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Atypical odontalgia: pathophysiology, diagnosis and management

ABSTRACT

BACKGROUND AND OBJECTIVES:

Atypical odontalgia, a subtype of persistent idiopathic facial pain, is characterized by continuous pain in one tooth or more, or inside the alveolus after exodontia, with no apparent clinical causes. These patients run the risk of going through unnecessary dental/surgical procedures which would worsen their pain. Since the pathophysiology, diagnosis, and management of atypical odontalgia are not clear, this article aims to present an integrative literature review about these aspects.

CONTENTS:

A review of articles related to the topic was conducted on the Pubmed database using the keywords “atypical odontalgia” OR “phantom tooth pain” OR “idiopathic tooth pain” OR “odontalgia” OR “odontalgias” OR “atypical toothache”. Applying the inclusion criteria (publications in the last ten years, in English, as clinical trials, multicenter studies, case reports, reviews, integrative and systematic reviews, 114 articles were found, and 39 were selected after the application of the exclusion criteria (articles with no relation to the topic).

CONCLUSION:

Although studies suggest the involvement of strong neuropathic mechanism, the psychological/psychiatric aspects might be considered not as a primary cause, but as an aggravator of the patient´s pain. Knowledge of other pathologies is recommended in order to determine the differential diagnosis. Also, complementary image tests, qualitative somatosensorial test, and reference to an orofacial pain specialist should be considered. In case of uncertain diagnosis, it is recommended to avoid any dental procedures because the pain can get worse.

Keywords:
Atypical odontalgia; Endodontic; Odontalgia

RESUMO

JUSTIFICATIVA E OBJETIVOS:

A odontalgia atípica, um subtipo da dor facial idiopática persistente, se caracteriza por dor contínua em um ou mais dentes, ou no alvéolo, após exodontia sem qualquer causa aparente e é um desafio para o dentista. O desconhecimento por parte do profissional pode levar a procedimentos odontológicos desnecessários e mutiladores, piorando e/ou cronificando a dor do paciente. Diante desse panorama, o objetivo deste estudo foi apresentar informações referentes à fisiopatologia, diagnóstico e tratamento da odontalgia atípica através de uma revisão integrativa da literatura.

CONTEÚDO:

A busca na base de dados Pubmed foi realizada com os termos: “atypical odontalgia” OR “phantom tooth pain” OR “idiopathic tooth pain” OR “odontalgia” OR “odontalgias” OR “atypical toothache”. Aplicando-se critérios de inclusão (publicações nos últimos 10 anos, de língua inglesa, tipo ensaio clínico, estudo multicêntrico, relato de caso, revisão, revisão integrativa científica e sistemática) foram encontrados 114 artigos, dos quais 39 foram selecionados após aplicação do critério de exclusão (trabalhos sem relação com o tema).

CONCLUSÃO:

Embora os estudos apontem forte envolvimento de mecanismos neuropáticos, aspectos psicogênicos/psiquiátricos devem ser levados em consideração como agravante do estado de dor do paciente. Sugere-se conhecimento sobre as outras doenças existentes para se realizar um diagnóstico diferencial, exames complementares de imagem, realização do teste somatossensorial qualitativo, encaminhamento a um especialista em dor orofacial e neurologista, e em casos de dúvida, não realizar nenhum procedimento a fim de não piorar a sua dor.

Descritores:
Endodontia; Odontalgia; Odontalgia atípica

INTRODUCTION

Atypical odontalgia (AO) represents a clinical challenge for most dentists11 Malacarne A, Spierings EL, Lu C, Maloney GE. Persistent dentoalveolar pain disorder: a comprehensive review. J Endod. 2018;44(2):206-11.. Generally, when a patient complains of pain, its origin is odontogenic, and the professional can identify and treat its cause - for example, a typical toothache due to pulpitis, caries or periodontal problem. But in some situations, pain continues in one or more teeth or in the socket after extraction without any apparent dental cause22 Headache Classification Committee of the International Headache Society (IHS) The International Classification of Headache Disorders, 3rd ed. Cephalalgia. 2018;38(1):1-211., and the dentist faces the challenge of determining the true non-odontogenic origin of pain and properly diagnosing it11 Malacarne A, Spierings EL, Lu C, Maloney GE. Persistent dentoalveolar pain disorder: a comprehensive review. J Endod. 2018;44(2):206-11.,33 Baad-Hansen L. Atypical odontalgia - pathophysiology and clinical management. J Oral Rehabil. 2008;35(1):1-11.

4 Goel R, Kumar S, Panwar A, Singh AB. Pontine infarct presenting with atypical dental pain: a case report. Open Dent J. 2015;9:337-9.
-55 Durham J, Stone SJ, Robinson LJ, Ohrbach R, Nixdorf DR. Development and preliminary evaluation of a new screening instrument for atypical odontalgia and persistent dentoalveolar pain disorder. Int Endod J. 2019;52(3):279-87..

According to the 3rd edition of the International Headache Society (ICHD-3)22 Headache Classification Committee of the International Headache Society (IHS) The International Classification of Headache Disorders, 3rd ed. Cephalalgia. 2018;38(1):1-211., the diagnostic criterion of AO is described by continuous pain in one or more teeth or socket after extraction, without any apparent dental and neurological causes. Pain lasts for more than two hours daily and persists for more than three months and may or may not be associated with a history of dental trauma (Table 1).

Table 1
Diagnostic criteria of the "International Headache Society 3"

The difficulty in diagnosing AO is because the reported pain is identical to those of odontogenic origin without clinical and radiographic alterations44 Goel R, Kumar S, Panwar A, Singh AB. Pontine infarct presenting with atypical dental pain: a case report. Open Dent J. 2015;9:337-9.,66 Woda A, Tubert-Jeannin S, Bouhassira D, Attal N, Fleiter B, Goulet JP, et al. Towards a new taxonomy of idiopathic orofacial pain. Pain. 2005;116(3):396-406.. The patient may have a history of extensive dental treatment without pain relief, which makes the diagnosis more complex77 Rees RT, Harris M. Atypical odontalgia. Br J Oral Surg. 1979;16(3):212-8.,88 Koratkar H, Pedersen J. Atypical odontalgia: a review. Northwest Dent. 2008;87(1): 37-8, 62.. Endodontic treatment, apicectomy and/or extraction may alleviate pain temporarily, but pain increases in intensity in a few days or weeks77 Rees RT, Harris M. Atypical odontalgia. Br J Oral Surg. 1979;16(3):212-8.,99 Marbach JJ. Phantom tooth pain. J Endod. 1978;4(12):362-72..

The physiopathology is not well defined, and several mechanisms have been suggested in the last 50 years33 Baad-Hansen L. Atypical odontalgia - pathophysiology and clinical management. J Oral Rehabil. 2008;35(1):1-11.,1010 Porporatti AL, Costa YM, Stuginski-Barbosa J, Bonjardim LR, Duarte MA, Conti PC. Diagnostic accuracy of quantitative sensory testing to discriminate inflammatory toothache and intraoral neuropathic pain. J Endod. 2015;41(10):1606-13.. There is great controversy regarding AO and psychological factors, with studies that indicate a large percentage of individuals with depression1111 Graff-Radford SB, Solberg WK. Is atypical odontalgia a psychological problem? Oral Surg Oral Med Oral Pathol. 1993;75(5):579-82. and others question whether they could be secondary factors to pain1212 Schnurr RF, Brooke RI. Atypical odontalgia. Update and comment on long-term follow-up. Oral Surg Oral Med Oral Pathol. 1992;73(4):445-8.

13 Clark GT. Persistent orodental pain, atypical odontalgia, and phantom tooth pain: when are they neuropathic disorders? J Calif Dent Assoc. 2006;34(8):599-609.

14 Takenoshita M, Sato T, Kato Y, Katagiri A, Yoshikawa T, Sato 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.

15 Ciaramella A, Paroli M, Lonia L, Bosco M, Poli P. Biopsychosocial aspects of atypical odontalgia. ISRN Neurosci. 2013;2013:413515.
-1616 Miura A, Tu TTH, Shinohara Y, Mikuzuki L, Kawasaki K, Sugawara S, et al. Psychiatric comorbidities in patients with atypical odontalgia. J Psychosom Res. 2018;104:35-40.. Vascular origin is a disorder of the pulp and ligament blood vessels and is described as a “dental migraine”77 Rees RT, Harris M. Atypical odontalgia. Br J Oral Surg. 1979;16(3):212-8.,1717 Kreisberg MK. Atypical odontalgia: differential diagnosis and treatment. J Am Dent Assoc. 1982;104(6):852-4..

Neuropathic origin is the most studied, being described in 1971 by Melzack based on Mitchell’s phantom limb pain in 1871 (Apud99 Marbach JJ. Phantom tooth pain. J Endod. 1978;4(12):362-72. and described by Marbach in 1978 for the first time as “phantom tooth pain”). In addition, other neuropathic mechanisms would explain the physiopathological process: deafferentation hypersensitivity1212 Schnurr RF, Brooke RI. Atypical odontalgia. Update and comment on long-term follow-up. Oral Surg Oral Med Oral Pathol. 1992;73(4):445-8.,1818 Campbell RL, Parks KW, Dodds RN. Chronic facial pain associated with endodontic therapy. Oral Surg Oral Med Oral Pathol. 1990;69(3):287-90. and central and/or peripheral sensitization33 Baad-Hansen L. Atypical odontalgia - pathophysiology and clinical management. J Oral Rehabil. 2008;35(1):1-11.,1919 List T, Leijon G, Svensson P. Somatosensory abnormalities in atypical odontalgia: a case-control study. Pain. 2008;139(2):333-41.

20 Bosch-Aranda ML, Vázquez-Delgado E, Gay-Escoda C. Atypical odontalgia: a systematic review following the evidence-based principles of dentistry. Cranio. 2011;29(3):219-26.
-2121 Pigg M, Svensson P, Drangsholt M, List T. Seven-year follow-up of patients diagnosed with atypical odontalgia: a prospective study. J Orofac Pain. 2013;27(2):151-64.. And, given this scenario, ICHD-3 classifies it as a subtype of persistent idiopathic facial pain (ICHD 13.12). If it is based on a history of trauma, it will be a subtype of posttraumatic trigeminal neuropathic pain (ICHD 13.1.2.3).

Although there is a well-defined diagnostic criterion by ICHD-3 for AO, there is no protocol on how to perform it2222 Abiko Y, Matsuoka H, Chiba I, Toyofuku A. Current evidence on atypical odontalgia: diagnosis and clinical management. Int J Dent. 2012;2012:518548.. Detailed clinical examination, work among various specialists, and listening to the patient about their dental history are proposed2323 Tarce M, Barbieri C, Sardella A. Atypical odontalgia: an up-to-date view. Minerva Stomatol. 2013;62(5):163-81.,2424 Forssell H, Jääskeläinen S, List T, Svensson P, Baad-Hansen L. An update on pathophysiological mechanisms related to idiopathic orofacial pain conditions with implications for management. J Oral Rehabil. 2015;42(4):300-22.. Some studies have shown that patients with AO have altered responses to qualitative and quantitative somatosensory tests33 Baad-Hansen L. Atypical odontalgia - pathophysiology and clinical management. J Oral Rehabil. 2008;35(1):1-11.,1919 List T, Leijon G, Svensson P. Somatosensory abnormalities in atypical odontalgia: a case-control study. Pain. 2008;139(2):333-41..

Currently, there is insufficient evidence to establish a treatment protocol for AO2525 García-Sáez R, Gutiérrez-Viedma A, González-García N, Gómez-Mayordomo V, Porta-Etessam J, Cuadrado ML. Onabotulinumtoxin A injections for atypical odontalgia: an open-label study on nine patients. J Pain Res. 2018;11:1583-8.. Tricyclic antidepressants, antiepileptics, anesthetics, and botulinum toxin, although reducing the pain of the patient33 Baad-Hansen L. Atypical odontalgia - pathophysiology and clinical management. J Oral Rehabil. 2008;35(1):1-11.,2222 Abiko Y, Matsuoka H, Chiba I, Toyofuku A. Current evidence on atypical odontalgia: diagnosis and clinical management. Int J Dent. 2012;2012:518548.,2525 García-Sáez R, Gutiérrez-Viedma A, González-García N, Gómez-Mayordomo V, Porta-Etessam J, Cuadrado ML. Onabotulinumtoxin A injections for atypical odontalgia: an open-label study on nine patients. J Pain Res. 2018;11:1583-8., have limited activity and have no proven effectiveness2525 García-Sáez R, Gutiérrez-Viedma A, González-García N, Gómez-Mayordomo V, Porta-Etessam J, Cuadrado ML. Onabotulinumtoxin A injections for atypical odontalgia: an open-label study on nine patients. J Pain Res. 2018;11:1583-8.

26 Cuadrado ML, García-Moreno H, Arias JA, Pareja JA. Botulinum neurotoxin type-A for the treatment of atypical odontalgia. Pain Med. 2016;17(9):1717-21.
-2727 Yatani H, Komiyama O, Matsuka Y, Wajima K, Muraoka W, Ikawa M, et al. Systematic review and recommendations for nonodontogenic toothache. J Oral Rehabil. 2014;41(11):843-52..

Generally, the dental surgeon is the first healthcare professional with whom the AO patient consults. The lack of knowledge of this situation by the dentist can lead to unnecessary and mutilating dental procedures, such as endodontic and surgical treatments ranging from apicectomy to extraction2828 Baad-Hansen L, Benoliel R. Neuropathic orofacial pain: facts and fiction. Cephalalgia. 2017;37(7):670-9.. Knowing the physiopathology and the diagnostic process allows the proper treatment, avoiding further injury to the patient2929 Ram S, Teruel A, Kumar SK, Clark G. Clinical characteristics and diagnosis of atypical odontalgia: implications for dentists. J Am Dent Assoc. 2009;140(2):223-8..

Given a scenario in which there is no consensus in the literature on the physiopathology, the diagnostic process, and its treatment, this study aimed to review these aspects, assisting the dentist in his/her professional activity.

CONTENTS

This is an integrative literature review with a qualitative approach to identify physiopathological, diagnostic, and treatment aspects of AO. The methodological process was divided into 5 steps, according to Whittemore and Knafl3030 Whittemore R, Knafl K. The integrative review: updated methodology. J Adv Nurs. 2005;52(5):546-53.: 1) problem identification; 2) literature search; 3) assessment of information; 4) critical analysis of the information; 5) presentation of results.

A search was performed in the Pubmed database. As a search strategy, the following terms were used: "atypical odontalgia" OR "phantom tooth pain" OR "idiopathic tooth pain" OR "odontalgia" OR "odontalgias" OR "atypical toothache”. The inclusion criteria were articles published in the last 10 years, in English, clinical trial, multicenter study, case report, review, integrative scientific review, and systematic review. After reading the title and the abstract, those who had no relation to the theme were excluded. In case of uncertainty of inclusion, the full article was read.

A total of 114 articles were found, and after applying the established inclusion criteria, 48 articles were chosen. Of these, 9 were excluded because they were not related to the theme, totaling 39 studies. The material was grouped according to the emphasis of the article: physiopathology, diagnosis and treatment. Information relevant to both steps is summarized in table 2.

Table 2
Selected studies and relevant information

PHYSIOPATHOLOGY

Current evidence suggests neuropathic mechanisms to explain the physiopathology of AO11 Malacarne A, Spierings EL, Lu C, Maloney GE. Persistent dentoalveolar pain disorder: a comprehensive review. J Endod. 2018;44(2):206-11.,2424 Forssell H, Jääskeläinen S, List T, Svensson P, Baad-Hansen L. An update on pathophysiological mechanisms related to idiopathic orofacial pain conditions with implications for management. J Oral Rehabil. 2015;42(4):300-22.,4141 Porporatti AL, Costa YM, Stuginski-Barbosa J, Bonjardim LR, Conti PC. Effect of topical anaesthesia in patients with persistent dentoalveolar pain disorders: A quantitative sensory testing evaluation. Arch Oral Biol. 2015;60(7):973-81.,4444 Baad-Hansen L, Pigg M, Ivanovic SE, Faris H, List T, Drangsholt M, et al. Intraoral somatosensory abnormalities in patients with atypical odontalgia--a controlled multicenter quantitative sensory testing study. Pain. 2013;154(8):1287-94.,4545 Baad-Hansen L, Pigg M, Ivanovic SE, Faris H, List T, Drangsholt M, et al. Chairside intraoral qualitative somatosensory testing: reliability and comparison between patients with atypical odontalgia and healthy controls. J Orofac Pain. 2013;27(2):165-70.,5050 Thorburn DN, Polonowita AD. Atypical odontalgia--a diagnostic dilemma. N Z Dent J. 2012;108(2):62-7.) and the somatosensory tests suggest its description in central and peripheral1919 List T, Leijon G, Svensson P. Somatosensory abnormalities in atypical odontalgia: a case-control study. Pain. 2008;139(2):333-41.,2323 Tarce M, Barbieri C, Sardella A. Atypical odontalgia: an up-to-date view. Minerva Stomatol. 2013;62(5):163-81.,3838 Rafael B, Sorin T, Eli E. Painful traumatic trigeminal neuropathy. Oral Maxillofac Surg Clin North Am. 2016;28(3):371-80.,4747 Tinastepe N, Oral K. Neuropathic pain after dental treatment. Agri. 2013;25(1):1-6.,5252 Zagury JG, Eliav E, Heir GM, Nasri-Heir C, Ananthan S, Pertes R, et al. Prolonged gingival cold allodynia: a novel finding in patients with atypical odontalgia. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2011;111(3):312-9..

One of the tests performed is local anesthesia, which, when observing pain reduction, suggests the neuropathic mechanism of peripheral origin (peripheral sensitization)4141 Porporatti AL, Costa YM, Stuginski-Barbosa J, Bonjardim LR, Conti PC. Effect of topical anaesthesia in patients with persistent dentoalveolar pain disorders: A quantitative sensory testing evaluation. Arch Oral Biol. 2015;60(7):973-81.. The quantitative somatosensory testing (QST) used are based on the pain threshold using a mechanical stimulus and heat pain threshold tests, for example4040 Porporatti AL, Costa YM, Stuginski-Barbosa J, Bonjardim LR, Conti PC, Svensson P. Quantitative methods for somatosensory evaluation in atypical odontalgia. Braz Oral Res. 2015;29. pii:S1806-83242015000100400.. Sensory alterations after cold application were identified in AO patients, also suggesting the involvement of central neuropathic mechanisms5252 Zagury JG, Eliav E, Heir GM, Nasri-Heir C, Ananthan S, Pertes R, et al. Prolonged gingival cold allodynia: a novel finding in patients with atypical odontalgia. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2011;111(3):312-9.. Qualitative somatosensory testing (QualST) were also used to confirm neuropathic involvement4545 Baad-Hansen L, Pigg M, Ivanovic SE, Faris H, List T, Drangsholt M, et al. Chairside intraoral qualitative somatosensory testing: reliability and comparison between patients with atypical odontalgia and healthy controls. J Orofac Pain. 2013;27(2):165-70..

In addition to these mechanisms, other neuropathic physiopathological events may be involved: nerve damage and ectopic activity due to the formation of neuromas, phenotypic changes, and increased sympathetic activity in times of stress or anxiety3838 Rafael B, Sorin T, Eli E. Painful traumatic trigeminal neuropathy. Oral Maxillofac Surg Clin North Am. 2016;28(3):371-80..

Recent studies do not point to psychiatric comorbidity as a determining cause for triggering AO1515 Ciaramella A, Paroli M, Lonia L, Bosco M, Poli P. Biopsychosocial aspects of atypical odontalgia. ISRN Neurosci. 2013;2013:413515., but professionals should be aware of this condition3636 Benoliel R, Gaul C. Persistent idiopathic facial pain. Cephalalgia. 2017;37(7):680-91. and cannot disregard it2222 Abiko Y, Matsuoka H, Chiba I, Toyofuku A. Current evidence on atypical odontalgia: diagnosis and clinical management. Int J Dent. 2012;2012:518548.. A high incidence of AO patients presents these comorbidities2222 Abiko Y, Matsuoka H, Chiba I, Toyofuku A. Current evidence on atypical odontalgia: diagnosis and clinical management. Int J Dent. 2012;2012:518548.,3333 Takenoshita M, Miura A, Shinohara Y, Mikuzuki R, Sugawara S, Tu TTH, et al. Clinical features of atypical odontalgia; three cases and literature reviews. Biopsychosoc Med. 2017;11:21., reaching 50% in another study1616 Miura A, Tu TTH, Shinohara Y, Mikuzuki L, Kawasaki K, Sugawara S, et al. Psychiatric comorbidities in patients with atypical odontalgia. J Psychosom Res. 2018;104:35-40.. “Neurotic and stressed”1616 Miura A, Tu TTH, Shinohara Y, Mikuzuki L, Kawasaki K, Sugawara S, et al. Psychiatric comorbidities in patients with atypical odontalgia. J Psychosom Res. 2018;104:35-40. and “resentful and depressed”1515 Ciaramella A, Paroli M, Lonia L, Bosco M, Poli P. Biopsychosocial aspects of atypical odontalgia. ISRN Neurosci. 2013;2013:413515. were striking characteristics described in individuals with AO. Moreover, such comorbidities may determine a predisposition to the development of chronic pain after extraction1515 Ciaramella A, Paroli M, Lonia L, Bosco M, Poli P. Biopsychosocial aspects of atypical odontalgia. ISRN Neurosci. 2013;2013:413515.. Tu et al.3232 Tu TTH, Miura A, Shinohara Y, Mikuzuki L, Kawasaki K, Sugawara S, et al. Evaluating burning mouth syndrome as a comorbidity of atypical odontalgia: the impact on pain experiences. Pain Pract. 2018;18(5):580-6., however, concluded that psychiatric comorbidity in patients with AO and mouth burning syndrome had little impact on pain experience.

The vascular cause presented by Rees and Harris77 Rees RT, Harris M. Atypical odontalgia. Br J Oral Surg. 1979;16(3):212-8. and Kreisberg1717 Kreisberg MK. Atypical odontalgia: differential diagnosis and treatment. J Am Dent Assoc. 1982;104(6):852-4. was mentioned in only two studies2020 Bosch-Aranda ML, Vázquez-Delgado E, Gay-Escoda C. Atypical odontalgia: a systematic review following the evidence-based principles of dentistry. Cranio. 2011;29(3):219-26.,2222 Abiko Y, Matsuoka H, Chiba I, Toyofuku A. Current evidence on atypical odontalgia: diagnosis and clinical management. Int J Dent. 2012;2012:518548., thus not being the main physiopathological mechanism of AO.

DIAGNOSIS

There is no gold standard diagnostic protocol for AO(28,34,) and existing ones are not sufficiently reliable for diagnosis11 Malacarne A, Spierings EL, Lu C, Maloney GE. Persistent dentoalveolar pain disorder: a comprehensive review. J Endod. 2018;44(2):206-11.. Since the physiopathology is not well defined2020 Bosch-Aranda ML, Vázquez-Delgado E, Gay-Escoda C. Atypical odontalgia: a systematic review following the evidence-based principles of dentistry. Cranio. 2011;29(3):219-26.,2323 Tarce M, Barbieri C, Sardella A. Atypical odontalgia: an up-to-date view. Minerva Stomatol. 2013;62(5):163-81., its diagnosis is often by exclusion3434 Tait RC, Ferguson M, Herndon CM. Chronic orofacial pain: burning mouth syndrome and other neuropathic disorders. J Pain Manag Med. 2017;3(1). pii: 120.,5050 Thorburn DN, Polonowita AD. Atypical odontalgia--a diagnostic dilemma. N Z Dent J. 2012;108(2):62-7..

Even in the face of insufficient information for the elaboration of a diagnostic protocol2222 Abiko Y, Matsuoka H, Chiba I, Toyofuku A. Current evidence on atypical odontalgia: diagnosis and clinical management. Int J Dent. 2012;2012:518548., after analyzing the data extracted from this study, it was possible to synthesize the main information for the professional who may be facing a diagnosis of AO:

  • 1) Importance of medical history: patient assessment should begin with its medical history, especially with regard to pain characteristics2323 Tarce M, Barbieri C, Sardella A. Atypical odontalgia: an up-to-date view. Minerva Stomatol. 2013;62(5):163-81.;

  • 2) Importance of the clinical examination: the odontogenic causes of toothache must be totally ruled out. For this, a thorough clinical examination is necessary55 Durham J, Stone SJ, Robinson LJ, Ohrbach R, Nixdorf DR. Development and preliminary evaluation of a new screening instrument for atypical odontalgia and persistent dentoalveolar pain disorder. Int Endod J. 2019;52(3):279-87.,1010 Porporatti AL, Costa YM, Stuginski-Barbosa J, Bonjardim LR, Duarte MA, Conti PC. Diagnostic accuracy of quantitative sensory testing to discriminate inflammatory toothache and intraoral neuropathic pain. J Endod. 2015;41(10):1606-13.,2424 Forssell H, Jääskeläinen S, List T, Svensson P, Baad-Hansen L. An update on pathophysiological mechanisms related to idiopathic orofacial pain conditions with implications for management. J Oral Rehabil. 2015;42(4):300-22.,4040 Porporatti AL, Costa YM, Stuginski-Barbosa J, Bonjardim LR, Conti PC, Svensson P. Quantitative methods for somatosensory evaluation in atypical odontalgia. Braz Oral Res. 2015;29. pii:S1806-83242015000100400.. One should not forget Rees and Harris’s observations77 Rees RT, Harris M. Atypical odontalgia. Br J Oral Surg. 1979;16(3):212-8. emphasizing that all possibilities of caries, pulp disease and crack/fracture of the crown or root should be excluded;

  • 3) Complementary imaging tests: despite the limitations of periapical radiographs5454 Durham J, Stone SJ, Robinson LJ, Ohrbach R, Nixdorf DR. Developing and preliminary evaluation of a new screening instrument for atypical odontalgia and persistent dentoalveolar pain disorder. Int Endod J. 2019;52(3):279-87.,5555 Velvart P, Hecker H, Tillinger G. Detection of the apical lesion and the mandibular canal in conventional radiography and computed tomography. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2001;92(6):682-8. they should be used to assess the periapical region. Volumetric computed tomography should be performed to rule out any possibility of periapical endodontic alteration1010 Porporatti AL, Costa YM, Stuginski-Barbosa J, Bonjardim LR, Duarte MA, Conti PC. Diagnostic accuracy of quantitative sensory testing to discriminate inflammatory toothache and intraoral neuropathic pain. J Endod. 2015;41(10):1606-13.,5151 Pigg M, List T, Petersson K, Lindh C, Petersson A. Diagnostic yield of conventional radiographic and cone-beam computed tomographic images in patients with atypical odontalgia. Int Endod J. 2011;44(12):1092-101.. The use of magnetic resonance imaging (MRI), in cases of suspected non-inflammatory dental pain, can be of great value as it excludes inflammation processes in the mandibular and maxillary region. When the diagnosis is uncertain, MRI reinforces the importance of noninvasive management4343 Pigg M, List T, Abul-Kasim K, Maly P, Petersson A. A comparative analysis of magnetic resonance imaging and radiographic examinations of patients with atypical odontalgia. J Oral Facial Pain Headache. 2014;28(3):233-42.;

  • 4) In order to facilitate and assist the diagnostic process, two tools should be highlighted:

    • a) Visual analog scale: diagnostic tool for pain measurement4141 Porporatti AL, Costa YM, Stuginski-Barbosa J, Bonjardim LR, Conti PC. Effect of topical anaesthesia in patients with persistent dentoalveolar pain disorders: A quantitative sensory testing evaluation. Arch Oral Biol. 2015;60(7):973-81.,4545 Baad-Hansen L, Pigg M, Ivanovic SE, Faris H, List T, Drangsholt M, et al. Chairside intraoral qualitative somatosensory testing: reliability and comparison between patients with atypical odontalgia and healthy controls. J Orofac Pain. 2013;27(2):165-70.:

    • b) QST and QualST: are important allies in the diagnosis of AO2424 Forssell H, Jääskeläinen S, List T, Svensson P, Baad-Hansen L. An update on pathophysiological mechanisms related to idiopathic orofacial pain conditions with implications for management. J Oral Rehabil. 2015;42(4):300-22.,2727 Yatani H, Komiyama O, Matsuka Y, Wajima K, Muraoka W, Ikawa M, et al. Systematic review and recommendations for nonodontogenic toothache. J Oral Rehabil. 2014;41(11):843-52.,4040 Porporatti AL, Costa YM, Stuginski-Barbosa J, Bonjardim LR, Conti PC, Svensson P. Quantitative methods for somatosensory evaluation in atypical odontalgia. Braz Oral Res. 2015;29. pii:S1806-83242015000100400.,4444 Baad-Hansen L, Pigg M, Ivanovic SE, Faris H, List T, Drangsholt M, et al. Intraoral somatosensory abnormalities in patients with atypical odontalgia--a controlled multicenter quantitative sensory testing study. Pain. 2013;154(8):1287-94.,4545 Baad-Hansen L, Pigg M, Ivanovic SE, Faris H, List T, Drangsholt M, et al. Chairside intraoral qualitative somatosensory testing: reliability and comparison between patients with atypical odontalgia and healthy controls. J Orofac Pain. 2013;27(2):165-70.,5252 Zagury JG, Eliav E, Heir GM, Nasri-Heir C, Ananthan S, Pertes R, et al. Prolonged gingival cold allodynia: a novel finding in patients with atypical odontalgia. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2011;111(3):312-9.. QST is performed through several stimuli, and only mechanical and thermal stimuli are related to AO. Of the patients with AO submitted to these stimuli, 83.7% had some QST abnormality4444 Baad-Hansen L, Pigg M, Ivanovic SE, Faris H, List T, Drangsholt M, et al. Intraoral somatosensory abnormalities in patients with atypical odontalgia--a controlled multicenter quantitative sensory testing study. Pain. 2013;154(8):1287-94.. Performing bilateral QST (pain side versus pain free side) also helps to detect neuropathic changes1010 Porporatti AL, Costa YM, Stuginski-Barbosa J, Bonjardim LR, Duarte MA, Conti PC. Diagnostic accuracy of quantitative sensory testing to discriminate inflammatory toothache and intraoral neuropathic pain. J Endod. 2015;41(10):1606-13.. Despite the indications, QST, when used outside hospitals and university clinics, is costly and often unfeasible, requiring the calibration and training of examiners4545 Baad-Hansen L, Pigg M, Ivanovic SE, Faris H, List T, Drangsholt M, et al. Chairside intraoral qualitative somatosensory testing: reliability and comparison between patients with atypical odontalgia and healthy controls. J Orofac Pain. 2013;27(2):165-70.. QualST detects hypersensitivity disorders to touch, cold, and bristle stimulation4646 Zakrzewska JM. Multi-dimensionality of chronic pain of the oral cavity and face. J Headache Pain. 2013;14:37..

  • 5) Exclude all hypotheses of non-odontogenic odontalgia. According to Yatani et al.2727 Yatani H, Komiyama O, Matsuka Y, Wajima K, Muraoka W, Ikawa M, et al. Systematic review and recommendations for nonodontogenic toothache. J Oral Rehabil. 2014;41(11):843-52. and ICHD-3, after discarding the hypothesis of dental pain, there are numerous other conditions of non-odontogenic origin that should be ruled out;

  • 6) Refer the patient to other specialists: Given the difficulty of properly diagnosing and the various physiopathological mechanisms that could be involved, it is recommended to refer the patient to other specialists2323 Tarce M, Barbieri C, Sardella A. Atypical odontalgia: an up-to-date view. Minerva Stomatol. 2013;62(5):163-81.,2424 Forssell H, Jääskeläinen S, List T, Svensson P, Baad-Hansen L. An update on pathophysiological mechanisms related to idiopathic orofacial pain conditions with implications for management. J Oral Rehabil. 2015;42(4):300-22.,3636 Benoliel R, Gaul C. Persistent idiopathic facial pain. Cephalalgia. 2017;37(7):680-91.,5757 Zakrzewska JM. Facial pain: an update. Curr Opin Support Palliat Care. 2009;3(2):125-30.. Interesting to note that in 1982, Kreisberg1717 Kreisberg MK. Atypical odontalgia: differential diagnosis and treatment. J Am Dent Assoc. 1982;104(6):852-4. already suggested referral to the neurologist;

  • 7) Consider psychological aspects: Although psychogenic and psychiatric factors have no determining relationship in the development of AO1616 Miura A, Tu TTH, Shinohara Y, Mikuzuki L, Kawasaki K, Sugawara S, et al. Psychiatric comorbidities in patients with atypical odontalgia. J Psychosom Res. 2018;104:35-40., there was a high incidence of these patients with psychiatric comorbidities1414 Takenoshita M, Sato T, Kato Y, Katagiri A, Yoshikawa T, Sato 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.

    15 Ciaramella A, Paroli M, Lonia L, Bosco M, Poli P. Biopsychosocial aspects of atypical odontalgia. ISRN Neurosci. 2013;2013:413515.
    -1616 Miura A, Tu TTH, Shinohara Y, Mikuzuki L, Kawasaki K, Sugawara S, et al. Psychiatric comorbidities in patients with atypical odontalgia. J Psychosom Res. 2018;104:35-40.,2222 Abiko Y, Matsuoka H, Chiba I, Toyofuku A. Current evidence on atypical odontalgia: diagnosis and clinical management. Int J Dent. 2012;2012:518548.,3232 Tu TTH, Miura A, Shinohara Y, Mikuzuki L, Kawasaki K, Sugawara S, et al. Evaluating burning mouth syndrome as a comorbidity of atypical odontalgia: the impact on pain experiences. Pain Pract. 2018;18(5):580-6.,3636 Benoliel R, Gaul C. Persistent idiopathic facial pain. Cephalalgia. 2017;37(7):680-91.. The professional should be aware of these comorbidities, giving AO a multifactorial etiology3131 Ghurye S, McMillan R. Orofacial pain - an update on diagnosis and management. Br Dent J. 2017;223(9):639-47.,3333 Takenoshita M, Miura A, Shinohara Y, Mikuzuki R, Sugawara S, Tu TTH, et al. Clinical features of atypical odontalgia; three cases and literature reviews. Biopsychosoc Med. 2017;11:21.. Thus, a biopsychosocial3131 Ghurye S, McMillan R. Orofacial pain - an update on diagnosis and management. Br Dent J. 2017;223(9):639-47. and interdisciplinary3636 Benoliel R, Gaul C. Persistent idiopathic facial pain. Cephalalgia. 2017;37(7):680-91. and no less priority3939 Toyofuku A. Psychosomatic problems in dentistry. Biopsychosoc Med. 2016;10:14. approach are necessary3939 Toyofuku A. Psychosomatic problems in dentistry. Biopsychosoc Med. 2016;10:14.;

  • 8) A more holistic, psychosocial, and not purely mechanical approach is important. It is recommended to listen carefully to the patient’s complaint and his/her history of treatments5656 Lofthag-Hansen S, Huumonen S, Gröndahl K, Gröndahl HG. Limited cone-beam CT and intraoral radiography for the diagnosis of periapical pathology. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2007;103(1):114-9.;

  • 9) Knowledge and training by professionals are important to avoid unnecessary and iatrogenic procedures2828 Baad-Hansen L, Benoliel R. Neuropathic orofacial pain: facts and fiction. Cephalalgia. 2017;37(7):670-9..

TREATMENT

Like diagnosis, AO treatment is challenging2323 Tarce M, Barbieri C, Sardella A. Atypical odontalgia: an up-to-date view. Minerva Stomatol. 2013;62(5):163-81.,5050 Thorburn DN, Polonowita AD. Atypical odontalgia--a diagnostic dilemma. N Z Dent J. 2012;108(2):62-7.. Currently, there is insufficient evidence to establish a treatment protocol2525 García-Sáez R, Gutiérrez-Viedma A, González-García N, Gómez-Mayordomo V, Porta-Etessam J, Cuadrado ML. Onabotulinumtoxin A injections for atypical odontalgia: an open-label study on nine patients. J Pain Res. 2018;11:1583-8..

Tricyclic antidepressants are the most cited drugs in case reports and case-control studies, and for many authors, they are considered the first choice in treatment2727 Yatani H, Komiyama O, Matsuka Y, Wajima K, Muraoka W, Ikawa M, et al. Systematic review and recommendations for nonodontogenic toothache. J Oral Rehabil. 2014;41(11):843-52.,3535 Kobayashi Y, Nagashima W, Tokura T, Yoshida K, Umemura E, Miyauchi T, et al. Duloxetine plasma concentrations and its effectiveness in the treatment of nonorganic chronic pain in the orofacial region. Clin Neuropharmacol. 2017;40(4):163-8.. However, these drugs cause adverse effects. Amitriptyline, for example, causes xerostomia, constipation, urinary retention, and weight gain2020 Bosch-Aranda ML, Vázquez-Delgado E, Gay-Escoda C. Atypical odontalgia: a systematic review following the evidence-based principles of dentistry. Cranio. 2011;29(3):219-26. and, depending on the dose and the patient, have varied responses regarding the effectiveness in pain remission3333 Takenoshita M, Miura A, Shinohara Y, Mikuzuki R, Sugawara S, Tu TTH, et al. Clinical features of atypical odontalgia; three cases and literature reviews. Biopsychosoc Med. 2017;11:21.. Serotonin and norepinephrine reuptake inhibitors, such as milnacipran and duloxetine, have also been used in the management of painful symptoms3535 Kobayashi Y, Nagashima W, Tokura T, Yoshida K, Umemura E, Miyauchi T, et al. Duloxetine plasma concentrations and its effectiveness in the treatment of nonorganic chronic pain in the orofacial region. Clin Neuropharmacol. 2017;40(4):163-8.,4848 Nagashima W, Kimura H, Ito M, Tokura T, Arao M, Aleksic B, et al. Effectiveness of duloxetine for the treatment of chronic nonorganic orofacial pain. Clin Neuropharmacol. 2012;35(6):273-7.,5353 Ito M, Kimura H, Yoshida K, Kimura Y, Ozaki N, Kurita K. Effectiveness of milnacipran for the treatment of chronic pain in the orofacial region. Clin Neuropharmacol. 2010;33(2):79-83., and although they have pain reduction, there is a need for randomized controlled trials (RCT) to prove its real effectiveness2020 Bosch-Aranda ML, Vázquez-Delgado E, Gay-Escoda C. Atypical odontalgia: a systematic review following the evidence-based principles of dentistry. Cranio. 2011;29(3):219-26.,2121 Pigg M, Svensson P, Drangsholt M, List T. Seven-year follow-up of patients diagnosed with atypical odontalgia: a prospective study. J Orofac Pain. 2013;27(2):151-64.,2323 Tarce M, Barbieri C, Sardella A. Atypical odontalgia: an up-to-date view. Minerva Stomatol. 2013;62(5):163-81..

As already described, current evidence suggests neuropathic mechanisms to explain the physiopathology of AO11 Malacarne A, Spierings EL, Lu C, Maloney GE. Persistent dentoalveolar pain disorder: a comprehensive review. J Endod. 2018;44(2):206-11.,2424 Forssell H, Jääskeläinen S, List T, Svensson P, Baad-Hansen L. An update on pathophysiological mechanisms related to idiopathic orofacial pain conditions with implications for management. J Oral Rehabil. 2015;42(4):300-22.,4141 Porporatti AL, Costa YM, Stuginski-Barbosa J, Bonjardim LR, Conti PC. Effect of topical anaesthesia in patients with persistent dentoalveolar pain disorders: A quantitative sensory testing evaluation. Arch Oral Biol. 2015;60(7):973-81.,4444 Baad-Hansen L, Pigg M, Ivanovic SE, Faris H, List T, Drangsholt M, et al. Intraoral somatosensory abnormalities in patients with atypical odontalgia--a controlled multicenter quantitative sensory testing study. Pain. 2013;154(8):1287-94.,4545 Baad-Hansen L, Pigg M, Ivanovic SE, Faris H, List T, Drangsholt M, et al. Chairside intraoral qualitative somatosensory testing: reliability and comparison between patients with atypical odontalgia and healthy controls. J Orofac Pain. 2013;27(2):165-70.,5050 Thorburn DN, Polonowita AD. Atypical odontalgia--a diagnostic dilemma. N Z Dent J. 2012;108(2):62-7.. Thus, treating it as a neuropathy sounds coherent3131 Ghurye S, McMillan R. Orofacial pain - an update on diagnosis and management. Br Dent J. 2017;223(9):639-47.. However, results with therapies employed for neuropathic orofacial pain have been disappointing in AO studies3434 Tait RC, Ferguson M, Herndon CM. Chronic orofacial pain: burning mouth syndrome and other neuropathic disorders. J Pain Manag Med. 2017;3(1). pii: 120..

More recent studies have assessed the action of botulinum neurotoxin type A (Onabotulinum toxin A) in pain control. The good results regarding pain remission point it as a promising drug in the treatment of AO. However, as with tricyclic antidepressants and serotonin and norepinephrine reuptake inhibitors, the use of botulinum neurotoxin type A should be proven to be effective through more RCT2020 Bosch-Aranda ML, Vázquez-Delgado E, Gay-Escoda C. Atypical odontalgia: a systematic review following the evidence-based principles of dentistry. Cranio. 2011;29(3):219-26.,2525 García-Sáez R, Gutiérrez-Viedma A, González-García N, Gómez-Mayordomo V, Porta-Etessam J, Cuadrado ML. Onabotulinumtoxin A injections for atypical odontalgia: an open-label study on nine patients. J Pain Res. 2018;11:1583-8.,2626 Cuadrado ML, García-Moreno H, Arias JA, Pareja JA. Botulinum neurotoxin type-A for the treatment of atypical odontalgia. Pain Med. 2016;17(9):1717-21..

Thus, the information obtained from the articles found can be summarized:

  • 1) In cases of doubt, not performing endodontic and surgical treatments, as AO would be unnecessary and worsen the patient’s pain4949 Patel SB, Boros AL, Kumar SK. Atypical odontalgia--an update. J Calif Dent Assoc. 2012;40(9):739-47.;

  • 2) Knowledge and training by professionals in the diagnostic process are essential to avoid unnecessary and iatrogenic procedures2828 Baad-Hansen L, Benoliel R. Neuropathic orofacial pain: facts and fiction. Cephalalgia. 2017;37(7):670-9.;

  • 3) Interdisciplinary work is important not only in the diagnosis, but also in the institution of the correct treatment3636 Benoliel R, Gaul C. Persistent idiopathic facial pain. Cephalalgia. 2017;37(7):680-91.,5050 Thorburn DN, Polonowita AD. Atypical odontalgia--a diagnostic dilemma. N Z Dent J. 2012;108(2):62-7.;

  • 4) RCTs are necessary to assess the effectiveness of tricyclic antidepressants, serotonin and norepinephrine reuptake inhibitors and botulinum neurotoxin type A2121 Pigg M, Svensson P, Drangsholt M, List T. Seven-year follow-up of patients diagnosed with atypical odontalgia: a prospective study. J Orofac Pain. 2013;27(2):151-64.,2323 Tarce M, Barbieri C, Sardella A. Atypical odontalgia: an up-to-date view. Minerva Stomatol. 2013;62(5):163-81.;

  • 5) Minimizing the pain of patients with the lowest drug dose is the main objective and inadvertent use without the need for several drugs should be avoided2020 Bosch-Aranda ML, Vázquez-Delgado E, Gay-Escoda C. Atypical odontalgia: a systematic review following the evidence-based principles of dentistry. Cranio. 2011;29(3):219-26.;

  • 6) And again, a holistic, psychosocial, and not purely mechanical approach is important. It is recommended to listen carefully to the patient’s complaint and his or her history of treatments5656 Lofthag-Hansen S, Huumonen S, Gröndahl K, Gröndahl HG. Limited cone-beam CT and intraoral radiography for the diagnosis of periapical pathology. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2007;103(1):114-9..

After these reflections, it is important to emphasize that this study had limitations regarding the choice of database for the selection of studies. However, Pubmed is considered the universal English language database with indexed high impact journals.

CONCLUSION

Recent studies use the 3rd edition of the ICHD classification, in which AO falls into the “persistent idiopathic facial pain” category (ICHD-13.12). Since the physiopathological process is not defined, the establishment of a protocol to make its diagnosis is fundamental. It is suggested knowledge about the other existing diseases to make a differential diagnosis, and the use of complementary exams such as volumetric computed tomography, MRI, and QualST. Tricyclic antidepressants and serotonin and norepinephrine reuptake inhibitors are the drugs of first choice in the treatment of AO. However, currently, the use of botulinum neurotoxin type A in pain management has been assessed. All these drugs require RCT to have their effectiveness proven. Given the possibility of AO, an interdisciplinary approach in the diagnostic process and definition of its treatment is guided.

  • Sponsoring sources: none.

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Publication Dates

  • Publication in this collection
    02 Dec 2019
  • Date of issue
    Oct-Dec 2019

History

  • Received
    04 Mar 2019
  • Accepted
    24 May 2019
Sociedade Brasileira para o Estudo da Dor Av. Conselheiro Rodrigues Alves, 937 Cj2 - Vila Mariana, CEP: 04014-012, São Paulo, SP - Brasil, Telefones: , (55) 11 5904-2881/3959 - São Paulo - SP - Brazil
E-mail: dor@dor.org.br