the radiation oncologist in minimizing salivary gland damage (parotid-sparing RT; cytoprotectants). This summary includes basic science, translational and clinical research topics with respect to radiation-induced salivary hypofunction, and provides an evidence-based management algorithm.

Long-term efficacy and safety of topical tacrolimus in the management of ulcerative/erosive oral lichen planus.

The long-term safety and clinical benefit of topical tacrolimus for the management of erosive or ulcerative oral lichen planus has not been evaluated. 50 adults (39 female 11 male; group median age 59, range 29-88 years) with symptomatic, erosive or ulcerative lichen planus recalcitrant to topical corticosteroids applied 0.1% topical tacrolimus ointment twice daily to symptomatic mucosal lesions. Topical tacrolimus was applied for a median time of 19.8 months (range 2-39 months) in this patient group. Fourteen percent of the patients had complete resolution of ulcers or erosions, 80% partial resolution and 6% reported no clinical benefit. The most common adverse effects were a burning sensation (16%) at the site of application and transient taste disturbance (8%). No significant, long-standing changes in hepatic or renal biochemistry were observed. The mean tacrolimus level decreased with duration of therapy from 2.7 microg/l (week 1) to 0.5 microg/l (week 32). 0.1% topical tacrolimus is an effective means of controlling the symptoms and signs of erosive or ulcerative oral lichen planus and has no notable adverse effects over a mean duration of application of 19.8 months.

Oral pemphigus vulgaris associated with HIV infection.

Vesiculobullous disease in association with HIV infection is extremely rare. This report details the presentation and management of pemphigus vulgaris in an individual with HIV infection. The clinical characteristics and response to therapy appear not to be modified by coexistent HIV infection. However, the management of pemphigus vulgaris relies on immunosuppressive therapy potentially resulting in HIV disease progression. Cyclosporine has in vitro antiretroviral activity and, currently, should be considered as a possible treatment for individuals with pemphigus vulgaris and HIV infection.

Topical tacrolimus: a novel therapeutic intervention for recalcitrant labial pemphigus vulgaris.

We report a case of a persistent lip ulcer in a patient with mucocutaneous pemphigus vulgaris, recalcitrant to various topical and systemic corticosteroids and immunosuppressants, which resolved following the administration of topical tacrolimus.

Mixed patterns of transmission of human herpesvirus-8 (Kaposi's sarcoma-associated herpesvirus) in Malawian families.

To study transmission patterns of human herpesvirus-8 (HHV-8) (Kaposi's sarcoma-associated herpesvirus) in families in Malawi, nucleotide sequences derived from two hypervariable loci of the HHV-8 genome, the V1 and V2 regions of open reading frame K1 (K1/V1 and K1/V2, respectively), were amplified from blood and mouth rinse samples of 22 patients with treated and untreated Kaposi's sarcoma (KS) and their first-degree relatives (n=67). In patients with KS, vincristine therapy was significantly associated with non-detectability of circulating, but not oral, K1/V1 DNA. Intra-familial K1/V1 phylogenetic comparisons of eight families were possible. Both identical and non-identical sequences were observed between family members, suggesting transmission of HHV-8 along both intra- and extra-familial transmission routes.