#PAGE_PARAMS# #ADS_HEAD_SCRIPTS# #MICRODATA#

Idiopathic orofacial pain


Authors: P. Řehulka 1;  A. Bártková 2
Authors‘ workplace: I. neurologická klinika, LF MU a FN u sv. Anny v Brně 1;  Neurologická klinika, LF UP a FN v Olomouci 2
Published in: Cesk Slov Neurol N 2025; 88(1): 39-46
Category: Review Article
doi: https://doi.org/10.48095/cccsnn202539

Overview

Idiopathic orofacial pain is a group of painful disorders affecting the facial region and/or oral cavity with unclear etiology. This group includes burning mouth syndrome, persistent idiopathic dentoalveolar pain, persistent idiopathic facial pain, and constant unilateral facial pain with additional attacks. These are acquired diseases of adulthood, most commonly affecting postmenopausal women. The etiology of these disorders is not entirely understood, but likely involves changes at both the peripheral and central levels of the somatosensory system, including small fiber neuropathy, subclinical peripheral neuropathy, alterations in brainstem functions, and changes in central inhibitory pain modulation. Although some patients mistakenly consider dental treatment as the triggering situation, a causal relationship with invasive procedures in the oral cavity has not been unequivocally proven. Diagnosis is based on evaluating the clinical characteristics of the pain and excluding secondary causes. Treatment strategies include acute topical application of certain medications or long-term off-label treatment (antidepressants, antiseizure medications). The effectiveness of pharmacological treatment may be limited, often due to side effects. Expert agreement is that early diagnosis and pharmacological therapy are positive prognostic factors, while repeated invasive procedures worsen the prognosis. Comorbid depressive and anxiety symptoms affect roughly one-third to one-half of patients. Psychosocial stress can exacerbate the disease course. Spontaneous improvement occurs in only a minority of patients (at most one-third), usually after two to seven years of disease duration. Providing sufficient information about the diagnosis and reassuring the patient about the neurobiological nature of the disease remains crucial. Supportive psychotherapeutic treatment is also recommended.

Keywords:

trigeminal neuralgia – facial pain – burning mouth syndrome – dentoalveolar pain


Sources

1. International Classification of Orofacial Pain, 1st edition (ICOP). Cephalalgia 2020; 40 (2): 29–221. doi: 10.1177/0333102419893823.

2. Kosek E, Cohen M, Baron R et al. Do we need a third mechanistic descriptor for chronic pain states? Pain 2016; 157 (7): 1382–1386. doi: 10.1097/j.pain.0000000 000000507.

3. Fitzcharles MA, Cohen SP, Clauw DJ et al. Nociplastic pain: towards an understanding of prevalent pain conditions. Lancet 2021; 97 (10289): 2098–2110. doi: 10.1016/S0140-6736 (21) 00392-5. PMID: 340 62144.

4. Granan LP. We do not need a third mechanistic descriptor for chronic pain states! Not yet. Pain 2017; 158 (1): 179. doi: 10.1097/j.pain.0000000000000735.

5. Puhakka A, Forssell H, Soinila S et al. Peripheral nervous system involvement in primary burning mouth syndrome – results of a pilot study. Oral Dis 2016; 22 (4): 338–344. doi: 10.1111/odi.12454.

6. Yilmaz Z, Renton T, Yiangou Y et al. Burning mouth syndrome as a trigeminal small fibre neuropathy: Increased heat and capsaicin receptor TRPV1 in nerve fibres correlates with pain score. J Clin Neurosci 2007; 14 (9): 864–871. doi: 10.1016/j.jocn.2006. 09.002.

7. Beneng K, Yilmaz Z, Yiangou Y et al. Sensory purinergic receptor P2X3 is elevated in burning mouth syndrome. Int J Oral Maxillofac Surg 2010; 39 (8): 815–819. doi: 10.1016/j.ijom.2010.03.013.

8. Seol SH, Chung G. Estrogen-dependent regulation of transient receptor potential vanilloid 1 (TRPV1) and P2X purinoceptor 3 (P2X3): Implication in burning mouth syndrome. J Dent Sci 2022; 17 (1): 8–13. doi: 10.1016/j.jds.2021.06.007.

9. Eliav E, Kamran B, Schaham R et al. Evidence of chorda tympani dysfunction in patients with burning mouth syndrome. J Am Dent Assoc 2007; 138 (5): 628–633. doi: 10.14219/jada.archive.2007.0234.

10. Spadari F, Venesia P, Azzi L et al. Low basal salivary flow and burning mouth syndrome: new evidence in this enigmatic pathology. J Oral Pathol Med 2015; 44 (3): 229–233. doi: 10.1111/jop.12240.

11. Imura H, Shimada M, Yamazaki Y et al. Characteristic changes of saliva and taste in burning mouth syndrome patients. J Oral Pathol Med 2016; 45 (3): 231–236. doi: 10.1111/jop.12350.

12. Nixdorf D, Moana-Filho E. Persistent dento-alveolar pain disorder (PDAP): working towards a better understanding. Rev Pain 2011; 5 (4): 18–27. doi: 10.1177/204946371100500404.

13. Neto HS, Camilli JA, Marques MJ. Trigeminal neuralgia is caused by maxillary and mandibular nerve entrapment: greater incidence of right-sided facial symptoms is due to the foramen rotundum and foramen ovale being narrower on the right side of the cranium. Med Hypotheses 2005; 65 (6): 1179–1182. doi: 10.1016/j.mehy.2005.06.012.

14. Grémeau-Richard C, Dubray C, Aublet-Cuvelier B et al. Effect of lingual nerve block on burning mouth syndrome (stomatodynia): a randomized crossover trial. Pain 2010; 149 (1): 27–32. doi: 10.1016/j.pain.2009. 11.016.

15. List T, Leijon G, Helkimo M et al. Effect of local anesthesia on atypical odontalgia-a randomized controlled trial. Pain 2006; 122 (3): 306–314. doi: 10.1016/j.pain.2006.02.005.

16. Tan Y, Wu X, Chen J et al. Structural and functional connectivity between the amygdala and orbital frontal cortex in burning mouth syndrome: an fMRI study. Front Psychol 2019; 10: 1700. doi: 10.3389/fpsyg. 2019.01700.

17. Schmidt-Wilcke T, Hierlmeier S, Leinisch E. Altered regional brain morphology in patients with chronic facial pain. Headache 2010; 50 (8): 1278–1285. doi: 10.1111/j.1526-4610.2010.01637.x.

18. Hagelberg N, Forssell H, Rinne JO et al. Striatal dopamine D1 and D2 receptors in burning mouth syndrome. Pain 2003; 101 (1–2): 149–154. doi: 10.1016/s0304-3959 (02) 00323-8.

19. Hagelberg N, Forssell H, Aalto S et al. Altered dopamine D2 receptor binding in atypical facial pain. Pain 2003; 106 (1–2): 43–48. doi: 10.1016/s0304-3959 (03) 0 0275-6.

20. Moseley GL. A pain neuromatrix approach to patients with chronic pain. Man Ther 2003; 8 (3): 130–140. doi: 10.1016/s1356-689x (03) 00051-1.

21. Bergdahl M, Bergdahl J. Burning mouth syndrome: prevalence and associated factors. J Oral Pathol Med 1999; 28 (8): 350–354. doi: 10.1111/j.1600-0714.1999.tb02052.x.

22. Jääskeläinen SK, Woda A. Burning mouth syndrome. Cephalalgia 2017; 37 (7): 627–647. doi: 10.1177/0333102417694883.

23. Klein B, Thoppay JR, De Rossi SS et al. Burning mouth syndrome. Dermatol Clin 2020; 38 (4): 477–483. doi: 10.1016/j.det.2020.05.008.

24. Cui Y, Xu H, Chen FM et al. Efficacy evaluation of clonazepam for symptom remission in burning mouth syndrome: a meta-analysis. Oral Dis 2016; 22 (6): 503–511. doi: 10.1111/odi.12422.

25. Kisely S, Forbes M, Sawyer E et al. A systematic review of randomized trials for the treatment of burning mouth syndrome. J Psychosom Res 2016; 86: 39–46. doi: 10.1016/j.jpsychores.2016.05.001.

26. Currie CC, Jääskeläinen SK. Burning mouth syndrome: a review. Oral Surg 2020; 13 (4): 379–388. doi: 10.1111/ors.12456.

27. Sanner F, Sonntag D, Hambrock N et al. Patients with persistent idiopathic dentoalveolar pain in dental practice. Int Endod J 2022; 55 (3): 231–239. doi: 10.1111/iej.13664.

28. Miura A, Tu TTH, Shinohara Y et al. Psychiatric comorbidities in patients with atypical odontalgia. J Psychosom Res 2018; 104: 35–40. doi: 10.1016/j.jpsychores.2017.11.001.

29. Takenoshita M, Sato T, Kato Y et al. Psychiatric diagnoses in patients with burning mouth syndrome and atypical odontalgia referred from psychiatric to dental facilities. Neuropsychiatr Dis Treat 2010; 6: 699–705. doi: 10.2147/NDT.S12605.

30. Tu TTH, Miura A, Shinohara Y et al. Pharmacotherapeutic outcomes in atypical odontalgia: determinants of pain relief. J Pain Res 2019; 12: 831–839. doi: 10.2147/JPR.S188362.

31. Coulter J, Nixdorf DR. A review of persistent idiopathic dentoalveolar pain (formerly PDAP/Atypical odontalgia). Oral Surg 2020; 13 (4): 371–378. doi: 10.1111/ors. 12472.

32. Vickers ER, Cousins MJ, Walker S et al. Analysis of 50 patients with atypical odontalgia. A preliminary report on pharmacological procedures for diagnosis and treatment. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1998; 85 (1): 24–32. doi: 10.1016/s1079-2104 (98) 9 0393-6.

33. List T, Leijon G, Helkimo M et al. Effect of local anesthesia on atypical odontalgia--a randomized controlled trial. Pain 2006; 122 (3): 306–314. doi: 10.1016/j.pain.2006.02.005.

34. Dawson A, Dawson J, Ernberg M. The effect of botulinum toxin A on patients with persistent idiopathic dentoalveolar pain – a systematic review. J Oral Rehabil 2020; 47 (9): 1184–1191. doi: 10.1111/joor.13053.

35. Forssell H, Tenovuo O, Silvoniemi P et al. Differences and similarities between atypical facial pain and trigeminal neuropathic pain. Neurology 2007; 69 (14): 1451–1459. doi: 10.1212/01.wnl.0000277274.83301.c0.

36. Maarbjerg S, Wolfram F, Heinskou TB et al. Persistent idiopathic facial pain – a prospective systematic study of clinical characteristics and neuroanatomical findings at 3.0 Tesla MRI. Cephalalgia 2017; 37 (13): 1231–1240. doi: 10.1177/0333102416675618.

37. Obermann M, Holle D, Katsarava Z. Trigeminal neuralgia and persistent idiopathic facial pain. Expert Rev Neurother 2011; 11 (11): 1619–1629. doi: 10.1586/ ern.11.156.

38. Ziegeler C, May A. Facial presentations of migraine, TACs, and other paroxysmal facial pain syndromes. Neurology 2019; 93 (12): e1138–e1147. doi: 10.1212/WNL.0000000000008124.

39. Kohorst JJ, Bruce AJ, Torgerson RR et al. The prevalence of burning mouth syndrome: a population-based study. Br J Dermatol 2015; 172 (6): 1654–1656. doi: 10.1111/bjd.13613.

40. Řehulka P, Bartáková J, Hudečková M et al. Mezinárodní klasifikace orofaciální bolesti (ICOP) – oficiální český překlad prvního vydání. Cesk Slov Neurol N 2020; 83/116 (6): 602–607. doi: 10.48095/cccsnn 2020602.

41. Ambler Z. Neuropatie nervus mentalis jako manifestace systémové malignity. Cesk Slov Neurol N 2009; 72/105 (6): 560–562.

42. Kadaňka Z Jr, Bednařík J. Klinické syndromy z oblasti cervikálního plexu. Cesk Slov Neurol N 2019; 82/115 (6): 616–620. doi: 10.14735/amcsnn2019616.

43. Keogh E, Asmundson GJG. Negative affectivity, catastrophizing, and anxiety sensitivity. In: Asmundson GJG, Vlaeyen JWS, Crombez G (eds). Understanding and treating fear of pain. New York: Oxford University Press 2004, reprinted 2007: 91–115.

44. Reme SE, Ljosaa TM, Stubhaug A et al. Perceived injustice in patients with chronic pain: prevalence, relevance, and associations with long-term recovery and deterioration. J Pain 2022; 23 (7): 1196–1207. doi: 10.1016/j.jpain.2022.01.007.

45. Paterson AJ, Lamb AB, Clifford TJ et al. Burning mouth syndrome: the relationship between the HAD scale and parafunctional habits. J Oral Pathol Med 1995; 24 (7): 289–292. doi: 10.1111/j.1600-0714.1995.tb0 1186.x.

46. Grossman S, Tan H, Gadiwalla Y. Cannabis and orofacial pain: a systematic review. Br J Oral Maxillofac Surg 2022; 60 (5): e677–e690. doi: 10.1016/j.bjoms.2021. 06.005.

47. Benoliel R, Gaul C. Persistent idiopathic facial pain. Cephalalgia 2017; 37 (7): 680–691. doi: 10.1177/03 33102417706349.

48. Mueller D, Obermann M, Yoon MS et al. Prevalence of trigeminal neuralgia and persistent idiopathic facial pain: a population-based study. Cephalalgia 2011; 31 (15): 1542–1548. doi: 10.1177/03331024114 24619.

49. Nixdorf DR, Moana-Filho EJ, Law AS et al. Frequency of nonodontogenic pain after endodontic therapy: a systematic review and meta-analysis. J Endod 2010; 36 (9): 1494–1498. doi: 10.1016/j.joen.2010. 06.020.

50. Woda A, Dao T, Gremeau-Richard C. Steroid dysregulation and stomatodynia (burning mouth syndrome). J Orofac Pain 2009; 23 (3): 202–210.

51. Nixdorf DR, Drangsholt MT, Ettlin DA et al. International RDC-TMD Consortium. Classifying orofacial pains: a new proposal of taxonomy based on ontology. J Oral Rehabil 2012; 39 (3): 161–169. doi: 10.1111/j.1365-2842.2011.02247.x.

52. Baad-Hansen L. Atypical odontalgia – pathophysiology and clinical management. J Oral Rehabil 2008; 35 (1): 1–11. doi: 10.1111/j.1365-2842.2007.01813.x.

53. Tan HL, Renton T. Burning mouth syndrome: an update. Cephalalgia Rep 2020; 3. doi: 10.1177/251581 6320970143

54. Taiminen T, Kuusalo L, Lehtinen L et al. Psychiatric (axis I) and personality (axis II) disorders in patients with burning mouth syndrome or atypical facial pain. Scand J Pain 2011; 2 (4): 155–160. doi: 10.1016/j.sjpain.2011.06.004. PMID: 29913754.

55. Sardella A, Lodi G, Demarosi F et al. Burning mouth syndrome: a retrospective study investigating spontaneous remission and response to treatments. Oral Dis 2006; 12 (2): 152–155. doi: 10.1111/j.1601-0825.2005.01174.x.

56. Kim MJ, Choi JH, Kho HS. Long-term prognosis of burning mouth syndrome following treatment. Int J Oral Maxillofac Surg 2022; 51 (12): 1538–1544. doi: 10.1016/j.ijom.2022.08.011.

57. Pigg M, Svensson P, Drangsholt M et al. Seven-year follow-up of patients diagnosed with atypical odontalgia: a prospective study. J Orofac Pain 2013; 27 (2): 151–164. doi: 10.11607/jop.1033.

58. Yoon JH, Chun YC, Park SY et al. Malignant lymphoma of the maxillary sinus manifesting as a persistent toothache. J Endod 2001; 27 (12): 800–802. doi: 10.1097/00004770-200112000-00023.

59. Forssell H, Jääskeläinen S, List T et al. An update on pathophysiological mechanisms related to idiopathic oro-facial pain conditions with implications for management. J Oral Rehabil 2015; 42 (4): 300–322. doi: 10.1111/joor.12256.

60. Fricová J, Kozák J, Lejčko J et al. (eds.). Metodické pokyny pro farmakoterapii chronické bolesti. Bolest 2022; 25 (Suppl 1): 1–44.

Labels
Paediatric neurology Neurosurgery Neurology

Article was published in

Czech and Slovak Neurology and Neurosurgery

Issue 1

2025 Issue 1

Most read in this issue
Login
Forgotten password

Enter the email address that you registered with. We will send you instructions on how to set a new password.

Login

Don‘t have an account?  Create new account

#ADS_BOTTOM_SCRIPTS#