Dopa responsive burning mouth syndrome: Restless mouth syndrome or oral variant of restless legs syndrome?

Prakash, Sanjay; Ahuja, Sunil K.; Rathod, Chirag

doi:10.1016/j.jns.2012.07.007

Cited by 35 publications

(22 citation statements)

References 36 publications

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“…Hagenberg et al reported a decrease in D2 receptor coupling, which led to a decrease in D1/D2 ratio in the striatum of BMS patients. Clinical reports support this intra‐cranial dopaminergic dysregulation, and some researchers reported comorbidity with Parkinson's disease and, more recently, with restless legs syndrome (RLS) . In addition, some studies reported that the D2 agonist pramipexole alleviated burning mouth symptoms .…”

Section: Introductionmentioning

confidence: 95%

An updated review on pathophysiology and management of burning mouth syndrome with endocrinological, psychological and neuropathic perspectives

Imamura

Shinozaki

Okada-Ogawa

et al. 2019

J of Oral Rehabilitation

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Burning mouth syndrome (BMS) is a chronic oro-facial pain disorder of unknown cause. It is more common in peri-and post-menopausal women, and sex hormone dysregulation is believed to be an important causative factor. Psychosocial events often trigger or exacerbate symptoms, and persons with BMS appear to be predisposed towards anxiety and depression. Atrophy of small nerve fibres in the tongue epithelium has been reported, and potential neuropathic mechanisms for BMS are now widely investigated. Historically, BMS was thought to comprise endocrinological, psychosocial and neuropathic components. Neuroprotective steroids and glial cell line-derived neurotrophic factor family ligands may have pivotal roles in the peripheral mechanisms associated with atrophy of small nerve fibres. Denervation of chorda tympani nerve fibres that innervate fungiform buds leads to alternative trigeminal innervation, which results in dysgeusia and burning pain when eating hot foods. With regard to the central mechanism of BMS, depletion of neuroprotective steroids alters the brain network-related mood and pain modulation. Peripheral mechanistic studies support the use of topical clonazepam and capsaicin for the management of BMS, and some evidence supports the use of cognitive behavioural therapy. Hormone replacement therapy may address the causes of BMS, although adverse effects prevent its use as a first-line treatment. Selective serotonin reuptake inhibitors (SSRIs) and serotonin and noradrenaline reuptake inhibitors (SNRIs) may have important benefits, and well-designed controlled studies are expected. Other treatment options to be investigated include brain stimulation and TSPO (translocator protein 18 kDa) ligands. K E Y W O R D Sburning mouth syndrome, central pain modulation, menopause, neuroprotective steroids | 575 IMAMURA et Al. | INTRODUC TI ONBurning mouth symptoms that occurred as secondary phenomena attributable to local conditions 1-9 were previously referred to as "secondary" burning mouth syndrome (BMS), 10 but BMS now refers only to burning symptoms not attributable to local or systemic causes. 11 After excluding such conditions, some common characteristics of BMS are important. Burning sensations are usually bilateral, and intensity fluctuates. 2 The most common site is the tip of the tongue, but pain is often noted at the lateral border of the tongue, lips and hard palate. 12 Affected persons often complain of dysgeusia, 13 which may be accompanied by subjective xerostomia. 14 Peri-and post-menopausal women are predisposed to the condition.Some patients exhibit depressive symptoms and anxiety 15-18 and may express concern regarding the presence of a malignant condition. 10,16 Psychosocial stressors can trigger or worsen pain, 15,19 while eating and drinking usually alleviate pain. 10,20 These manifestations are characteristic features of BMS and should not be excluded as secondary signs or symptoms of primary disease. Agreement on these clinical features yields important clues in understanding the underlying pathoph...

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Section: Introductionmentioning

confidence: 95%

An updated review on pathophysiology and management of burning mouth syndrome with endocrinological, psychological and neuropathic perspectives

Imamura

Shinozaki

Okada-Ogawa

et al. 2019

J of Oral Rehabilitation

View full text Add to dashboard Cite

show abstract

“…The best evidence for BMS treatment includes only topical capsaicin, alpha-lipoic acid (ALA), and clonazepam [16, 17]. Interestingly, there are reports of BMS comorbidity with Parkinson disease (PD) or Restless Syndrome [18–20] and dopamine agonists showed potential efficacy in reducing BMS symptoms [21, 22]. Non-pharmaceutical treatments may help in addition, like cognitive psychotherapy [23].…”

Section: Introductionmentioning

confidence: 99%

Refractory burning mouth syndrome: clinical and paraclinical evaluation, comorbidities, treatment and outcome

2017

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BackgroundBurning Mouth Syndrome (BMS) is a chronic pain condition characterized by persistent intraoral burning without related objective findings and unknown etiology that affects elderly females mostly. There is no satisfactory treatment for BMS. We aimed to observe the long-term efficacy of high velanfaxine doses combined with systemic and topical administered clonazepam in a particular subgroup of BMS patients who do not respond to current clinical management.ResultsEight (66.1 ± 6.2 years old females) out of 14 BMS patients fulfilled the inclusion criteria and were treated with venlafaxine (300 mg/d) and clonazepam (5 mg/d) for 35.4 ± 12.1 (mean ± SD) months. The average duration of the symptoms at baseline was 4.3 ± 1.4 years and the overall mean daily pain intensity score was 8.6 ± 1.3 (VAS); pain was in tongue and within the oral mucosa, accompanying by oral and facial dysesthesia. In five patients tasting was abnormal. All patients had positive history of concomitant primary headache. The average score of Hamilton Rating scale for Anxiety and Depression was 21 ± 4.2, and 26.1 ± 2.9, respectively. Previous ineffective treatments include anticonvulsants and anti-depressants. All patients responded (more than 50% decrease in VAS) after three months treatment (mean VAS 3.2 ± 2.2) with no remarkable adverse events.ConclusionBMS deserves bottomless psychiatric evaluation and management when current available treatments fail. Treatment with venlafaxine combined with topical and systemic clonazepam may be effective in refractory BMS cases but further investigation in a large-scale controlled study is needed to confirm these results.

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“…Dieb et al (13) reported that nigrostriatal dopaminergic depletion produces trigeminal neuropathic pain that at least involves a segmental mechanism in a rat model. Khan et al (14) showed multiple lines of evidence of disturbed structure and function in the medial prefrontal cortex and hippocampus in BMS patients, which might also affect mood.…”

Section: Discussionmentioning

confidence: 99%

“…Three sonographic abnormalities were found in patients with typical restless legs syndrome (13)(14)(15): substantia nigra hypoechogenicity (17%), nuclei raphe hypoechogenicity (26%) and red nucleus hyperechogenicity (26%). These results suggest similar underlying mechanism of both conditions.…”

Section: Discussionmentioning

confidence: 99%

The Significance of Brain Transcranial Sonography in Burning Mouth Syndrome: a Pilot Study

Zavoreo¹,

Boras²,

Zadravec³

et al. 2017

Acta Stomatol Croat

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Dopa responsive burning mouth syndrome: Restless mouth syndrome or oral variant of restless legs syndrome?

Cited by 35 publications

References 36 publications

An updated review on pathophysiology and management of burning mouth syndrome with endocrinological, psychological and neuropathic perspectives

An updated review on pathophysiology and management of burning mouth syndrome with endocrinological, psychological and neuropathic perspectives

Refractory burning mouth syndrome: clinical and paraclinical evaluation, comorbidities, treatment and outcome

The Significance of Brain Transcranial Sonography in Burning Mouth Syndrome: a Pilot Study

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