ABSTRACT
Introduction
Debilitating facial pain can seriously affect an individual’s daily living. Given that the pathophysiology behind neuropathic and myofascial pain is not fully understood, when chronic facial pain goes undiagnosed, it has been proposed that one of the two is the likely cause. Since their discovery, glossopharyngeal neuralgia (GN) and Eagle’s syndrome have been considered mostly conditions afflicting the adult population. However, when pediatric patients present with symptoms resembling GN or Eagle’s syndrome, physicians are less apt to include these as a differential diagnosis simply due to the low prevalence and incidence in the pediatric population.
Materials and methods
A literature review was performed with the aim to better understand the history of reported cases and to provide a comprehensive report of the anatomical variations that lead to these two conditions as well as the way these variations dictated medical and surgical management. Articles were obtained through Google Scholar and PubMed. Search criteria included key phrases such as pediatric glossopharyngeal neuralgia and pediatric Eagle syndrome. These key phrases were searched independently. PubMed was searched primarily then cross-referenced articles were found via Google Scholar. Results from non-English articles were excluded.
Results
A total of 58 articles were reviewed. Most of the articles focused on adult glossopharyngeal neuralgia, and the majority was comprised of case reports. When searched via PubMed, a total of 16 articles and 2 articles returned for glossopharyngeal neuralgia and Eagle’s syndrome, respectively. After criteria selection and cross-referencing, a total of seven articles were found with respect to pediatric glossopharyngeal neuralgia.
Conclusions
While they are rare conditions, there are multiple etiologies that lead to the debilitating symptoms of GN and Eagle’s syndrome. The clinical anatomy proved notable as multiple causes of GN and Eagle’s syndrome are due to variation in the anatomy of the neurovascular structures surrounding the glossopharyngeal nerve, an elongated styloid process, a calcified stylohyoid ligament as well as a calcified stylomandibular ligament. Due to the success of different treatment modalities, the treatment of choice is dependent on clinical judgment.
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Abbreviations
- GN:
-
Glossopharyngeal neuralgia
- TN:
-
Trigeminal neuralgia
- PICA:
-
Posterior inferior cerebellar artery
References
(IHS) HCCotIHS (2013) The International Classification of Headache Disorders, 3rd edition (beta version). Cephalalgia 33:629–808. https://doi.org/10.1177/0333102413485658
Baddour H, McAnear J, Tilson H (1978) Eagle’s syndrome. Report of a case. Oral Surg Oral Med Oral Pathol 46:486–494
Bitner DP, Uzbelger Feldman D, Axx K, Albandar JM (2015) Description and evaluation of an intraoral cervical plexus anesthetic technique. Clin Anat 28:608–613. https://doi.org/10.1002/ca.22543
Buch HA (2017) Lingual nerve: in search of a landmark for its position. Clin Anat 30:1009–1012. https://doi.org/10.1002/ca.22921
Cesmebasi A, Spinner RJ (2015) An anatomic-based approach to the iatrogenic spinal accessory nerve injury in the posterior cervical triangle: how to avoid and treat it. Clin Anat 28:761–766. https://doi.org/10.1002/ca.22555
Childs AM, Meaney JF, Ferrie CD, Holland PC (2000) Neurovascular compression of the trigeminal and glossopharyngeal nerve: three case reports. Arch Dis Child 82:311–315
Davies JC, Ravichandiran M, Agur AM, Fattah A (2016) Evaluation of clinically relevant landmarks of the marginal mandibular branch of the facial nerve: a three-dimensional study with application to avoiding facial nerve palsy. Clin Anat 29:151–156. https://doi.org/10.1002/ca.22570
De Ridder D, Møller A, Verlooy J, Cornelissen M, De Ridder L (2002) Is the root entry/exit zone important in microvascular compression syndromes? Neurosurgery 51:427–433 discussion 433-424
Dubey A, Mujoo S, Sakarde SB, Dubey AK (2012) Paroxysmal neuralgia in pediatric population--a diagnostic dilemma for physicians and dental practioners. Kathmandu Univ Med J (KUMJ) 10:74–77
Flory T, Chung J, Ozner J (2015) Resolution of facial neuralgia following reduction of atlas subluxation complex: a case study. J Upper Cervical Chiro:6–13
Golden AS, Haut SR, Moshé SL (2006) Nonepileptic uses of antiepileptic drugs in children and adolescents. Pediatr Neurol 34:421–432. https://doi.org/10.1016/j.pediatrneurol.2005.08.017
Gracco A, De Stefani A, Bruno G, Balasso P, Alessandri-Bonetti G, Stellini E (2017) Elongated styloid process evaluation on digital panoramic radiograph in a North Italian population. J Clin Exp Dent 9:e400–e404. https://doi.org/10.4317/jced.53450
Grazzi L, Usai S, Rigamonti A (2005) Facial pain in children and adolescents. Neurol Sci 26(Suppl 2):s101–s103. https://doi.org/10.1007/s10072-005-0419-4
Harris W (1921) Persistent pain in lesions of the peripheral and central nervous system. Br Med J 2:896–900
Heir GM, Masterson M (2016) Bilateral glossopharyngeal neuropathy following chemo and radiation therapy for a primitive neuroectodermal tumour. J Oral Rehabil 43:154–158. https://doi.org/10.1111/joor.12347
Hodnett BL, Schmitt NC, Clayburgh DR, Burkowsky A, Balzer J, Thirumala PD, Duvvuri U (2015) Superior laryngeal nerve monitoring using laryngeal surface electrodes and intraoperative neurophysiological monitoring during thyroidectomy. Clin Anat 28:460–466. https://doi.org/10.1002/ca.22487
Holloway M, Wason S, Willging J, Myer CR, Wood B (1991) Radiological case of the month. A pediatric case of Eagle’s syndrome. Am J Dis Child 3:339–340. doi:https://doi.org/10.1001/archpedi.1991.02160030109031, 145
Iwanaga J (2017) The clinical view for dissection of the lingual nerve with application to minimizing iatrogenic injury. Clin Anat 30:467–469. https://doi.org/10.1002/ca.22877
Iwanaga J, Saga T, Tabira Y, Nakamura M, Kitashima S, Watanabe K, Kusukawa J, Yamaki K (2015) The clinical anatomy of accessory mental nerves and foramina. Clin Anat 28:848–856. https://doi.org/10.1002/ca.22597
Iwanaga J, Watanabe K, Henry B, Tomaszewski KA, Walocha JA, Oskouian RJ, Tubbs RS (2017) Anatomical study of the internal nasal branch of the infraorbital nerve: application to minimizing nerve damage with surgery in and around the nose. Clin Anat 30:817–820. https://doi.org/10.1002/ca.22927
Iwanaga J, Watanabe K, Kusukawa J, Fisahn C, Alonso F, Oskouian RJ, Tubbs RS (2017) A novel treatment for keratitis sicca (dry eye): anatomical feasibility study. Clin Anat 30:839–843. https://doi.org/10.1002/ca.22946
Iwanaga J, Watanabe K, Oskouian RJ, Tubbs RS (2017) Previously undescribed palpebral branch from the infraorbital canal: application to surgery of the eyelid and treatment of orbital floor fractures. Clin Anat 30:835–838. https://doi.org/10.1002/ca.22940
Iwanaga J, Watanabe K, Saga T, Tabira Y, Kitashima S, Kusukawa J, Yamaki K (2016) Accessory mental foramina and nerves: application to periodontal, periapical, and implant surgery. Clin Anat 29:493–501. https://doi.org/10.1002/ca.22635
Joo W, Rhoton AL (2015) Microsurgical anatomy of the trochlear nerve. Clin Anat 28:857–864. https://doi.org/10.1002/ca.22602
Kandt RS, Daniel FL (1986) Glossopharyngeal neuralgia in a child. A diagnostic and therapeutic dilemma. Arch Neurol 43:301–302
Katusic S, Williams DB, Beard CM, Bergstralh EJ, Kurland LT (1991) Epidemiology and clinical features of idiopathic trigeminal neuralgia and glossopharyngeal neuralgia: similarities and differences, Rochester, Minnesota, 1945-1984. Neuroepidemiology 10:276–281. https://doi.org/10.1159/000110284
Kehrer A, Engelmann S, Bauer R, Taeger C, Grechenig S, Kehrer M, Prantl L, Tamm ER, Bleys RLAW, Mandlik V (2018) The nerve supply of zygomaticus major: variability and distinguishing zygomatic from buccal facial nerve branches. Clin Anat 31:560–565. https://doi.org/10.1002/ca.23044
Kent D, Rath T, Snyderman C (2015) Conventional and 3-dimensional computerized tomography in Eagle’s syndrome, glossopharyngeal neuralgia, and asymptomatic controls. Otolaryngol Head Neck Surg 153:41–47. https://doi.org/10.1177/0194599815583047
Kondev L, Minster A (2003) Headache and facial pain in children and adolescents. Otolaryngol Clin N Am 36:1153–1170
Lindeman P (1985) The elongated styloid process as a cause of throat discomfort. Four case reports. J Laryngol Otol 99:505–508
Ling XY, Smoll NR (2016) A systematic review of variations of the recurrent laryngeal nerve. Clin Anat 29:104–110. https://doi.org/10.1002/ca.22613
Lobón MJ, Bautista F, Riet F, Dhermain F, Canale S, Dufour C, Blauwblomme T, Zerah M, Beccaria K, Saint-Rose C, Puget S, Carrie C, Lartigau E, Bondiau PY, Valteau-Couanet D, Grill J, Bolle S (2016) Re-irradiation of recurrent pediatric ependymoma: modalities and outcomes: a twenty-year survey. Springerplus 5:879. https://doi.org/10.1186/s40064-016-2562-1
López-Elizalde R, Campero A, Sánchez-Delgadillo T, Lemus-Rodríguez Y, López-González MI, Godínez-Rubí M (2018) Anatomy of the olfactory nerve: a comprehensive review with cadaveric dissection. Clin Anat 31:109–117. https://doi.org/10.1002/ca.23003
McLaughlin MR, Jannetta PJ, Clyde BL, Subach BR, Comey CH, Resnick DK (1999) Microvascular decompression of cranial nerves: lessons learned after 4400 operations. J Neurosurg 90:1–8. https://doi.org/10.3171/jns.1999.90.1.0001
Moffat DA, Ramsden RT, Shaw HJ (1977) The styloid process syndrome: aetiological factors and surgical management. J Laryngol Otol 91:279–294
Monsour PA, Young WG (1986) Variability of the styloid process and stylohyoid ligament in panoramic radiographs. Oral Surg Oral Med Oral Pathol 61:522–526
Müderris T, Bercin S, Sevil E, Beton S, Kırıs M (2014) Surgical management of elongated styloid process: intraoral or transcervical? Eur Arch Otorhinolaryngol 271:1709–1713. https://doi.org/10.1007/s00405-013-2664-0
Mueller D, Obermann M, Yoon MS, Poitz F, Hansen N, Slomke MA, Dommes P, Gizewski E, Diener HC, Katsarava Z (2011) Prevalence of trigeminal neuralgia and persistent idiopathic facial pain: a population-based study. Cephalalgia 31:1542–1548. https://doi.org/10.1177/0333102411424619
Nam YS, Kim IB, Shin SY (2017) Detailed anatomy of the abducens nerve in the lateral rectus muscle. Clin Anat 30:873–877. https://doi.org/10.1002/ca.22918
Park HK, Rha HK, Lee KJ, Chough CK, Joo W (2017) Microsurgical anatomy of the oculomotor nerve. Clin Anat 30:21–31. https://doi.org/10.1002/ca.22811
Polednak AP (2017) Relationship of the recurrent laryngeal nerve to the inferior thyroid artery: a comparison of findings from two systematic reviews. Clin Anat 30:318–321. https://doi.org/10.1002/ca.22851
Quereshy F, Gold E, Arnold J, Powers MP (2001) Eagle’s syndrome in an 11-year-old patient. J Oral Maxillofac Surg 59:94–97. https://doi.org/10.1053/joms.2001.19302
Resnick DK, Jannetta PJ, Bissonnette D, Jho HD, Lanzino G (1995) Microvascular decompression for glossopharyngeal neuralgia. Neurosurgery 36:64–68 discussion 68–69
Restrepo CE, Tubbs RS, Spinner RJ (2015) Expanding what is known of the anatomy of the spinal accessory nerve. Clin Anat 28:467–471. https://doi.org/10.1002/ca.22492
Rushton JG, Stevens JC, Miller RH (1981) Glossopharyngeal (vagoglossopharyngeal) neuralgia: a study of 217 cases. Arch Neurol 38:201–205
Sharma N, Ram R, Kamal R (2016) Unusually elongated styloid process: a report of two cases with literature review. Ann Maxillofac Surg 6:297–299. https://doi.org/10.4103/2231-0746.200343
Slavin KV (2002) Eagle syndrome: entrapment of the glossopharyngeal nerve? Case report and review of the literature. J Neurosurg 97:216–218. https://doi.org/10.3171/jns.2002.97.1.0216
Spina A, Boari N, Gagliardi F, Bailo M, Morselli C, Iannaccone S, Mortini P (2017) The emerging role of gamma knife radiosurgery in the management of glossopharyngeal neuralgia. Neurosurg Rev. https://doi.org/10.1007/s10143-017-0886-0
Tseng SC, Cheng AM, Fu Y (2017) Retrospective study to identify trigeminal-cervical ocular referred pain as a new causative entity of ocular pain. J Pain Res 10:1747–1754. https://doi.org/10.2147/JPR.S140895
Tubbs RS, Loukas M, Dixon J, Cohen-Gadol AA (2010) Compression of the cervical internal carotid artery by the stylopharyngeus muscle: an anatomical study with potential clinical significance. Laboratory investigation. J Neurosurg 113:881–884. https://doi.org/10.3171/2010.1.JNS091407
Tubbs RS, Mortazavi MM, Loukas M, Shoja MM, Cohen-Gadol AA (2011) Intraoperative and anatomical descriptions of intracranial connections between the glossopharyngeal and vagus nerves: clinical implications. J Neurosurg 115:179–181. https://doi.org/10.3171/2011.2.JNS101757
Vanelderen P, Lataster A, Levy R, Mekhail N, van Kleef M, Van Zundert J (2010) 8. Occipital neuralgia. Pain Pract 10:137–144. https://doi.org/10.1111/j.1533-2500.2009.00355.x
Walco GA, Dworkin RH, Krane EJ, LeBel AA, Treede RD (2010) Neuropathic pain in children: special considerations. Mayo Clin Proc 85:S33–S41. https://doi.org/10.4065/mcp.2009.0647
Wong K, Opimo AB, Olch AJ, All S, Waxer JF, Clark D, Cheng J, Chlebik A, Erdreich-Epstein A, Krieger MD, Tamrazi B, Dhall G, Finlay JL, Chang EL (2016) Re-irradiation of recurrent pineal germ cell tumors with radiosurgery: report of two cases and review of literature. Cureus 8:e585. https://doi.org/10.7759/cureus.585
Yglesias A, Narbona J, Vanaclocha V, Artieda J (1996) Chiari type I malformation, glossopharyngeal neuralgia and central sleep apnoea in a child. Dev Med Child Neurol 38:1126–1130
Yomo S, Arkha Y, Donnet A, Régis J (2009) Gamma knife surgery for glossopharyngeal neuralgia. J Neurosurg 110:559–563. https://doi.org/10.3171/2008.8.17641
Yoshihara N, Okuda M, Takano K, Wada T, Osaka H (2012) Idiopathic cranial polyneuropathy with unilateral IX and X and contralateral XI nerve palsy in a 4-year-old boy. Pediatr Neurol 47:198–200. https://doi.org/10.1016/j.pediatrneurol.2012.04.021
Zeiler FA, Janik MK, McDonald PJ, Kaufmann AM, Fewer D, Butler J, Schroeder G, West M (2016) Gamma knife radiosurgery for pediatric arteriovenous malformations: a Canadian experience. Can J Neurol Sci 43:82–86. https://doi.org/10.1017/cjn.2015.267
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Shereen, R., Gardner, B., Altafulla, J. et al. Pediatric glossopharyngeal neuralgia: a comprehensive review. Childs Nerv Syst 35, 395–402 (2019). https://doi.org/10.1007/s00381-018-3995-3
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DOI: https://doi.org/10.1007/s00381-018-3995-3