ABSTRACT
OBJECTIVES: The underlying mechanisms of burning mouth syndrome (BMS) remain unclear leading to challenges and unsatisfactory management. Current treatments focus primarily on symptom relief, with few consistently achieving a 50% reduction in pain. This review aims to explore animal models of BMS to gain a better understanding of the underlying mechanisms and to discuss potential and existing knowledge gaps. METHODS: A comprehensive review of PubMed®, Google Scholar, and Scopus was performed to assess advances and significant gaps of existing rodent models that mimic BMS-related symptoms. RESULTS: Rodent models of BMS involve reproduction of dry-tongue, chorda tympani transection, or overexpression of artemin protein. Existing preclinical models tend to highlight one specific etiopathogenesis and often overlook sex- and hormone-specific factors. CONCLUSION: Combining aspects from various BMS models could prove beneficial in developing comprehensive experimental designs and outcomes encompassing the multifaceted nature of BMS.
Subject(s)
Burning Mouth Syndrome , Disease Models, Animal , Burning Mouth Syndrome/therapy , Burning Mouth Syndrome/physiopathology , Burning Mouth Syndrome/etiology , Animals , Humans , RatsABSTRACT
Burning mouth, also referred to as oral dysesthesia, is an underreported condition among cancer patients that may represent an early symptom of cancer or an adverse effect of treatment. This review sought to characterize this symptom in oncology care where burning symptoms may occur. A systematic review of the literature was performed based on the PRISMA statement, and the protocol was registered at PROSPERO database. A structured search was done using eight databases. The process of study selection was conducted in two distinct phases. The JBI Critical Appraisal Tools were utilized to evaluate the risk of bias in the studies included. Of the total number of studies assessed, sixteen met the eligibility criteria. Of these studies included, 7 were case reports, 7 cross-sectional studies, and 2 non-randomized clinical trials. Most studies presented low risk of bias (n = 9), while the remaining studies were evaluated and scored as moderate (n = 5) or high (n = 2) risk of bias. Burning mouth was reported as a first symptom of cancer in three studies, and as an adverse event of radiotherapy (n = 2), chemoradiotherapy (n = 2), and chemotherapy (n = 9). Burning mouth was a first symptom in 0.62% of oral squamous cell carcinoma (OSCC), and 3.3% of patients with pain as chief complaint. Oral dysesthesia prevalence was 13.6% in patients experiencing chemotherapy-induced oral adverse events. The symptom of burning mouth should be examined in oncology care, as it may be underreported and therefore undertreated. New therapies may be related to a higher risk of oral burning and studies assessing approach to management are needed. Current management borrows from the current management of burning mouth in the non-cancer setting.
Subject(s)
Burning Mouth Syndrome , Neoplasms , Humans , Burning Mouth Syndrome/etiology , Burning Mouth Syndrome/therapy , Neoplasms/therapy , Neoplasms/complications , Antineoplastic Agents/adverse effects , Antineoplastic Agents/administration & dosageABSTRACT
Oral dysaesthesia is a condition characterised by persistent alteration to oral sensation, perceived by the patient to be abnormal and/or unpleasant, in the absence of any mucosal pathology. The condition can be difficult to detect and diagnose. A possible peripheral or central neuropathic aetiology has been proposed. Burning mouth syndrome (BMS) is the most common idiopathic oral dysesthesia in which long-term suffering is often reported by patients. Recent efforts from professional organisations and study groups have provided a consensus on BMS disease definition and diagnostic criteria. Large-scale epidemiological studies are required to provide an accurate estimate for prevalence and incidence of the condition. Meticulous diagnostic investigations which may require interdisciplinary teamwork are often warranted to reach an accurate diagnosis. A combination of interventional modalities, with a holistic approach, is key for successful management and improvement in patients' quality of life.
Subject(s)
Burning Mouth Syndrome , Paresthesia , Humans , Paresthesia/complications , Burning Mouth Syndrome/diagnosis , Burning Mouth Syndrome/etiology , Burning Mouth Syndrome/therapy , Quality of LifeABSTRACT
OBJECTIVES: To study the diagnostic value of salivary pepsin tests for detecting laryngopharyngeal reflux (LPR) in patients with primary burning mouth syndrome (BMS). METHODS: Patients with BMS and asymptomatic individuals were consecutively recruited from September 2018 to June 2023. Patients underwent hypopharyngeal-esophageal impedance pH-monitoring (HEMII-pH) and saliva collections to measure pepsin. Stomatology evaluation was carried out to exclude other causes of BMS. Oral, pharyngeal and laryngeal signs and symptoms were evaluated with Reflux Sign Assessment (RSA) and Reflux Symptom Score (RSS). Sensitivity, specificity, positive (PPV) and negative (NPV) predictive values of pepsin test were calculated considering the highest values of pepsin tests at ≥ 16, ≥ 36, and ≥ 100 ng/mL cutoffs. Receiver operating characteristic curve (ROC) was evaluated. RESULTS: Forty-nine patients with both BMS and LPR at the HEMII-pH and 21 asymptomatic individuals were recruited. Pepsin test was 83.7%, 79.6%, and 71.4% sensitive at cutoffs ≥ 16, ≥ 36, and ≥ 100 ng/mL, respectively. The ROC analysis reported that a threshold of ≥ 21.5 ng/mL was associated with sensitivity, specificity, PPV and NPV of 81.6%, 81.0%, 90.1% and 65.4%, respectively. The severity score of burning mouth symptom was significantly associated with the saliva pepsin concentration (rs = 0.263; p = 0.029) and the oral RSA (rs = 0.474; p = 0.007). CONCLUSION: Pepsin test is a valuable diagnostic approach for detecting LPR in patients with BMS. Patients with high level of saliva pepsin reported more severe burning mouth symptoms. Future studies are needed to confirm the role of LPR in the primary BMS.
Subject(s)
Burning Mouth Syndrome , Laryngopharyngeal Reflux , Humans , Saliva/chemistry , Pepsin A/analysis , Burning Mouth Syndrome/etiology , Burning Mouth Syndrome/complications , Prospective Studies , Esophageal pH Monitoring , Laryngopharyngeal Reflux/complications , Laryngopharyngeal Reflux/diagnosis , Electric ImpedanceABSTRACT
BACKGROUND: Burning mouth syndrome (BMS) is a chronic oral pain disorder characterized by a generalized burning sensation in the oral mucosa without apparent medical or dental causes. Despite various hypotheses proposed to explain BMS pathogenesis, a clear understanding of the cellular-level events and associated histologic and molecular findings is lacking. Advancing our understanding of BMS pathogenesis could facilitate the development of more targeted therapeutic interventions. TYPES OF STUDIES REVIEWED: The authors conducted an extensive literature search and review of cellular mechanisms, focusing on evidence-based data that support a comprehensive hypothesis for BMS pathogenesis. The authors explored novel and detailed mechanisms that may account for the characteristic features of BMS. RESULTS: The authors proposed that BMS symptoms arise from the uncontrolled activation of proapoptotic transmembrane calcium permeable channels expressed in intraoral mucosal nerve fibers. Elevated levels of reactive oxygen species or dysfunctional antiapoptosis pathways may lead to uncontrolled oxidative stress-mediated apoptosis signaling, resulting in upregulation of transmembrane transient receptor potential vanilloid type 1 and P2X 3 calcium channels in nociceptive fibers. Activation of these channels can cause nerve terminal depolarization, leading to generation of action potentials that are centrally interpreted as pain. CONCLUSIONS AND PRACTICAL IMPLICATIONS: The authors present a novel hypothesis for BMS pathogenesis, highlighting the role of proapoptotic transmembrane calcium permeable channels and oxidative stress-mediated apoptosis signaling in the development of BMS symptoms. Understanding these underlying mechanisms could provide new insights into the development of targeted therapeutic interventions for BMS. Additional research is warranted to validate this hypothesis and explore potential avenues for effective management of BMS.
Subject(s)
Burning Mouth Syndrome , Chronic Pain , Humans , Burning Mouth Syndrome/etiology , Burning Mouth Syndrome/diagnosis , Burning Mouth Syndrome/drug therapy , Calcium/metabolism , Calcium/therapeutic use , Mouth Mucosa , Oxidative StressABSTRACT
Emerging evidence has revealed a cross-talk in the etiopathogenesis of burning mouth syndrome (BMS) related to peripheral nerve fibers (NF) and neuropeptides secreted by mast cells. Here, we investigated the S-100+ density and PGP 9.5+ integrity of peripheral NF and the tryptase+ mast cell density in the oral mucosa of BMS patients and healthy individuals. A total of 23 oral mucosa specimens (12 BMS and 11 controls) were evaluated. The clinical diagnosis of BMS was based on a careful examination, excluding other local and systemic causes. Samples were taken from an incisional biopsy of the tongue mucosa of individuals with symptomatic BMS, while the margins of the non-neoplastic tongue biopsy served as controls of healthy individuals. Immunohistochemistry was performed to determine the density/mm2 of S-100+, PGP 9.5+ peripheral NF, and tryptase+ mast cells. Similar densities of S-100+, PGP 9.5+ peripheral NF, and tryptase+ mast cells were found in cases of BMS, with a median value of 3.70, 0.70, and 29.24/mm2, respectively, and in the control group, with a median value of 2.60, 0.80, and 26.01/mm2, respectively (p > 0.05). Moreover, the relationship between S100+ and PGP 9.5+ peripheral NF was the same in both groups (p = 0.70). This study demonstrated that there were no alterations in the density and integrity of peripheral NF in the tongue of symptomatic BMS patients. However, the sensitization of peripheral NF in this disease may not depend on mast cell density.
Subject(s)
Burning Mouth Syndrome , Mast Cells , Humans , Mast Cells/pathology , Burning Mouth Syndrome/diagnosis , Burning Mouth Syndrome/etiology , Burning Mouth Syndrome/pathology , Tryptases , Tongue , Peripheral Nerves/pathologyABSTRACT
BACKGROUND: Burning mouth disorder (BMD) is a complex medical condition characterized by a burning sensation in the mouth of fluctuating intensity. BMD is considered a diagnosis of exclusion, as oral burning can occur secondary to local or systemic conditions. Parkinson's disease (PD) is one such condition. OBJECTIVE: To provide a scoping review of the literature by assessing all articles written in English that investigated the relationship between BMD and PD. MATERIALS AND METHODS: Various databases (PubMed, Ovid, Web of Science, Science Direct and Scopus) and a search platform (EBSCOhost) were searched following similar investigative approaches. Duplicates were removed and reference lists of original studies were scrutinized for additional articles. Any decision about the inclusion/exclusion in the review was by consensus among the co-authors. RESULTS: Twenty-five original articles and one supplemental article were included in the final review, of which 13 met the inclusion criteria. These were further divided into five categories based on the study design/article, which included Prevalence studies (n = 6), Letter to the editor (n = 1), Incidence study (n = 1), Case reports (n = 2) and Experimental studies (n = 3). Strongest data was provided by epidemiological studies, which suggest BMD and PD are poorly associated. CONCLUSIONS: A scoping review of the existing literature does not suggest that PD patients are any more at risk of developing BMD compared to the general population. While there may be a link through the dopaminergic system as determined by imaging studies, it is unlikely that the pathogenesis of PD disease shares significant commonality with BMD.
Subject(s)
Burning Mouth Syndrome , Parkinson Disease , Humans , Parkinson Disease/complications , Burning Mouth Syndrome/etiology , Dopamine , Diagnosis, OralABSTRACT
Background: Burning mouth syndrome is an idiopathic condition characterized by burning pain in a normal-appearing oral mucosa lasting at least four to six months. In the case of secondary burning mouth syndrome is associated with local or systemic factors (such as thyroid disorders) that can cause these symptoms. The aim of this review was to study the relationship between thyroid disorders and burning mouth syndrome.Material and methods: The present study followed the PRISMA guidelines. An electronic search strategy was developed for PubMed/Medline, Scopus and Cochrane. The following combination of keywords and Boolean operators were used: Thyroid AND burning mouth; Thyroid AND burning mouth syndrome; Hypothyroidism AND burning mouth; Hypothyroidism AND burning mouth syndrome; Hyperthyroidism AND burning mouth; Hyperthyroidism AND burning mouth syndrome. The results were processed by existing free software in https://www.graphpad.com/. To evaluate the association of the categorical variables we used the Fisher test at a level of significance of p-value ≤ 0,05. As a primary summary measure the Odds Ratio (OR) has been used. To analyze the risk of bias the guidelines of the GRADE guide were used and the grade of evidence was analyzed by the guide of Joanna Briggs Institute: Levels of Evidence and Grades of Recommendations.Results: After applying the inclusion and exclusion criteria, 5 studies were selected for review. The Chi-square was 10.92 and the Odds Ratio was 3.31 with respect to TSH values with p <0.0001 (Fisher's test). The population of patients with TSH alterations is increased in 80.49% and decreased in 19.51%.Conclusions: It can be concluded that thyroid hormone abnormalities are a factor in secondary burning mouth syndrome; specially in patients with hypothyroidism. (AU)
Subject(s)
Humans , Hyperthyroidism/complications , Hyperthyroidism/diagnosis , Burning Mouth Syndrome/etiology , Thyroid Hormones , ThyrotropinABSTRACT
Burning mouth syndrome (BMS) and pelvodynia are chronic pain still poorly understood and the links between them are all the more so. Health professionals therefore have few resources to understand, diagnose and treat these pains. They may consider and treat these ailments individually, which does not represent optimal care management for the patient and leads to overmedication. This article aims to highlight their contiguity from epidemiological, etiological, diagnostic, and therapeutic perspectives. This study was based on articles which were found using databases such as PubMed and Web of Science. No exclusion criteria were used. Fourteen studies were reviewed. This present work shows that the clinical presentations of these syndromes are similar, as exemplified by their strong association with anxiety and depression. The neurophysiological mechanisms involved in these conditions are similar to those in patients. The diagnosis is essentially based on visual examination and an elimination of all other possible causes. In addition, this work promotes the fact that a common therapy can be implemented when BMS and pelvodynia co-occur. However, the literature on the subject is still very limited. This can be deepened by exploring all the effective treatments in BMS and vulvodynia for penoscrotodynia. Finally, for all these pains, there is a therapeutic order to respect starting with a psychological approach, then topical treatments, systemic therapy and surgical. This therapeutic gradient assists practitioner in their patient's pain management. This article also allows health care providers to quickly find an effective systemic treatment for a patient with both BMS and pelvodynia.
Subject(s)
Burning Mouth Syndrome , Chronic Pain , Female , Humans , Burning Mouth Syndrome/diagnosis , Burning Mouth Syndrome/etiology , Burning Mouth Syndrome/therapy , Anxiety , Anxiety Disorders/complications , Chronic Pain/complications , Pain Management/adverse effectsABSTRACT
OBJECTIVE: The International Classification of Disease defines burning mouth syndrome (BMS) as a chronic intraoral burning sensation, with no identifiable local or systemic cause. Since current management is often unsatisfactory, the aim of this report is to describe a new treatment modality (i.e., low-dose naltrexone [LDN]). STUDY DESIGN: A 62-year-old woman presented with the complaint of burning on the tongue of 3 years' duration. Existing comorbidities were fibromyalgia, irritable bowel syndrome, headache, and interstitial cystitis. Her reported pain intensity ranged from 2/10 (morning) to 8/10 (evening) on a numeric rating scale. With the diagnosis of BMS and hyposalivation, and in light of her current clonazepam use and fibromyalgia, a dry mouth protocol and LDN (3 mg) were prescribed. RESULTS: After 1 month, her pain intensity decreased by 50%, with no pain upon awakening. After 2 months, the widespread pain associated with her chronic morbidities also reduced by 50%, and her headache disappeared. After adjusting LDN dose to 4.5 mg, the patient was stable at 6 months, with 50% reduction of widespread pain and 2/10 BMS pain, and no reported side effects. CONCLUSIONS: These preliminary results suggest that LDN may be a feasible and effective treatment for BMS, especially in patients' refractory to traditional treatment.
Subject(s)
Burning Mouth Syndrome , Fibromyalgia , Humans , Female , Middle Aged , Naltrexone/therapeutic use , Burning Mouth Syndrome/drug therapy , Burning Mouth Syndrome/diagnosis , Burning Mouth Syndrome/etiology , Clonazepam/therapeutic use , Pain/complications , Pain/drug therapy , HeadacheABSTRACT
BACKGROUND: Burning mouth syndrome is an idiopathic condition characterized by burning pain in a normal-appearing oral mucosa lasting at least four to six months. In the case of secondary burning mouth syndrome is associated with local or systemic factors (such as thyroid disorders) that can cause these symptoms. The aim of this review was to study the relationship between thyroid disorders and burning mouth syndrome. MATERIAL AND METHODS: The present study followed the PRISMA guidelines. An electronic search strategy was developed for PubMed/Medline, Scopus and Cochrane. The following combination of keywords and Boolean operators were used: Thyroid AND burning mouth; Thyroid AND burning mouth syndrome; Hypothyroidism AND burning mouth; Hypothyroidism AND burning mouth syndrome; Hyperthyroidism AND burning mouth; Hyperthyroidism AND burning mouth syndrome. The results were processed by existing free software in https://www.graphpad.com/. To evaluate the association of the categorical variables we used the Fisher test at a level of significance of p-value ≤ 0,05. As a primary summary measure the Odds Ratio (OR) has been used. To analyze the risk of bias the guidelines of the GRADE guide were used and the grade of evidence was analyzed by the guide of Joanna Briggs Institute: Levels of Evidence and Grades of Recommendations. RESULTS: After applying the inclusion and exclusion criteria, 5 studies were selected for review. The Chi-square was 10.92 and the Odds Ratio was 3.31 with respect to TSH values with p <0.0001 (Fisher's test). The population of patients with TSH alterations is increased in 80.49% and decreased in 19.51%. CONCLUSIONS: It can be concluded that thyroid hormone abnormalities are a factor in secondary burning mouth syndrome; specially in patients with hypothyroidism.
Subject(s)
Burning Mouth Syndrome , Hyperthyroidism , Hypothyroidism , Humans , Burning Mouth Syndrome/etiology , Hypothyroidism/complications , Hypothyroidism/diagnosis , Thyroid Hormones , Hyperthyroidism/complications , ThyrotropinABSTRACT
Burning mouth syndrome (BMS) is a rare chronic neuropathic pain condition characterized by recurring burning pain or dysesthesia in the absence of any local or systemic causes of symptoms. The exact pathophysiology of BMS is unknown, but recent research suggests a medley of neuropathic, endocrinological, and psychosocial elements. This article presents a case history and reviews the epidemiology, diagnostic criteria, clinical features, diagnostic investigations, pathophysiology, and management of BMS.
Subject(s)
Burning Mouth Syndrome , Neuralgia , Humans , Burning Mouth Syndrome/diagnosis , Burning Mouth Syndrome/etiology , Burning Mouth Syndrome/therapy , Neuralgia/complicationsABSTRACT
BACKGROUND AND AIM: An association between reflux and burning mouth syndrome (BMS) has been proposed. Aims of this study were: 1) to investigate the frequency of BMS in a sample of GERD patients; 2) to measure G17, in a sample of BMS patients; 3) to assess the efficacy of different therapeutical schedules for GERD in BMS patients. METHODS: We divided the study in 3 main steps. In step one, we analyzed 500 consecutive GERD patients' type and frequency of extraesophageal manifestations including BMS. In step two, we collected 124 consecutive BMS patients' symptoms and G17. In step three, we evaluate the efficacy of 3 different drugs on BMS. RESULTS: In step one, 204 patients complained heartburn; 31 globus pharyngeus; 52 chronic cough; 54 pharyngitis; 31 postnasal drip; 56 burning mouth symptoms; 34 noncardiac chest pain; 17 asthma and 21 sleep apnea. In step two, 29 patients had G17 ≤ 1 pg/L; 64 patients between 1 and 3; and 31 patients ≥ 3. In step three, 49 patients reported slight benefit with PPI, 75 no benefit. 61 patients reported slight benefit with sodium alginate and sodium bicarbonate, 63 no benefit. 23 reported an almost complete remission with HYCHSA, 26 slight benefit, 33 no benefit. CONCLUSIONS: Prevalence of BMS in GERD patients was similar to that reported for chronic chough and pharyngitis. Low levels of G17 were found in the majority of BMS patients. Finally, we observed a greater benefit from barrier drugs therapy than from PPI therapy in BMS patients. (www.actabiomedica.it).
Subject(s)
Burning Mouth Syndrome , Gastroesophageal Reflux , Pharyngitis , Humans , Burning Mouth Syndrome/epidemiology , Burning Mouth Syndrome/etiology , Gastroesophageal Reflux/complications , Gastroesophageal Reflux/drug therapy , Gastroesophageal Reflux/epidemiology , Cough , Chest Pain , Pharyngitis/complicationsABSTRACT
Burning mouth syndrome (BMS) is defined as "idiopathic orofacial pain with intraoral burning or dysesthesia recurring daily for more than 2 hours per day and more than 3 months, without any identifiable causative lesions, with or without somatosensory changes" in International Classification of Orofacial Pain, 2020. Worldwide prevalence of BMS was estimated to be 1.73% in population-based studies, while female and elderly are at higher risk of BMS. The aim of this narrative review is to clarify the main etiopathogenetic factors of BMS investigated so far in the scientific literature. There is growing evidence of an important role of peripheral neuropathology in BMS, supported by immunohistochemical studies which have demonstrated a significant loss of epithelial and subepithelial nerve fibers. Other possible etiopathogenetic factors emerging from literature are laryngopharyngeal reflux and hormonal and salivary changes related to aging and menopause. Finally, the role of the oral microbiota in BMS has not yet been thoroughly investigated. Further studies are necessary to investigate the probably multifactorial etiopathogenesis of primary BMS, a pathology which has a serious impact on the quality of life of our patients, a disease we find ourselves treating without the adequate therapy and the necessary knowledge.
Subject(s)
Burning Mouth Syndrome , Aged , Burning Mouth Syndrome/diagnosis , Burning Mouth Syndrome/epidemiology , Burning Mouth Syndrome/etiology , Facial Pain/complications , Female , Humans , Quality of LifeABSTRACT
PURPOSE: To evaluate whether the prevalence of glossodynia increased among patients affected by COVID-19 compared to other hospital populations. METHODS: The i2b2 patient registry platform at the University of Florida Health Center was used to generate a count of patients using the international classification of diseases (ICD)-10 diagnosis codes from October 2015 to June 2021. Logistic regression of the aggregates was used for analysis. RESULTS: Of the patients with both glossodynia and COVID-19, 60% were females, 32% were African American, 64% were white, and 100% were adults. There were 72% females, 19% African Americans, 72% whites, and 93% adults with glossodynia only. For COVID-19 patients, 57% were females, 23% were African American, 56% whites, and 90% were adults. The odds ratio (OR) for glossodynia in the COVID-19 patients was significant (OR = 2.9; 95% CI, 1.94-4.32; P < 0.0001). CLINICAL SIGNIFICANCE: Glossodynia is significantly more common in COVID-19 patients and should be considered in the differential diagnoses among the oral complications of this infection.
Subject(s)
Burning Mouth Syndrome , COVID-19 , Glossalgia , Adult , Black or African American , Burning Mouth Syndrome/diagnosis , Burning Mouth Syndrome/epidemiology , Burning Mouth Syndrome/etiology , COVID-19/complications , Female , Glossalgia/complications , Glossalgia/diagnosis , Humans , Male , White PeopleABSTRACT
Resumen El síndrome de la boca ardiente (SBA) es una condición de dolor crónico en la cavidad oral, que se presenta mayoritariamente en mujeres de edad media. Diversas causas locales y sistémicas pueden producirlo en forma secundaria, o bien, puede representar un cuadro primario, sin etiología específica identificable. Su etiopatogenia y evolución clínica es poco comprendida. Las opciones terapéuticas son variadas y en general es necesario un tratamiento multidisciplinario. A continuación, se presenta una revisión de la literatura respecto a esta patología para difusión en nuestro medio.
Abstract Burning mouth syndrome (BMS) is a chronic pain condition of the oral cavity, which occurs more frequently in middle-aged women. It can be secondary to different local and systemic causes, or represent a primary condition, with no specific identifiable etiology. Its etiopathogenesis and clinical evolution are poorly understood. Therapeutic options are multiple and a multidisciplinary treatment is necessary. We present a review of the literature regarding BMS, to provide information relevant to our area of expertise.