Persistent idiopathic facial pain
https://doi.org/10.21886/2712-8156-2025-6-4-51-60
Abstract
Currently, among all types of orofacial pain, the least studied is persistent idiopathic facial pain (PIFP), also known by the old name atypical facial pain. There are few studies on this topic, which is why this type of prosopalgia continues to be a diagnosis of exclusion, and the frequency of misdiagnosis is extremely high. A search was conducted for literary sources in the databases PubMed, Web of Science, Scopus, and eLibrary to select articles published from 1924 to 2025 using the keywords “persistent idiopathic facial pain” and “atypical facial pain”. The review examines the prevalence, views on etiopathogenesis, clinical manifestations, diagnosis and treatment of PILB.
About the Authors
E. V. Balyazina
Rostov State Medical University
Russian Federation
Russian Federation
Elena V. Balyazina, Professor of the Department of Neurology and Neurosurgery
Rostov-on-Don
O. M. Zykova
Rostov State Medical University
Russian Federation
Russian Federation
Oksana M. Zykova, PhD student of the Department of Neurology and Regenerative Medicine with a course in osteopathy
Rostov-on-Don
A. V. Afanasyeva
Rostov State Medical University
Russian Federation
Russian Federation
Anastasia V. Afanasyeva, student
Rostov-on-Don
L. Z. Suleymanova
Rostov State Medical University
Russian Federation
Russian Federation
Luara Z. Suleymanova, neurologist
Rostov-on-Don
References
1. Forssell H, Tasmuth T, Tenovuo O, Hampf G, Kalso E. Venlafaxine in the treatment of atypical facial pain: a randomized controlled trial. J Orofac Pain. 2004;18(2):131-137. PMID: 15250433.
2. Gerwin R. Chronic Facial Pain: Trigeminal Neuralgia, Persistent Idiopathic Facial Pain, and Myofascial Pain Syndrome-An Evidence-Based Narrative Review and Etiological Hypothesis. Int J Environ Res Public Health. 2020;17(19):7012. doi: 10.3390/ijerph17197012
3. Parkhomenko E.V., Nartov S.E., Karpov D.Yu. Persistent idiopathic facial pain: a difficult path to a complex diagnosis. RMJ. 2017;(24):1738–1744. (In Russ.) eLIBRARY ID: 32244038 EDN: YLAYNT
4. Zakrzewska JM, Jensen TS. History of facial pain diagnosis. Cephalalgia. 2017;37(7):604-608. doi: 10.1177/0333102417691045
5. Balyazin VA, Balyazin EV. Ways of managing patients and relapses after microvascular decompression in patients with trigeminal neuralgia. Burdenko’s Journal of Neurosurgery. 2003;2:6-9. (In Russ.).
6. Zakrzewska JM. Multi-dimensionality of chronic pain of the oral cavity and face. J Headache Pain. 2013;14(1):37. doi: 10.1186/1129-2377-14-37
7. Patel SK, Markosian C, Choudhry OJ, Keller JT, Liu JK. The historical evolution of microvascular decompression for trigeminal neuralgia: from Dandy's discovery to Jannetta's legacy. Acta Neurochir (Wien). 2020;162(11):2773-2782. doi: 10.1007/s00701-020-04405-7
8. Frazier CH, Russell EC. Neuralgia of the face, an analysis of 754 cases with relation to pain and other sensory phenomena before and after operation. Arch NeurPsych. 1924;11(5):557–563. doi: 10.1001/archneurpsyc.1924.02190350063005
9. McElin TW, Horton BT. Atypical face pain; a statistical consideration of 66 cases. Ann Intern Med. 1947;27(5):749-768. doi: 10.7326/0003-4819-27-5-749
10. Rasmussen P. Facial pain. A clinical study with special reference to the symptomatology, aetiology and surgical therapy. Dan. Med. Bull. 1966;13(4):115-118.
11. Rushton JG, Gibilisco JA, Goldstein NP. Atypical face pain. J Am Med Assoc. 1959;171:545-548. doi: 10.1001/jama.1959.03010230043010
12. Campbell JK. Facial pain due to migraine and cluster headache. Semin Neurol. 1988;8(4):324-331. doi: 10.1055/s-2008-1041397
13. Raskin NH. On the origin of head pain. Headache. 1988;28(4):254-257. doi: 10.1111/j.1526-4610.1988.hed2804254.x
14. Benoliel R, Gaul C. Persistent idiopathic facial pain. Cephalalgia. 2017;37(7):680-691. doi: 10.1177/0333102417706349
15. Harris M, Feinmann C. Psychosomatic disorders. In: Jones HJ, Mason DK, eds. Oral manifestations of systemic disease, 2nd edn. Bailliere Tindall, London; 1990.
16. Woda A, Pionchon P. A unified concept of idiopathic orofacial pain: pathophysiologic features. J Orofac Pain. 2000;14(3):196-212. PMID: 11203755.
17. Clarkson E, Jung E. Atypical Facial Pain. Dent Clin North Am. 2020;64(1):249-253. doi: 10.1016/j.cden.2019.08.014
18. Evans RW, Agostoni E. Persistent idiopathic facial pain. Headache. 2006;46(8):1298-1300. doi: 10.1111/j.1526-4610.2006.00541.x
19. Cornelissen P, van Kleef M, Mekhail N, Day M, van Zundert J. Evidence-based interventional pain medicine according to clinical diagnoses. 3. Persistent idiopathic facial pain. Pain Pract. 2009;9(6):443-448. doi: 10.1111/j.1533-2500.2009.00332.x
20. Merskey H, Bogduk N. Classification of Chronic Pain, Task Force on Taxonomy, International Association for the Study of Pain. 2nd ed. Seattle: ISAP Press, 1994.
21. Türp JC, Gobetti JP. Trigeminal neuralgia versus atypical facial pain. A review of the literature and case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1996;81(4):424-432. doi: 10.1016/s1079-2104(96)80018-7
22. Classification and diagnostic criteria for headache disorders, cranial neuralgias and facial pain. Headache Classification Committee of the International Headache Society. Cephalalgia. 1988;8 Suppl 7:1-96. PMID: 3048700..
23. Headache Classification Subcommittee of the International Headache Society. The International Classification of Headache Disorders: 2nd edition. Cephalalgia. 2004;24 Suppl 1:9-160. doi: 10.1111/j.1468-2982.2003.00824.x
24. Maksimova M.Yu., Fedin P.A., Suanova E.T., Tyurnikov V.M. Neurophysiological features of atypical facial pain. Annals of clinical and experimental neurology. 2013;7(3):9-16. (In Russ.) eLIBRARY ID: 20313018 EDN: RCNGLF
25. Medvedev V.EH., Fofanova YU.S., Frolova V.I. Persistiruyushchie idiopaticheskie litsevye boli s pozitsii psikhosomaticheskoĬ meditsiny. The Russian Archives of Internal Medicine. 2015;(4):10-16. (In Russ.) https://doi.org/10.20514/2226-6704-2015-0-4-10-16
26. Headache Classification Committee of the International Headache Society (IHS) The International Classification of Headache Disorders, 3rd edition. Cephalalgia. 2018;38(1):1-211. doi: 10.1177/0333102417738202
27. International Classification of Orofacial Pain, 1st edition (ICOP). Cephalalgia. 2020;40(2):129-221. doi: 10.1177/0333102419893823
28. Fay T. Atypical facial neuralgia, a syndrome of vascular pain. Ann. Otol. Rhino1. Laryngol. 1932;41:1030-1062.
29. Friedman AP. Atypical facial pain. Headache. 1969;9(1):27-30. doi: 10.1111/j.1526-4610.1969.hed0901027.x
30. Sjaastad O. Headache and the influence of stress. A personal view. Ann Clin Res. 1987;19(2):122-128. PMID: 3662406.
31. Glaser MA. Atypical neuralgia, so called. A critical analysis of one hundred and forty-three cases. Arch. Neurol. Psychiatry. 1928;20:537-558.
32. Campbell AM, Lloyd JK. Atypical facial pain. Lancet. 1954;267(6847):1034-1038. doi: 10.1016/s0140-6736(54)90602-9
33. Nijensohn DE, Araujo JC, MacCarty CS. Meningiomas of Meckel's cave. J Neurosurg. 1975;43(2):197-202. doi: 10.3171/jns.1975.43.2.0197
34. Fee WE Jr, Epsy CD, Konrad HR. Trigeminal neurinomas. Laryngoscope. 1975;85(2):371-376. doi: 10.1288/00005537-197502000-00014
35. Garen PD, Powers JM, King JS, Perot PL Jr. Intracranial fibro-osseous lesion. Case report. J Neurosurg. 1989;70(3):475-477. doi: 10.3171/jns.1989.70.3.0475
36. Rushton JG, Gibilisco JA, Goldstein NP. Atypical face pain. J Am Med Assoc. 1959;171:545-548. doi: 10.1001/jama.1959.03010230043010
37. Bullitt E, Tew JM, Boyd J. Intracranial tumors in patients with facial pain. J Neurosurg. 1986;64(6):865-871. doi: 10.3171/jns.1986.64.6.0865
38. Pradat P, Guilly P, David M, Metzger J. Neuralgie faciale atypique datant de 35 ans: kyste epidermoide latero-protuberantiel; interet de la tomographie hypocycloide. Neurochirurgie. 1969;15:497-502.
39. Roberts AM, Person P. Etiology and treatment of idiopathic trigeminal and atypical facial neuralgias. Oral Surg Oral Med Oral Pathol. 1979;48(4):298-308. doi: 10.1016/0030-4220(79)90027-6
40. Roberts AM, Person P, Chandran NB, Hori JM. Further observations on dental parameters of trigeminal and atypical facial neuralgias. Oral Surg Oral Med Oral Pathol. 1984;58(2):121-129. doi: 10.1016/0030-4220(84)90123-3
41. List T, Leijon G, Helkimo M, Oster A, Dworkin SF, Svensson P. Clinical findings and psychosocial factors in patients with atypical odontalgia: a case-control study. J Orofac Pain. 2007;21(2):89-98. PMID: 17547120.
42. Garcia O, Okada M, Yeng L, et al. Atypical facial pain: Multidisciplinary assessment and treatment. Proceedings of the 9th World Congress on Pain, 22–27 August. Vienna, Austria; 1999.
43. Issrani R, Prabhu N, Mathur S. Atypical facial pain and atypical odontalgia: A concise review. Int. J. Contemp. Dent. Med. Rev. 2015;2015:8-11. doi: 10.15713/ins.ijcdmr.28
44. Melzack R, Wall PD. Pain mechanisms: a new theory. Science. 1965;150(3699):971-979. doi: 10.1126/science.150.3699.971
45. Sicuteri F, Nicolodi M, Fusco BM, Orlando S. Idiopathic headache as a possible risk factor for phantom tooth pain. Headache. 1991;31(9):577-581. doi: 10.1111/j.1526-4610.1991.hed3109577.x
46. Rees RT, Harris M. Atypical odontalgia: Differential diagnosis and treatment. Br. J. Oral Surg. 1978;16:212-214.
47. Jaeger B. Persistent Idiopathic Facial Pain. In: Gary Jay W, ed. Clinician’s Guide to Chronic Headache and Facial Pain. Location Boca; CRC Press, 2009.
48. List A, Feinmann C. Persistent idiopathic facial pain (atypical facial pain). In: Zakrzewska JM, ed. Orofacial Pain. Oxford: Oxford University Press, 2009.
49. Maier C, Hoffmeister B. Führung und Behandlung von Patienten mit atypischem Gesichtsschmerz [Management and treatment of patients with atypical facial pain]. Dtsch Zahnarztl Z. 1989;44(12):977-983. (In German). PMID: 2700719.
50. Baad-Hansen L, Benoliel R. Neuropathic orofacial pain: Facts and fiction. Cephalalgia. 2017;37(7):670-679. DOI: 10.1177/0333102417706310
51. Mock D, Frydman W, Gordon AS. Atypical facial pain: a retrospective study. Oral Surg Oral Med Oral Pathol. 1985;59(5):472-474. doi: 10.1016/0030-4220(85)90086-6
52. Remick RA, Blasberg B, Barton JS, Campos PE, Miles JE. Ineffective dental and surgical treatment associated with atypical facial pain. Oral Surg Oral Med Oral Pathol. 1983;55(4):355-358. doi: 10.1016/0030-4220(83)90189-5
53. Korjachkin V.A. Kompleksnyj regionarnyj bolevoj sindrom. Travmatologija i ortopedija Rossii. 2014;(3):147-156 (In Russ.).
54. LeResche L. Epidemiology of temporomandibular disorders: implications for the investigation of etiologic factors. Crit Rev Oral Biol Med. 1997;8(3):291-305. doi: 10.1177/10454411970080030401
55. LeResche L, Mancl L, Sherman JJ, Gandara B, Dworkin SF. Changes in temporomandibular pain and other symptoms across the menstrual cycle. Pain. 2003;106(3):253-261. doi: 10.1016/j.pain.2003.06.001
56. Raphael KG, Marbach JJ, Klausner J. Myofascial face pain. Clinical characteristics of those with regional vs. widespread pain. J Am Dent Assoc. 2000;131(2):161-171. doi: 10.14219/jada.archive.2000.0143
57. Puri V, Cui L, Liverman CS, Roby KF, Klein RM, Welch KM, et al. Ovarian steroids regulate neuropeptides in the trigeminal ganglion. Neuropeptides. 2005;39(4):409-417. doi: 10.1016/j.npep.2005.04.002
58. Gerwin RD. Myofascial Trigger Point Pain Syndromes. Semin Neurol. 2016;36(5):469-473. doi: 10.1055/s-0036-1586262
59. Mueller D, Obermann M, Yoon MS, Poitz F, Hansen N, Slomke MA, et al. Prevalence of trigeminal neuralgia and persistent idiopathic facial pain: a population-based study. Cephalalgia. 2011;31(15):1542-1548. doi: 10.1177/0333102411424619
60. Koopman JS, Dieleman JP, Huygen FJ, de Mos M, Martin CG, Sturkenboom MC. Incidence of facial pain in the general population. Pain. 2009;147(1-3):122-127. doi: 10.1016/j.pain.2009.08.023
61. Maarbjerg S, Wolfram F, Heinskou TB, Rochat P, Gozalov A, Brennum J, 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
62. Ziegeler C, Brauns G, May A. Characteristics and natural disease history of persistent idiopathic facial pain, trigeminal neuralgia, and neuropathic facial pain. Headache. 2021;61(9):1441-1451. doi: 10.1111/head.14212
63. Wirz S, Wartenberg HC, Wittmann M, Baumgarten G, Knüfermann P, Korthaus T, et al. Versorgung von Patienten mit chronischem orofazialem Schmerz. Ergebnisse einer Befragung in ambulanten zahnärztlichen und Mund-Kiefer-Gesichts-Chirurgie-Einrichtungen [Managing patients with chronic orofacial pain in the outpatient departments of dental and maxillofacial surgeons. Results of a survey]. Schmerz. 2003;17(5):325-331. (In German). doi: 10.1007/s00482-003-0230-1
64. Zebenholzer K, Wöber C, Vigl M, Wessely P, Wöber-Bingöl C. Facial pain in a neurological tertiary care centre--evaluation of the International Classification of Headache Disorders. Cephalalgia. 2005;25(9):689-699. doi: 10.1111/j.1468-2982.2004.00936.x
65. Benoliel R, Birman N, Eliav E, Sharav Y. The International Classification of Headache Disorders: accurate diagnosis of orofacial pain? Cephalalgia. 2008;28(7):752-762. doi: 10.1111/j.1468-2982.2008.01586.x
66. Wirz S, Ellerkmann RK, Buecheler M, Putensen C, Nadstawek J, Wartenberg HC. Management of chronic orofacial pain: a survey of general dentists in german university hospitals. Pain Med. 2010;11(3):416-424. doi: 10.1111/j.1526-4637.2010.00805.x
67. Prakash S, Rathore C, Makwana P, Dave A. A Cross-Sectional Clinic-Based Study in Patients With Side-Locked Unilateral Headache and Facial Pain. Headache. 2016;56(7):1183-1193. doi: 10.1111/head.12842
68. Aggarwal VR, Lovell K, Peters S, Javidi H, Joughin A, Goldthorpe J. Psychosocial interventions for the management of chronic orofacial pain. Cochrane Database Syst Rev. 2011;(11):CD008456. Update in: Cochrane Database Syst Rev. 2015;(12):CD008456. doi: 10.1002/14651858.CD008456.pub2.
69. Macfarlane TV, Blinkhorn AS, Davies RM, Kincey J, Worthington HV. Association between female hormonal factors and oro-facial pain: study in the community. Pain. 2002;97(1-2):5-10. doi: 10.1016/s0304-3959(01)00396-7
70. Galli U, Ettlin DA, Palla S, Ehlert U, Gaab J. Do illness perceptions predict pain-related disability and mood in chronic orofacial pain patients? A 6-month follow-up study. Eur J Pain. 2010;14(5):550-558. doi: 10.1016/j.ejpain.2009.08.011
71. Sipilä K, Ylöstalo PV, Ek E, Zitting P, Knuuttila ML. Association between optimism and self-reported facial pain. Acta Odontol Scand. 2006;64(3):177-182. doi: 10.1080/00016350500514816
72. Madland G, Newton-John T, Feinmann C. Chronic idiopathic orofacial pain: I: What is the evidence base? Br Dent J. 2001;191:22–24. doi: 10.1038/sj.bdj.4801081
73. Aggarwal VR, Macfarlane GJ, Farragher TM, McBeth J. Risk factors for onset of chronic oro-facial pain--results of the North Cheshire oro-facial pain prospective population study. Pain. 2010;149(2):354-359. doi: 10.1016/j.pain.2010.02.040
74. Vickers ER, Cousins MJ, Woodhouse A. Pain description and severity of chronic orofacial pain conditions. Aust Dent J. 1998;43(6):403-409. doi: 10.1111/j.1834-7819.1998.tb00200.x
75. Breivik H, Borchgrevink PC, Allen SM, Rosseland LA, Romundstad L, Hals EK, et al. Assessment of pain. Br J Anaesth. 2008;101(1):17-24. doi: 10.1093/bja/aen103
76. Agostoni E, Frigerio R, Santoro P. Atypical facial pain: clinical considerations and differential diagnosis. Neurol Sci. 2005;26 Suppl 2:s71-4. doi: 10.1007/s10072-005-0412-y
77. Galhardoni R, Ciampi de Andrade D, Puerta MY, Brunoni AR, Varotto BL, de Siqueira JT, et al. Altered cortical excitability in persistent idiopathic facial pain. Cephalalgia. 2019;39(2):219-228. doi: 10.1177/0333102418780426
78. Jääskeläinen SK, Forssell H, Tenovuo O. Electrophysiological testing of the trigeminofacial system: aid in the diagnosis of atypical facial pain. Pain. 1999;80(1-2):191-200. doi: 10.1016/s0304-3959(98)00203-6
79. Didier H, Marchetti C, Borromeo G, Tullo V, Bussone G, Santoro F. Persistent idiopathic facial pain: multidisciplinary approach and assumption of comorbidity. Neurol Sci. 2010;31 Suppl 1:S189-95. doi: 10.1007/s10072-010-0324-3
80. Juniper RP, Glynn CJ. Association between paroxysmal trigeminal neuralgia and atypical facial pain. Br J Oral Maxillofac Surg. 1999;37(6):444-447. doi: 10.1054/bjom.1999.0135
81. Sarlani E, Schwartz AH, Greenspan JD, Grace EG. Facial pain as first manifestation of lung cancer: a case of lung cancer-related cluster headache and a review of the literature. J Orofac Pain. 2003;17(3):262-267. PMID: 14558496.
82. Agbaje J, De Laat A, Constantinus P, Svensson P, Baad-Hansen L. Agreement between quantitative and qualitative sensory testing of changes in oro-facial somatosensory sensitivity. J Oral Rehabil. 2017;44(1):30-42. doi: 10.1111/joor.12455
83. Teerijoki-Oksa T, Forssell H, Jääskeläinen SK. Validation of diagnostic methods for traumatic sensory neuropathy and neuropathic pain. Muscle Nerve. 2019;59(3):342-347. doi: 10.1002/mus.26400
84. Dyck PJ. Evaluative procedures to detect, characterize, and assess the severity of diabetic neuropathy. Diabet Med. 1991;8:S48-51. doi: 10.1111/j.1464-5491.1991.tb02156.x
85. Jääskeläinen SK, Teerijoki-Oksa T, Forssell H. Neurophysiologic and quantitative sensory testing in the diagnosis of trigeminal neuropathy and neuropathic pain. Pain. 2005;117(3):349-357. doi: 10.1016/j.pain.2005.06.028
86. Baad-Hansen L, Abrahamsen R, Zachariae R, List T, Svensson P. Somatosensory sensitivity in patients with persistent idiopathic orofacial pain is associated with pain relief from hypnosis and relaxation. Clin J Pain. 2013;29(6):518-526. doi: 10.1097/AJP.0b013e318268e4e7
87. Benoliel R, Eliav E, Elishoov H, Sharav Y. Diagnosis and treatment of persistent pain after trauma to the head and neck. J Oral Maxillofac Surg. 1994;52(11):1138-1147; discussion 1147-1148. doi: 10.1016/0278-2391(94)90530-4
88. Sullivan MJL, Bishop SR, Pivik J. The Pain Catastrophizing Scale: Development and validation. Psychol. Assessment. 1995;7(4):524-532. doi:10.1037/1040-3590.7.4.524
89. Whyte A, Boeddinghaus R, Matias MATJ. Diagnostic Imaging Principles and Applications in Head and Neck Pathology. In: Farah C, Balasubramaniam R, McCullough M, eds. Contemporary Oral Medicine. Springer, Cham; 2017. doi: 10.1007/978-3-319-28100-1_6-1
90. Hirata K, Nakaura T, Okuaki T, Kidoh M, Oda S, Utsunomiya D, et al. Comparison of the image quality of turbo spin echo- and echo-planar diffusion-weighted images of the oral cavity. Medicine (Baltimore). 2018;97(19):e0447. doi: 10.1097/MD.0000000000010447
91. Whyte A, Matias MATJ. Imaging of orofacial pain. J Oral Pathol Med. 2020;49(6):490-498. doi: 10.1111/jop.13063
92. Baliazina EV, Evusyak OM, Kharitonova KP. Place of hypovitaminosis D in the formation of chronic pain syndrome in patients with persistent idiopathic facial pain. Russian Journal of Pain. 2020;18(2):9‑13. (In Russ.) doi: 10.17116/pain2020180219
93. Schweiger V, Nocini R, De Santis D, Procacci P, Zanette G, Secchettin E, et al. Persistent Idiopathic Facial Pain (PIFP) in Patients Referred to a Multidisciplinary Centre in Italy: A Retrospective Observational Study. J Clin Med. 2022;11(13):3821. doi: 10.3390/jcm11133821
94. Forssell H, Jääskeläinen S, List T, Svensson P, Baad-Hansen L. 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
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Balyazina E.V., Zykova O.M., Afanasyeva A.V., Suleymanova L.Z. Persistent idiopathic facial pain. South Russian Journal of Therapeutic Practice. 2025;6(4):51-60. (In Russ.) https://doi.org/10.21886/2712-8156-2025-6-4-51-60
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