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REVIEW ARTICLE
Year : 2008  |  Volume : 20  |  Issue : 4  |  Page : 129-134 Table of Contents   

Burning mouth syndrome: Clinical dilemma?


1 Department of Dentistry, Indira Gandhi Government Medical College and Hospital, Nagpur-440 018, India
2 Department of Oral Medicine and Radiology, Government Dental College and Hospital, Nagpur-440 003, India

Date of Web Publication18-Jun-2009

Correspondence Address:
Kanchan R Patil
Plot No. 52, Lane No. 05, Jawaharnagar, Manewada Road, Nagpur-440 024, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0972-1363.52825

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   Abstract 

Burning Mouth Syndrome (BMS) is a chronic orofacial burning pain condition usually in the absence of clinical and laboratory findings that affects many adults worldwide, yet its etiology and treatment remain poorly understood. Though it has been associated with numerous oral and systemic conditions, there has been no clear consensus on its etiology, pathogenesis and treatment. As a result, patients with inexplicable oral complaints are often referred from one health care professional to another without effective management having significant emotional impact on patients. As the dental profession expands its scope of care to oral medicine and geriatrics, BMS will be more effectively diagnosed and managed by these dental surgeons. Hence, they should be more involved in evaluation and management of these patients. The present article provides updated information on BMS including possible etiological factors and current treatment options, although data on the effectiveness of these treatment modalities remain limited. Recently researchers found that treatment with a familiar nutritional supplement- lipoic acid- is of remarkable benefit with minimal adverse effects. ALA (alpha-lipoic acid) may be the effective treatment modality in management of BMS.

Keywords: Atypical facial pain, burning mouth syndrome, dysgeusia, glossodynia, glossopyrosis, oral dysesthesia, stomatopyrosis


How to cite this article:
Patil KR, Sathawane R S. Burning mouth syndrome: Clinical dilemma?. J Indian Acad Oral Med Radiol 2008;20:129-34

How to cite this URL:
Patil KR, Sathawane R S. Burning mouth syndrome: Clinical dilemma?. J Indian Acad Oral Med Radiol [serial online] 2008 [cited 2019 May 25];20:129-34. Available from: http://www.jiaomr.in/text.asp?2008/20/4/129/52825


   Introduction Top


Burning Mouth Syndrome (BMS) is a chronic intra-oral burning pain disorder in which the patient complains of a burning sensation and pain, in a mouth that appears clinically normal, and which may not be accompanied by other clinical signs and laboratory findings. [1],[2],[3]

BMS, glossodynia, glossopyrosis, stomatopyrosis, or oral dysesthesia is a variant of an atypical facial pain and is usually described as a steady, continuous, though variable, typical superficial somatic pain. [4],[5] It is predominantly seen in women, mostly postmenopausal women. [4],[5],[6],[7] A characteristic of this condition is the fact that the location of the pain corresponds to areas of greatest movement, thus revealing the cause as the abrasive effect of the tissues rubbing against themselves and the teeth. The tongue and inner surface of the lips are common sites. Other mucosal surfaces are also involved. Though the palate and gingiva proper are more resistant to such irritation, localized burning pain may occur. All oral irritants like highly spiced food, hot fluids, carbonated drinks, strong coffee and tea, certain juices, mouth rinses, dentifrices, and medications increase the patient's discomfort. Due to painful mastication nutritional deficiencies may arise. Anxiety and emotional tension may induce or aggravate pain and discomfort. [5]

Symptoms of BMS include oral burning, dry mouth, thirst, dysgeusia (taste alterations), changes in eating habits, sleep disturbances, constant pain, headaches, nonspecific health problems, severe menopausal symptoms, mood changes, irritability, anxiety, depression and a decreased desire to socialize. [6],[7],[8] Typically these patients awake without pain and burning but note increasing symptoms through the day which peak by early evening. [6]

Whether treated or not, BMS may disappear as mysteriously as it arose but it can also spontaneously worsen. The resolution of symptoms in BMS subjects is variable and poorly predictable, and pharmacologic therapy fails to relieve the symptoms. [9],[10] A number of epidemiologic, clinical, laboratory, neurologic and behavioral studies have been performed with no clear consensus on the etiology, pathogenesis and treatment. [1],[2],[6],[11]


   Epidemiology Top


Based on most studies published to date oral burning appears to be most prevalent in postmenopausal women. [4],[6],[7] It has been reported in about 10 to 40% of women presenting for treatment of menopausal symptoms.[12] BMS has been estimated to affect over one million (0.71%) US adults.[7] One study performed by Basker et al . [13] mentions a frequency of 2.6 to 5.1%. In another study [14] conducted in Sweden, BMS was found in 3.9% of persons. However, Maresky et al ., [4] found the number of patients with BMS to be 35%.


   Etiologic Factors Top


Because of longstanding difficulty in understanding the pain of BMS and its complex clinical picture, a number of etiologic factors have been suggested. These factors are divided into two basic categories: local (oral) disorders and systemic conditions.


   I) Local Causes Top


The possible local causes include

Denture allergy,
Galvanic current,
Mechanical irritation,
Parafunctional habits,
Salivary dysfunction (xerostomia),
Taste disturbances,
Ulcerative or erosive lesions,
Gingivitis,
Periodontitis,
Candidiasis,
Geographic tongue, and
Peripheral nerve disease.

Denture allergy, galvanic current, mechanical irritation due to denture related problems and parafunctional habits

Allergic reaction to high residual monomer levels in acrylic denture bases has been suggested as a causative factor of BMS. [15] Though galvanic current was suggested as a cause for BMS, a few studies have found that the currents of similar intensity occur in both BMS and asymptomatic control subjects. [16],[17] Chemical irritation, allergic reaction to dental material and galvanic currents have not been found to be important causes for BMS. [18] Mechanical irritation due to ill fitting dentures and parafunctional habits may cause oral burning, [7] but this is contradicted in some studies. [6]

Salivary dysfunction (xerostomia)

Oral burning in BMS subjects may be due to dry, easily irritated oral mucosa. [4] Xerostomia (dry mouth) occurring with advancing age or menopause has been suggested as a causative agent in pathogenesis of BMS. [19] However there is no significant evidence of diminution in salivary function in post-menopausal females. [7]

However, there is evidence that the xerostomia occurs only after onset of mouth burning. [6] Some studies have demonstrated decreased output of saliva in patients with BMS compared to control subjects, [20] whereas others have documented no significant flow rate differences of stimulated or unstimulated saliva. [21],[22],[23] In contrast to flow rate, changes in composition have been detected in some BMS patients. [21],[22] The relationship between the salivary composition and BMS is not known, but it may be due to altered sympathetic output related to stress. [12] Complaint of xerostomia in subjects with BMS may not be necessarily related to salivary gland dysfunction, [24] but certain medications, [25] chemotherapy or irradiation treatment for head, neck, face cancer [26] will cause xerostomia as well as salivary gland dysfunction. Medications that induce xerostomia include certain antispasmodics, antidepressants, antipsychotics, skeletal muscle relaxants,  Parkinsonism More Details medications, antiarrhythemics, antihistamines, appetite suppressants, convulsants, anxiolytics, diuretics and antihypertensives. [27] Also included are antibiotics that may alter the oral microbial flora and allow candidial proliferation to take place with resultant mucosal irritation. [28] Xerostomia may be an early symptom of several morbid systemic conditions including polyglandular failure syndrome, autoimmune exocrinopathy such as Sjogren's syndrome, systemic lupus erythematosus, primary biliary cirrhosis and autoimmune hemolytic anemia. [29] However, systemic diseases can cause salivary gland dysfunction either primarily or secondary to drugs. [30]

Taste disturbances

Persistent taste or dysgeusia (usually bitter or metallic) or altered taste perception have been reported in BMS subjects. Studies have suggested that the possible increase in the spontaneous firing rate of certain of afferent taste fibers (e.g. bitter) or afferent inhibition of others may be responsible for such taste disturbances. [31],[32] Some studies demonstrated decrease in dysgeusia with rinsing, [33],[34] hence it could imply that alteration in composition by the addition of substances from the crevicular fluids may be responsible. [35],[36] Subjective improvement in some BMS subjects has been reported on exposure to hot and spicy foods. In contrast, subjects with burning and dysgeusia have reported decrease or abolished dysgeusia symptoms after application of topical anesthesia, whereas the burning sensation increased. These findings could suggest that the dysgeusia has peripheral origin and burning group subjects have a centrally based neuropathic condition. [7] There is an increased prevalence of 'supertasters' (persons with enhanced abilities to detect taste) among subjects with BMS which may be because of their higher density of taste buds, each of which is surrounded by a basket-like collection of pain neurons of the trigeminal nerve. [3]

Ulcerative or erosive lesions, gingivitis, periodontitis

Some studies documented a significant relationship between BMS and ulcerative or erosive lesions, gingivitis and periodontitis. [4],[18] but others have not reported significant changes in intra-oral soft or hard tissues. [6] However, any break in the continuity of the oral mucosa may manifest as a burning mouth. The mild irritation associated with gingivitis and periodontitis may be interpreted as burning mouth. Hence such BMS patients need maintenance of optimal oral hygiene. However, such clearly defined oral infection should preclude categorizing patients with BMS. [4]


   II) Systemic Conditions Top


Burning pain may be associated with systemic conditions such as

Anemias,

Nutritional disorders,
Hematological disorders,
Immunologic abnormalities,
Diabetes mellitus,
Psychiatric and psychological disorders,
Central nervous system disorders,
Neoplasms,
Polyglandular failure syndrome, Sjogren's syndrome, systemic lupus erythematosus, primary biliary cirrhosis and autoimmune hemolytic anemia, and Menopause (Hormonal changes)

Anemias, nutritional and hematological disorders, immunologic abnormalities and diabetes mellitus

Many researchers have found a higher occurrence of many "other health complaints" in BMS patients, a common phenomenon linked to "help seeking behavior" in studies of other chronic sensory disorders. [1]

Though many systemic conditions have been associated with BMS, very few provide statistical support for this interrelationship. Studies that show a statistically significant association between women with BMS, anemia, hematological and nutritional disorders state that the burning could be because the metabolism and integrity of oral mucosal lining is sensitive to these deficiencies. Hence atrophy and functional disturbances of peripheral nerve tissue can occur that may manifest as BMS. [4] Literature indicates that nutritional deficiencies including iron, B 1 , B 2 , B 6 , B 12 , zinc, folic acid are unlikely to be important causative factors for BMS. [6],[20],[37],[38] This also explains why replacement therapy was no more effective than placebo in altering symptoms of BMS. [6],[39] However, one study showed that replacement therapy of vitamins produced resolution of symptoms in 30% of BMS subjects. [40]

A study showed that 58% of BMS subjects had evidence of immunologic abnormalities and 63% had elevated ESR. [6] The possibility of mildly elevated ESR may be associated with these immunologic abnormalities which may be responsible for damage to small blood vessels, thereby resulting in ischemia, pain, and sensory nerve dysfunction. Recent findings suggest that preliminary evidence of ischemia may cause decreased dorsal tongue temperature in BMS subjects as compared to controls. [6]

It was documented long ago that many patients with BMS have high blood glucose levels, but no consistent or casual relationship has been recorded. [13] The incidence of oral burning is seen in only 2 to 10% subjects with diabetes, indicating that it may not be an important cause of BMS. [20],[38],[40] As diabetes predisposes oral candidiasis which causes mucosal irritation this may, therefore, be responsible for oral burning. [10]

Psychiatric and psychological disorders

The psychological aspect has long been recognized as a factor in BMS. [2] A few studies undertaken using a questionnaire to assess the psychological status of such patients suggested that personality and mood changes (especially anxiety, depression, and neurotic tendencies) were found in patients with BMS and hence BMS is viewed as a psychogenic problem. [2],[41] A study reported psychological factors in more than half of patients complaining of BMS, with higher incidence in patients with history of constant burning (type 2) than in patients who develop oral burning as the day goes on and is worse in the evening (type 1). Anxious patients with type 2 BMS were likely to be the most difficult to treat successfully. [2] Another study documented that BMS patients had a significantly higher depression trait (an index of severity of chronic symptoms) and anxiety state than those of control subjects, but lower than those of psychiatric patients. [42]

Central nervous system (CNS) disorders, neoplasms

A study has shown a statistically significant association between BMS subjects and CNS disturbances. [4] CNS disorders and neoplasms (apart from drugs used to treat CNS disorders which cause xerostomia) may cause changes in taste sensation and peripheral sensory nerve function that may contribute to burning sensation of the oral mucosa. [4]

Recent studies have pointed to dysfunction of several cranial nerves associated with taste sensation as a possible cause of BMS. [3]

To date the exact mechanism for BMS, perhaps neuropathic, that involves the peripheral or CNS or both systems, has not been definitely identified. Nerve injury or dysfunction resulting from oral, facial or systemic trauma or from medical conditions might be the source of oral burning. [7]

Menopause (Hormonal changes)

Age related mucosal changes are minimal in healthy women. A triad of oral symptoms - burning mouth, dry mouth and altered taste have been reported in 20-90% of perimenopausal and postmenopausal women. [6],[13],[43],[44] Of 90% postmenopausal women, the highest frequency of onset was reported from 3 years before to 12 years after menopause. [6] This finding may be the result of decreased estrogen during menopause. [43],[44]

Although some studies have demonstrated little improvement in oral symptoms after initiation of estrogen replacement therapy (ERT) and progesterone supplements, [13],[43] several studies have claimed successful treatment of these symptoms and decreased psychological factors in postmenopausal women with this approach. [37,45] The result of this success may be due to ERT's alleviation of psychological distress [37] or due to the presence of estrogen receptor protein in oral mucosa. [45] Lack of response to this form of treatment in some cases may be related to absence of estrogen and progesterone receptors in oral mucosa in some affected individuals. [45]


   Other Possible Causes Top


Many other etiologic factors such as oral candidiasis, fusospirochetal infections, benign migratory glossitis, pernicious anemia, climacteric, cancer phobia, inflammation or irritation of the lymphoid tissue in the foliate papillae of the tongue, periodontal disease, reflux esophagitis, flatulence, gingivitis, ulcerative/erosive lesions, viral infections, acoustic nerve neuroma, myofacial referred pain from sites in the suprahyoid musculature, chronic respiratory infection, drug allergies, and medications have been proposed. [4],[6],[7],[10],[13],[16],[40] However these are not considered the perfect etiologic factors for BMS, probably due to lack of methodological standardization in studies.

Use of angiotensin-converting enzyme (ACE) inhibiters has been linked with BMS. Once these medicines were reduced or discontinued, oral burning was found to remit within several weeks. [46],[47],[48]


   Diagnosis Top


Diagnosing BMS is by a process of elimination - ruling out anything else that could cause pain and burning in the mouth such as canker sores or other kinds of lesions, benign or malignant. If the patient's mouth looks normal and tastes fine but burns anyway, it is diagnosed and labeled as BMS. Clinical history is helpful in diagnosing BMS. In these patients, the laboratory values are usually normal.


   Management Top


Though the treatment of BMS is usually directed at its symptoms, the treatment of BMS based on the above- mentioned causes is usually unsatisfactory. In those studies where success has been claimed, inadequate controls and/or inadequate follow-up periods were used. Therefore, no therapy for BMS has been proven effective.

As a group, BMS subjects display increased help-seeking behavior and the personality and psychiatric characteristics described for patients with chronic pain. It is to be expected that no single etiologic factor will be identified that is common to all patients included under the umbrella of BMS. Hence the treatment needs to be customized to the etiologic factors identified in an individual patient, with more attention to be given to symptomatic relief and management of any associated behavioral or psychiatric disorder. The patient should first be acquainted with the results of history and diagnostic tests carried out. These procedures are by themselves a good therapeutic procedure.

Depending on the individual cause, the treatment is followed logically which includes removal of local irritants, construction of plastic retainers to cover irregularities of the occlusion that magnify the side-effects of tongue habits, treatment of the muscular tension by correction of the malocclusion, fabrication of a night-guard or Hawley appliance, or by muscle relaxants such as diazepam or treatment of the systemic disease such as a connective tissue disease or diabetes mellitus. Neurosurgical exploration of the lingual nerve of neuropathic origin may relieve some of the more distressing symptoms. [1]

Symptomatic treatment is usually obtained from the use of topical analgesics such as 0.5% aqueous diphenhydramine alone or mixed with 0.5% dyclonine alone, or lidocaine, or other analgesic ointments applied to the affected area. [1]

For idiopathic problem associated with depression, treatment of the depression frequently controls the burning sensation. [1] The analgesic effects of antidepressants are seen at doses lower than those usually effective in patients who are not depressed. Tricyclic antidepressant drugs such as amitriptyline and doxepin which inhibit re-uptake of both serotonin and norepinephrine are more effective than drugs which are selective for either neurotransmitter. A daily 25 mg dose of amitriptyline for 3 weeks was demonstrated to be superior to placebo in a variety of patients with chronic pain. [49] Lower dosages (25-75 mg) should be used initially for patients who are not depressed, whereas antidepressant doses should be reserved for depressed patients and should be prescribed in collaboration with the physician. When local factors such as an irregular denture and muscular tension are prominent factors, small doses of benzodiazepines are effective. [1],[50] Benzodiazepines should not be prescribed in large quantities, and patients should not be permitted to increase their doses without professional supervision as it develops dependence over the long term. It is recommended to reduce the dose or discontinue its use if the medication appears to be ineffective or if sedative side effects or depressive symptoms arise. Studies also support the utility of a low dosage of clonazepam [51] and gabapentin [52] in treating BMS.

Estrogen and progesterone supplementation have been used effectively in the treatment of burning mouth in postmenopausal women. [37],[45]

Topical capsaicin has been used as a desensitizing agent in BMS patients. Capsaicin depletes the substance P (a neurotransmitter) sufficiently to decrease the peripheral burning pain perception. [53]

According to recent studies [54],[55] alpha-lipoic acid (ALA) the trometamol salt of thioctic acid, significantly reduces symptoms of BMS. BMS has features of a neuropathy and could be related to the production of toxic free radicals that are released in stress situations. ALA has been shown to be neuroprotective (especially effective in treating diabetic neuropathy) [56],[57],[58] in clinical studies.

The clinical trial of efficacy of ALA on the symptomatology of BMS was performed in a study which involved 42 patients with BMS and no clinical or laboratory evidence of organic oral disease. [54] The patients were divided into two groups (Test and Control), each consisting of 21 subjects, matched for age and sex. Significant improvements were seen in two-thirds of patients with BMS receiving ALA (600 mg / day orally for 20 days followed by 200 mg / day for 10 days); in about 15% of those using placebo and in up to two-thirds of those who having tried placebo were switched to ALA, their improvement rate increased. They found ALA of remarkable benefit with minimal adverse effects.

Encouraged by these results, the same research team undertook a more rigorous study with the same objective. [55] This randomized, placebo-controlled and double blind study was conducted for two months on 60 patients (two groups of 30 each) with constant burning. In this study the results were even better: more than 96% of those taking ALA (600 mg / day for 2 months) showed significant improvement in their symptoms, compared with 40% of those taking placebo. In ALA group, 4 patients (13%) showed a complete cessation of pain, which amounts to cure. None of the patients on ALA got worse during the course of the study, whereas 20% of the placebo group did. The improvement achieved with ALA was maintained in over 70% of patients at the 1-year follow-up, whereas all those who had taken a placebo had worsened. This study supports the hypothesis that BMS is a neuropathy.

A familiar nutritional supplement, ALA is a natural substance (our bodies make it in tiny quantities) soluble in both water and fat. ALA is a potent antioxidant that helps to conserve other antioxidants such as vitamin E and C, and increases the intracellular levels of glutathione, another potent antioxidant. It eliminates free radicals and prevents damage to nerve cells. [55] Thus, it protects the body against damage from free radicals. However, more research is needed to clarify its mechanism of action in the treatment of BMS.

However, if no organic basis is found for BMS, repeated reassurance about the benign nature of the condition should be given and it is a good policy to refrain from recommending or prescribing too many medicines. These patients should be handled with tender loving care and should be followed up at periodic intervals.

 
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56.Reljanovic M, Reichel G, Rett K, Lobisch M, Schuette K, Moller W, et al . Treatment of diabetic polyneuropathy with the antioxidant thioctic acid (alpha-lipoic acid): A two year multicenter randomized double blind placebo controlled trial (ALADIN II), Alpha-Lipoic Acid in Diabetic Neuropathy. Free Radic Res 1999;31:171-9.  Back to cited text no. 56    
57.Ziegler D, Hanefeld M, Ruhnau KJ, Hasche H, Lobisch M, Schütte K, et al . Treatment of symptomatic diabetic polyneuropathy with the antioxidant alpha-lipoic acid: A 7 month multicenter randomized controlled trial (ALADIN III Study) ALADIN III Study Group: Alpha-Lipoic Acid in Diabetic Neuropathy. Diabetes Care 1999;22: 1296-1301.  Back to cited text no. 57    
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